Squamous cell carcinoma (SCC) of the anal canal is a rare condition comprising only 2–4% of all cancers of the colon, rectum and anus. The most common sites of metastases are liver and lung.
We report a case of 34-year-old male, who presented with diarrhoeal illness and an acutely tender protruding anal lesion initially thought to be thrombosed external hemorrhoid. Examination under anaesthesia revealed a hard and fixed mass at the level of dentate line, which was biopsied. On histopathological examination, it was confirmed as primary moderately differentiated keratinising squamous cell carcinoma. Despite initial response to concomitant chemo-radiation, 4 months later, the patient presented with large bowel obstruction. On laparotomy, a large mass involving the terminal ileum and caecum was identified. Histopathological examination revealed metastatic spread from primary anal lesion. To the best of our knowledge, this is the first reported case of a squamous cell carcinoma of anus with bowel metastasis.

INTRODUCTION

Anal tumours are uncommon tumours of the gastrointestinal tract that constitute only 5% of anorectal malignancies with the peak incidence seen during the seventh decade of life. (1) Nearly 80% of anal canal tumours are squamous cell carcinomas (SCC). Anal cancer is primarily a loco-regional disease, which rarely (<10% of cases) metastasises. (2) Most common sites of extra-pelvic metastases from squamous cell cancer of anal canal are the liver, lungs and extra-pelvic lymph nodes, although spread to peritoneum, bone and other sites may also occur. (3) We herein report an unusual case of SCC of anus with small and large bowel metastases in a young male presented with large intestinal obstruction four months after the primary diagnosis.

CASE REPORT

A 34-year-old white heterosexual male presented with a 3-week history of diarrhoeal illness and an acutely tender protruding anal lesion after evacuation thought to be a thrombosed external hemorrhoid at left lateral position on initial examination.

Examination under general anaesthesia revealed a hard and fixed mass at the level of dentate line protruding down into anal canal and two peri-anal abscesses along with bilateral palpable inguinal lymphadenopathy. Incision and drainage of the abscesses was performed and biopsy was taken from anal lesion and anal verge. Histopathological analysis revealed primary moderately differentiated keratinising SCC with no evidence of vascular invasion.

Staging MRI scan showed large tumour protruding through anal canal involving left levator ani muscle and left seminal vesicle and enlarged bilateral pelvic lymph nodes with no evidence of liver metastasis. Subsequently, de-functioning colostomy was done and patient received chemotherapy with an initial cycle of Cisplatin and Fluorouracil, followed by combination of Fluorouracil, Mitomycin and radiotherapy. His tumour responded well and showed considerable reduction in size on subsequent imaging.

Fig 1: Squamous cell carcinoma metastasis infiltrating peritoneal fat and serosa evident of right hemicolectomy specimen

Despite an initial response to treatment, four months later, the patient presented with clinical picture suggestive of large bowel obstruction. An explorative laparotomy unveiled a large mass involving the terminal ileum and caecum along with dense adhesions of ileocaecal loops. A 4 cm nodule in the right lobe of liver was also discovered. The patient underwent right hemicolectomy with an end-to-end ileocolic anastomosis. The surgical specimen consisted of 21 and 15 cm length of small and large bowel, respectively. The central 10 cm of the bowel was of undeterminable nature due to its tortuous nature caused by innumerable adhesions. The mucosa was unremarkable except for focal oedema. Histological examination showed poorly-differentiated squamous cell carcinoma infiltrating serosa with prominent intravascular spread. The tumour was predominantly confined to the peritoneal fat and serosa with no obvious invasion of the muscularis, submucosal, and mucosal layers (Figure 1 and 2). The appearance was consistent with metastatic spread from primary anal lesion. Unfortunately, the patient did not recover and died from multiple organ failure on the fourth post-operative day.

Fig 2: Keratin pearls confirming squamous cell carcinoma metastasis in the tumour specimen

DISCUSSION

Metastatic tumours to the large bowel are rare and can pose diagnostic and management difficulties. In small bowel, metastatic tumours outnumber the primary tumours but it is rarely involved by metastasis from a tumour originating outside the peritoneal cavity. (4)

The malignancies, known to cause secondary deposits in large bowel, are stomach, breast, ovary, kidney, bladder, prostate, lung, cervix, and melanoma. (4) The common primary tumours that metastasise to small bowel are carcinoma of lung, cervix uteri, melanoma, other parts of the gastro-intestinal tract (stomach and colon), and kidney. (5,6) In the present case, the metastasis was from a moderately-differentiated SCC of the anus which has never been reported in the literature. The possibility of a primary SCC of the bowel was excluded since the tumour was located in serosa with no evidence of dysplasia or atypia in intestinal epithelium and staging MRI did not show any evidence of primary bowel tumour.

The route of metastasis to the ileum depends upon the site of the primary lesion. The different mechanisms postulated for small bowel metastasis from abdomen or pelvis include; retrograde lymphatic spread following initial blockade of para-aortic or mediastinal lymph nodes, peritoneal seedlings, direct extension by continuity or by permeation of the lymphatic spaces in connective tissues and haematogenous route. (7) In our case, haematogenous route appeared to be the most likely mode of spread due to the presence of liver metastasis and wide spread vascular invasion on histopathology.

The most common presenting clinical picture of metastatic small bowel tumour is that of partial or complete small bowel obstruction. Less often, the patient may present with perforation of the bowel or haemorrhage from the lesion. The causes of malignant bowel obstruction include extrinsic or intrinsic occlusion of the lumen and intestinal motility disorders. (8) In this case, malignant bowel obstruction can be explained by the extensive adhesions in the intestinal wall seen intra-operatively and caused by metastasis.

Even though management of metastatic tumor to bowel is only palliative, considerable relief can be obtained by resection of obstructed bowel and therefore, in the absence of overwhelming systemic disease, surgical intervention is still an option. (9) Karanikas et al, (10) in their study on 31 patients with metastases to large bowel concluded that clinically-evident metastases to large bowel confer a poor prognosis but surgical resection can, in selected patients, produce a worthwhile survival.

To sum up, our case report reveals the possibility of intestinal obstruction caused by metastasis from primary SCC of the anus, a scenario which has not been published in the literature before.

REFERENCES

1. Boman BM, Moertel CG, O’Cornell MJ, Scott M, Weiland LH, Beart RW, et al. Carcinoma of the anal canal: a clinical and pathological study of 188 cases. Cancer 1984; 54:114-25
2. Klas JV, Rothenberger DA, Wong WD, Madoff RD. Malignant tumours of the anal canal: the spectrum of disease, treatment and outcomes. Cancer 1999; 85(8):1686-93
3. Cummings BJ. Metastatic anal cancer: the search for cure. Onkologie 2006; 29(1-2):5-6
4. John AK, Kotru A, Pearson HJ. Colonic metastasis from bronchogenic carcinoma presenting as pancolitis. J Postgrad Med 2002; 48(3):199-200
5. Arulraj P, Damodaran V, Raman ML, Nagarajan V, Tulasi NR. Small bowel metastases from esophageal and oropharyngeal cancers. Indian J Gastroenterol 2005; 24(3):116-8
6. Mathur SK, Pandya GP. Solitary metastatic malignant stricture of the ileum: a rare cause of small bowel obstruction (a case report). J Postgrad Med 1984; 30(3):186-8
7. Neve RS, Qureshi SS, Mistry RC. Ileal metastases from oesophageal carcinoma causing intestinal obstruction. J Postgrad Med 2005; 51(1):74-5
8. Kelvin JF, Scagliola J. Metastases involving the gastrointestinal system. Seminars in Oncology Nursing 1998; 14 (3):187-98
9. Wong TH, Tan YM. Surgery for the palliation of intestinal obstruction in advanced abdominal malignancy. Singapore Med J 2009; 50(12):1139-44
10. Karanikas ID, Hartley JE, Walton AP, McDonald AW, Lee PWR, Duthie GS, Monson JRT. Non-primary malignancies of the colon and rectum. Brit J of Surg 1997; 84(2):32

Desmoid tumours are a rare entity and are usually detected with a certain delay, as they initially often grow without causing any specific clinical symptoms. Although these tumours grow slowly and do not metastasise, their growth pattern remains aggressive and local tissue displacement or even destruction is common. Surgery remains the only potentially curative treatment option. Here we report on the findings of a 43-year old female patient with a large intra-abdominal desmoid tumour, resulting in the displacement of most intra-abdominal organs to the right of the patient’s abdominal cavity. She successfully underwent surgical removal of the mass and recovered without complications.

INTRODUCTION

Desmoid tumours comprise 0.03% of all neoplasms, less than 3% of all soft tissue tumours (1), with an incidence of 2-4 cases / 1’000’000 per year (2). They are usually detected relatively early on in life (10-40 years of age), with a slight female predominance. While normally occurring sporadically, they are more common in patients with familial adenomatous polyposis (FAP), where the incidence is 850 times higher than in the general population (3).

Most desmoid tumours typically tend to grow slowly and do not metastasise, although their growth pattern remains quite aggressive, with organ infiltration, local tissue destruction and organ compression/displacement. The tumours often grow without causing any clinical symptoms and are therefore usually only detected relatively late or during a check-up for another ailment, when they may have already reached substantial sizes of 15cm in diameter or more. Problems associated with intestinal, vascular or neural compression are quite common.

CASE REPORT

During a routine check-up for hypertension, the 43-year old female patient’s general practitioner noted a painless mass in the abdomen. The patient reported increased fatigue and a reduced overall performance for the previous two years. Further questioning revealed occasional dyspepsia and an increased urge to urinate, although micturition was otherwise without any associated discomfort. She was otherwise in good general health and without previous abdominal surgery. Routine haematology and biochemistry were normal. An abdominal computed tomography revealed a very large, well-defined intra-abdominal mass, resulting in the displacement of most intra-abdominal organs to the right of the patient’s abdominal cavity (Fig. 1).

Fig 1: The abdominal CT scan reveals a large tumour, resulting in the displacement of the intra-abdominal organs to the right of the abdominal cavity.

Radiological evaluation did not make it possible to tell from which organ the tumour originated. Intra-operative findings revealed a tumour spanning from the symphysis to the xyphoid (Fig. 2) and showed multiple adherences between the omentum and the caudal pole of the spleen. In order to achieve an R0-resection, a partial splenectomy and partial removal of the omentum was necessary. The tumour weighed 5.6kg and measured 28cm.

Fig 2: Intra-operative aspect. The extensive tumour takes up nearly the entire intra-abdominal space.

DISCUSSION

Diagnostic imaging includes abdominal sonography, computed tomography or magnetic resonance imaging (MRI). MRI usually is the examination of choice as it provides the surgeon with a better evaluation of exact tumour size, location and infiltration in to the surrounding tissues with tumour recurrences being more easily detected. Upon diagnosis of a desmoid tumour all patients should have a colonoscopy and a gastroscopy to exclude FAP.

Histologically, desmoid tumours are benign and characterised by a monoclonal fibroblastic proliferation typically arising from muscular or aponeurotic structures. Although upon initial inspection an intact capsule seems to surround the periphery, the tumour characteristically extends beyond this pseudo-capsule. Tumours are classified as intra- or extra-abdominal, with extra-abdominal tumours most often located around the neck and shoulders, the pelvis, or within the thoracic wall. Sporadic desmoid tumours are mainly located extra-abdominal and are relatively easy to resect.  High recurrence rates and difficult locations make treatment of the intra-abdominal sub-group particularly difficult.

Treatment of desmoid tumours is challenging and the optimal therapeutic strategy remains a matter of debate. While spontaneous tumour regression has been observed, usually in menopausal women or post- oophorectomy, suggesting a certain estrogen-dependency (4,5), rapid growing tumours require an aggressive therapy if cure is to be sought. Their high recurrence rate and their ability and tendency to infiltrate vital organs and structures can result in difficult and highly complex operations. Treatment for small, non-infiltrating tumours can be delayed in completely asymptomatic patients and / or in patients unfit for surgery, as the tumours can remain stable over years without any significant morbidity.  However, these patients require routine radiological imaging to exclude rapid growth or new organ infiltration.

Surgery remains the main therapeutic option, with complete tumour resection providing the only potentially curative treatment. Although, as with classical cancer surgery, the aim is to achieve an R0 resection margin, surgery should nevertheless be kept to the required minimum, avoiding any mutilating intervention. If resected early on, the removal of abdominal-wall desmoid tumours is associated with a low morbidity and mortality.  Overall recurrence rates lie by 20% to 75% (6,7) and are more frequent for the rarer extra-abdominal tumours. Surgical interventions for intra-abdominal tumours are often associated with a high mortality and morbidity. Many are found at the root of the small bowel mesenterium and up to 65% of the tumours are unresectable upon intraoperative inspection (8).

There is little evidence for the benefit of pharmacological therapies, which include the use of non-steroidal anti-inflammatory drugs, such as sulindac or anti-estrogens such as tamoxifen (9). The aim is tumour regression / growth stop and it can be used as a first-line treatment for multiple, intra-abdominal tumours. The high recurrence rate can be reduced by adjuvant and / or neoadjuvant radiotherapy (10) both for abdominal wall and extra-abdominal tumours. The usefulness of alternative therapies, such as radio frequency ablation is currently under scrutiny. Other options include placement of a percutaneous catheter for use in chemical ablation using acids. The possibility of gene transfer is also subject to intense research.

Desmoid tumours are rare and are either found sporadically or more commonly associated with FAP. A classification into abdominal wall tumours as well as extra- and intra-abdominal tumours is important with regard to further treatment and outcome. Clear guidelines outlining possible therapy options – including helpful algorithms – are missing. However, symptomatic tumours, especially if located intra-abdominal, should be treated aggressively. Surgery still remains the treatment of choice, although recurrence rates are high, even after R0 resection.

REFERENCES

1. Nuyttens JJ, Rust PF, Thomas CR, Jr., Turrisi AT, 3rd. Surgery versus radiation therapy for patients with aggressive fibromatosis or desmoid tumors: A comparative review of 22 articles. Cancer. 2000 Apr 1;88(7):1517-23
2. Papagelopoulos PJ, Mavrogenis AF, Mitsiokapa EA, Papaparaskeva KT, Galanis EC, Soucacos PN. Current trends in the management of extra-abdominal desmoid tumours. World J Surg Oncol. 2006;4:21
3. Gurbuz AK, Giardiello FM, Petersen GM, Krush AJ, Offerhaus GJ, Booker SV, et al. Desmoid tumours in familial adenomatous polyposis. Gut. 1994 Mar; 35(3):377-81
4. Easter DW, Halasz NA. Recent trends in the management of desmoid tumors. Summary of 19 cases and review of the literature. Ann Surg. 1989 Dec;210(6):765-9
5. Reitamo JJ, Scheinin TM, Hayry P. The desmoid syndrome. New aspects in the cause, pathogenesis and treatment of the desmoid tumor. Am J Surg. 1986 Feb;151(2):230-7
6. Heiskanen I, Jarvinen HJ. Occurrence of desmoid tumours in familial adenomatous polyposis and results of treatment. Int J Colorectal Dis. 1996;11(4):157-62
7. Clark SK, Neale KF, Landgrebe JC, Phillips RK. Desmoid tumours complicating familial adenomatous polyposis. Br J Surg. 1999 Sep;86(9):1185-9
8. Dozois EJ. Desmoid disease. In: Kelly KA, editor. Mayo Clinic gastrointestinal Surgery Philadpelphia: Saunders; 2004. p.563-5
9. Hansmann A, Adolph C, Vogel T, Unger A, Moeslein G. High-dose tamoxifen and sulindac as first-line treatment for desmoid tumors. Cancer. 2004 Feb 1;100(3):612-20
10. Tsukada K, Church JM, Jagelman DG, Fazio VW, Lavery IC. Systemic cytotoxic chemotherapy and radiation therapy for desmoid in familial adenomatous polyposis. Dis Colon Rectum. 1991 Dec;34(12):1090-2

Bowel perforation can be potentially fatal. We describe the case of a 42-year-old male who presented with severe abdominal pain following anal fisting. Clinical examination revealed tenderness of the complete abdomen with signs of peritonism. A CT-scan with rectal contrast showed a perforation of the sigmoid 40 cm above the anus. At laparoscopy, a perforation in the sigmoid colon was found and successfully repaired. Patient recovered uneventful and was discharged in 5 days. We present a unique case of a sigmoid perforation after anal fisting which was laparoscopically repaired without formation of a protective colostomy.

INTRODUCTION

In men having sex with men high risk sexual behavior can be seen. Often with concomitant use of recreational drugs such as ecstasy, cocaine, ketamine, and poppers. (1,2) Activities include fisting and unprotected anal intercourse. Also transanal introduction of foreign bodies is a well known phenomenon.

Bowel perforation as a complication of such activities can be potentially fatal. The treatment of a bowel perforation is most commonly performed by laparotomy. (3) Procedures which can be performed in such patients are suturing of the perforation with or without formation of a protective colostomy, a Hartmann procedure or bowel resection with primary anastomosis. (3)

Bowel perforation can also be caused as a complication of a colonoscopy. The risk of perforation after colonoscopy is small, around 0.12%. (4)  The retrieval of upper gastro-intestinal foreign bodies and bowel perforation after colonoscopy can be managed by laparotomy or laparoscopy (5,6), although complex complications due to foreign bodies in the rectosigmoid are usually managed by laparotomy. (1) Here we present a case of a laparoscopic repair of a sigmoid perforation due to a foreign body, in this case a human fist after anal fisting.

CASE REPORT

A 42-year-old HIV-infected man presented himself to our emergency department with severe abdominal pain five hours following anal fisting. Clinical examination showed an agitated male with no tachycardia, hypotension or fever. Physical examination of his abdomen revealed tenderness of the complete abdomen with signs of peritonism. A plain abdominal radiograph was reported to have normal findings. Laboratory results showed no abnormalities. Based on his clinical condition, a CT-scan with rectal contrast of the abdomen was performed. The CT-scan showed a perforation of the sigmoid 40 cm above the anus. Figure 1 shows rectal contrast throughout the complete abdomen as high as the diaphragm.

Fig 1: CT abdomen (coronal) with rectal contrast:1. Liver, 2. Contrast in the sub hepatic region, 3. Contrast in the left abdomen, 4. Sigmoid with extra luminal contrast

Under general anesthesia patient underwent a laparoscopy in which a defect of the sigmoid was found with only minimal signs of local peritonitis. The defect had a diameter of 9 centimeters and was located approximately 20 cm above the transverse rectal fold (figure 2). Because of the absence of general fecal peritonitis we opted to suture the perforation in two layers with a Vicryl 2.0, without formation of a protective ileostomy. Patient made an uneventful recovery and was discharged in 5 days.

Fig 2: Laparoscopy: defect of the sigmoid:1. Large defect of the Sigmoid, 2. Sigmoid mesocolon, 3. Abdominal wall

Patient was frequently reviewed in our outpatient department. At the time of the last telephone contact with the patient he had made a complete recovery without any sequelae of his abdominal surgery.

DISCUSSION

A large bowel perforation can be potential fatal due to the development of peritonitis by contamination of the peritoneal cavity. This can lead to a septic shock, which requires immediate surgery. There is a high risk of mortality and morbidity in patients with a septic shock due to peritonitis caused by bowel perforation. (7) Most common causes of colon perforation which lead to peritonitis are colorectal cancer and complications of colonic diverticulitis representing between 64% and 100% of cases with peritonitis due to different etiologies. (8)  In literature the incidence of iatrogenic colon perforation after colonoscopy, which also can lead to peritonitis, has been described between 0.06 % and 0.1 %. (9)

Laparotomy was always considered to be  the first choice to manage colon perforation due to its advantages such as adequate localization of the perforation, closure or repair of the defect and the possibility for peritoneal lavage. A more recent study showed that outcomes in laparoscopic repair of colonic perforation are the same as in open surgery. (5)

In our patient, we performed a successful laparoscopic repair of his sigmoid perforation. Because of the short time (7 hours) between the trauma and surgery we decided to only suture the large defect without a protective ileostomy. Laparoscopy has certain advantages compared to open surgery in post-operative outcomes. Less paralytic ileus, faster return to normal bowel function, less pain, lavage of the peritoneum and a shorter duration of hospital stay. (10)

We present a unique case of a sigmoid perforation after anal fisting, which was laparoscopically repaired without formation of a protective colostomy. Laparoscopic repair of traumatic colon perforation, for example after colonoscopy, can be performed in experienced surgeons.

REFERENCES

1. Arora S, Ashrafian H, Smock ED, Ng P. Total laparoscopic repair of sigmoid foreign body perforation. J Laparoendosc Adv Surg Tech A 2009;19:401-403
2. Lee SJ, Galanter M, Dermatis H, McDowell D. Circuit parties and patterns of drug use in a subset of gay men. J Addict Dis 2003;22:47-60
3. Rodriguez-Hermosa JI, Codina-Cazador A, Sirvent JM, Martin A, Girones J, Garsot E. Surgically treated perforations of the gastrointestinal tract caused by ingested foreign bodies. Colorectal Dis 2008;10:701-707
4. Luning TH, Keemers-Gels ME, Barendregt WB, Tan AC, Rosman C. Colonoscopic perforations: a review of 30,366 patients. Surg Endosc 2007;21:994-997
5. Hansen AJ, Tessier DJ, Anderson ML, Schlinkert RT. Laparoscopic repair of colonoscopic perforations: indications and guidelines. J Gastrointest Surg 2007;11:655-659
6. Linos D, Nestoridis E. Laparoscopic removal of an ingested foreign body and repair of colonic perforation. Gastrointest Endosc 2000;52:424-426
7. Nespoli A, Ravizzini C, Trivella M, Segala M. The choice of surgical procedure for peritonitis due to colonic perforation. Arch Surg 1993;128:814-818
8. Bielecki K, Kaminski P, Klukowski M. Large bowel perforation: morbidity and mortality. Tech Coloproctol 2002;6:177-182
9. Mai CM, Wen CC, Wen SH, Hsu KF, Wu CC, Jao SW, Hsiao CW. Iatrogenic colonic perforation by colonoscopy: a fatal complication for patients with a high anesthetic risk. Int J Colorectal Dis 2010;25:449-454
10. Degiuli M, Mineccia M, Bertone A, Arrigoni A, Pennazio M, Spandre M, Cavallero M, Calvo F. Outcome of laparoscopic colorectal resection. Surg Endosc 2004;18:427-432

Acalculous cholecystitis is a relatively rare form of cholecystitis appearing in severely ill patients. We chose the case of a young patient under chemotherapy for myeloid leukaemia who developed a severe septic shock secondary to an alithiasic cholecystitis. Because of hemodynamic instability needing high-dose of vasoactive amines, percutaneous gallbladder drainage was done. After this intervention, the septic shock could be controlled, but a bleeding liver laceration was observed, fortunately without morbidity consequences.

Abdominal infections are life-threatening complications in neutropenic patients. Neutropenic enterocolitis is the most important entity, but the acute cholecystitis, even rarer, had been described in several reports, suggesting that this infection could represent a difficult trap.

INTRODUCTION

Patients with leukaemia undergoing aggressive chemotherapy are often affected by abdominal infections becoming life-threatening conditions. It is a diagnostic challenge for the physician to detect the cause of abdominal symptoms (infectious and non-infectious) in these patients (1,2).

Acalculous cholecystitis is a form of acute cholecystitis seen more frequently in immunosuppressed and high risk patients. The treatment of choice for acute cholecystitis is surgical, but in selected cases conservative treatment followed by elective cholecystectomy later or cholecystostomy could represent an alternative (2,3).

CASE REPORT

We report the case of a 36-year-old man with the diagnosis of acute myeloid leukemia type M4 (>90% blasts). The patient was treated firstly by cytoreduction chemotherapy followed by induction chemotherapy (cytarabine + idarubicine). During this period the patient was admitted in hospital with diarrhoea and febrile agranulocytosis. Neutropenic colitis was diagnosed and confirmed by CT scan, the patient being treated with broad spectrum antibiotics leading to a good recovery.

Fig 1: Computed tomography scan demonstrating gallbladder wall thickening and pericholecystic liquid, without gallbladder lithiasis, confirmed by ultrasonography.

One month later the first consolidation chemotherapy (cytarabine + amsacrine) was done and 6 days afterwards the patient presented diarrhoea without abdominal pain or fever. The laboratory investigations revealed a total leukocyte count of 500/mm³, a haemoglobin level of 68 g/L and a platelet count of 11 000/mm³. Despite supportive treatment and antibiotics for a supposed improving colitis, the patient complained of right upper quadrant abdominal pain, high fever (40°C) and hypotension needing ICU admission with the diagnose of abdominal sepsis. A positive Murphy’s sign was observed at physical abdominal examination. The radiological imaging was suggestive for an alithiasic cholecystitis, even though a transverse colon wall thickening was also seen in computed tomography.

Fig 2: Computed tomography scan showing peritoneal bleeding from hepatic laceration after gallbladder percutaneous transhepatic drainage.

The patient was considered to unstable for a surgical intervention and an emergency percutaneous CT-guided cholecystostomy was done, in spite of a very low platelet count (7 000/mm³). 24 hours later, the septic shock rapidly improved, but hemoglobin level fell down from 83 to 61 g/L. A CT scan showed a bleeding liver laceration with intraperitoneal haemorrhage secondary to the transhepatic percutaneous gallbladder drainage.

The patient was treated conservatively receiving several transfusions and went well without any further complications.

DISCUSSION

Alithiasic cholecystitis is an entity occurring typical in the elderly, severely ill and high risk patients typically hospitalised in the ICU. The frequency of acute cholecystitis between patients with myelosupressive chemotherapy and neutropenia is estimated at 0.4% in the literature, with a nearly 30% mortality. The majority of the neutropenic patients develop acalculous cholecystitis, in contrast with non-neutropenic patients to whom acalculous cholecystitis comprises only approximately 5% of all cases of acute cholecystitis (1).

The clinical diagnosis of acute cholecystitis (AC) remains difficult in the patient population with multiple comorbid conditions, the typical signs and symptoms being easily masked among ventilated, paralyzed or immunosuppressed patients. Ultrasound is an established valuable imaging method if cholecystitis is suspected and a thickened gallbladder wall greater than 3mm is considered typical for diagnosis. Other frequent sonographic findings are pericholecystic fluid and over-distension. Similar findings can be demonstrated by CT, computed tomography being extremely useful in the assessment of septic shock of unknown origin in this patient population (1,4).

Laparoscopic cholecystectomy is widely considered as the treatment of choice for acute cholecystitis and it can be performed safely in selected patients. Surgery in patients with neutropenia, thrombopenia and sepsis who present with an acute abdomen is however associated with a high mortality. Percutaneous cholecystostomy (PC) was described as a minimally invasive, image-guided procedure and has become an alternative treatment strategy for high-risk patients with acute calculous or acalculous cholecystitis who are unfit for urgent surgery (1,5).

Percutaneous cholecystostomy could be also used as a method of exclusion of the gallbladder/biliary tree as a source of sepsis in unstable severely ill patients. The positive response is defined as resolution of right upper quadrant abdominal pain and normalization of general clinical state, usually within 48 hours (4).

The procedure can be performed CT-guided or US-guided, even at the bedside in the ICU. The safety of the procedure and its minimal invasiveness made it a valid treatment option for a patient not responding to antibiotic therapy or being in severe shock and unfit for immediate cholecystectomy. The results from several series prove that the percutaneous gallbladder drainage is safe and that it was effective in more than 85% of the patients. There were no bile duct injuries or any other major surgical complications in this group of patients (6,7).

REFERENCES

1. Gorschlüter M et al. Cholecystitis in neutropenic patients: Retrospective study and systematic review. Leukemia Research 30 (2006) 521–528
2. Hatzidakis AA et al. Acute cholecystitis in high-risk patients: percutaneous cholecystostomy vs conservative treatment. Eur Radiol (2002) 12(7):1778-84
3. Tsuyuguchi T. Techniques of biliary drainage for acute cholecystitis: Tokyo Guidelines J Hepatobiliary Pancreat Surg (2007) 14:46–51
4. Beardsley SL et al. Predicting Infected Bile among patients undergoing percutaneous cholecystostomy. Cardiovasc Intervent Radiol (2005) 28:319–325
5. Silberfein EJ et al. Percutaneous cholecystostomy for acute cholecystitis in high-risk patients: experience of a surgeon-initiated interventional program. The American Journal of Surgery 194 (2007) 672–677
6. Spira RM, Nissan A, Zamir O, Cohen T, Fields SI, Freund HR. Percutaneous transhepatic cholecystostomy and delayed laparoscopic cholecystectomy in critically ill patients with acute calculus cholecystitis. The American Journal of Surgery 183 (2002) 62–66
7. Barie PS, Eachempati SR Acute acalculous cholecystitis. Curr Gastroenterol Rep. 2003 Aug;5(4):302-9

Primary gallbladder carcinoma is a malignant neoplasm with an incidence of 1.2 cases per 100 000 people each year. Its occurrence increases with age, particularly in women. It is the fifth most common cancer of the gastrointestinal tract, and the most common malignancy in the biliary tract. Adenocarcinoma is the most common type of gallbladder neoplasm whereas sarcomatoid carcinoma is rare with an incidence of less than one percent of all malignant gall bladder neoplasms. Sarcomatoid carcinomas are composed of malignant epithelial and sarcomatous components, sometimes with heterologous sarcomatous elements. In general, conservative treatments are thought to be unfavourable to the tumor. Prognosis is poor following curative resection for carcinosarcoma of the gall bladder because of recurrence as systemic metastasis of the liver and peritoneal dissemination.

INTRODUCTION

Primary gallbladder carcinoma is a malignant neoplasm with an incidence of 1.2 cases per 100 000 people each year. Its occurrence increases with age, particularly in women (1). It is the fifth most common cancer of the gastrointestinal tract, and the most common malignancy in the biliary tract (2). Adenocarcinoma is the most common type of gallbladder neoplasm whereas sarcomatoid carcinoma is rare with an incidence of less than one percent of all malignant gall bladder neoplasms (1). Sarcomatoid carcinomas are composed of malignant epithelial and sarcomatous components, sometimes with heterologous sarcomatous elements (3). Therapeutic methods of the tumor include surgery, chemotherapy, radiotherapy, and medical treatment. If possible, however, the patients with this carcinoma are usually treated by either radical or palliative surgery. In general, conservative treatments are thought to be unfavourable to the tumor (4). To our knowledge, only 78 cases with sarcomatoid carcinoma of the gallbladder have been reported in the English-language literature (5,6).

CASE REPORT

A 55-year-old woman presented with a 12 month history of intermittent right upper quadrant pain, anorexia, and weight loss. A physical examination revealed a non-tender and hard mass ~ 5×4 cm in the right upper quadrant of her abdomen. Laboratory examinations showed a normal leukocyte count, normal haemoglobin levels and liver function tests as well as a normal urine analysis. The serum levels of carcinoembryonic antigen and carbohydrate antigen 19-9 were within the normal reference range. Her family history and medical histories were unremarkable. Ultrasound Abdomen showed a 5-cm irregularly shaped heterogeneous mass in the gallbladder.  On Computed tomography, a partially dilated gall bladder with multiple calculi and heterogeneously enhancing solid soft tissue density mass in the region of fundus with mild IHBR dilatation was seen. Liver and adjacent organs were all normal (Figure 1A).

The patient underwent cholecystectomy & wedge resection of the liver with resection of transverse colon and paraduodenal lymph nodes. Intra-operatively, a hard mass in the fundal region of gall bladder, adherent to the transverse colon was seen. Grossly, the tumor measured 7×5x3 cm and was solid grey-white (Figure 1B). The tumor was adherent to the colon but not infiltrating the wall of colon or underlying liver. Microscopic sections showed features of sarcomatoid carcinoma with focal squamous differentiation (Figure 2). The tumor cells were immunopositive for cytokeratin and vimentin. The tumor infiltrated into the adventitia of colon, however muscularis propria and mucosa were free of tumor. The underlying liver parenchyma, resected margins of liver, intestine, cystic duct and eleven lymphnodes dissected from specimen were free of tumor. Paraduodenal lymph nodes were also free of tumor.

Fig 1: A. CECT Abdomen shows soft tissue density in the region of fundus; B. Cut surface of the resected specimen showing a large variegated tumor in gall bladder

Two months after surgery, the patient visited the hospital because of a mild fever and tenderness on the operation site. A follow computed tomographic scan showed multiple intrahepatic metastases and omental seedlings. She could not receive any treatment for cancer because of her poor general medical condition.

Fig 2: A. H&E stained section shows presence of both malignant epithelial (squamous) and mesenchymal (spindle) components (X100); B. Tumor at higher magnification (X200); C. Mesenchymal (spindle) component of tumor (X100)

DISCUSSION

Adenocarcinoma is the most common type of gallbladder neoplasm whereas sarcomatoid carcinoma is rare with an incidence of less than one percent of all malignant gall bladder neoplasms (1). It has been described that sarcomatoid carcinoma arises from totipotent stromal stem cells and is composed of epithelial and mesenchymal components that contain undifferentiated spindle or stellate cells (7). The cancer was first reported by Landsteiner in 1907, and until now, only 78 cases have been reported in the English-language literature, with mean age range from 66.5-72 years (5,6). Similar to adenocarcinoma, gallbladder sarcomatoid carcinoma usually present with abdominal pain, jaundice, nausea, and poor oral intake, and some may present with a palpable abdominal mass and weight loss. Liver function tests are normal, except in patients with obstructive jaundice caused by tumor invasion of the biliary tract (8). Carcinosarcoma of the gall bladder is not associated with specific radiological findings or serum data, including tumor markers (carcinoembryonic antigen, carbohydrate antigen 19-9, or squamous cell carcinoma antigen). Carcinosarcoma of the gall bladder should be considered as a differential diagnosis of neoplasms of the gall bladder, especially when patients present with severe abdominal symptoms and/or a large tumor size (9). Race, tumor size, genetic variation, earlier detection, and more extensive surgery may be contributing factors for better prognosis. While gallstone, epithelial and mesenchymal component types, age and sex are of little prognostic value (5).

Surgery is suggested as the only recognized treatment for gallbladder sarcomatoid carcinoma. Previous studies reported the use of chemoradiotherapy did not significantly improve patient prognosis (4,8). Exploration of new radiation techniques and of chemotherapeutic regimens with new drugs is required for the treatment of carcinosarcoma of the gall bladder because conventional chemotherapy and radiotherapy do not increase patient survival. Novel ‘molecularly targeted’ agents may improve surgical outcome. In a meta-analysis by Zhang L et al reported the mean survival of CSGB as 17.5 months, and the median was 5 months with a 1year survival rate of 19±5% and 5-year of 16±5% (5). The longest survivor was reported in 2002, who had survived 5 years and 7 months post-operatively (10). Therefore, in comparison with the reported 0-10% 5-year survivals of adenocarcinoma, Sarcomatous carcinoma of gallbladder has a similar but slightly better prognosis (5).

REFERENCES

1. Albores-Saavedra J, Cruz-Ortiz H, Alcantara-Vazques A, Henson DE. Unusual types of gallbladder carcinoma. A report of 16 cases. Arch Pathol Lab Med 1981; 105:287-293
2. Parkin DM, Bray F, Ferlay J, Pisani P. Global cancer statistics, 2002. CA Cancer J Clin 2005; 55:74-108
3. Mehrotra TN, Gupta SC, Naithani YP. Carcino-sarcoma of the gall bladder. J Pathol 1971; 104:145-148
4. Liu KH, Yeh TS, Hwang TL, Jan YY, Chen MF. Surgical management of gallbladder sarcomatoid carcinoma. World J Gastroenterol 2009; 15:1876-1879
5. Zhang L, Chen Z, Fukuma M, Lee LY, Wu M. Prognostic Significance of Race and Tumor Size in Carcinosarcoma of Gallbladder: a Meta-Analysis of 68 Cases. Int J Clin Exp Pathol. 2008; 1(1): 75–83
6. Hu ZH, Li ZW, Shen L, Zhang M, Zheng SS. Surgical therapy and prognosis of sarcomatoid carcinoma of the gallbladder. Hepatobiliary Pancreat Dis Int 2010; 9: 175-179
7. Chao TC, Wang CS, Jeng LB, Jan YY, Chen MF. Primary carcinoma of the gallbladder in Taiwan. J Surg Oncol 1996; 61: 49-55
8. Huguet KL, Hughes CB, Hewitt WR. Gallbladder carcinosarcoma: a case report and literature review. J Gastrointest Surg 2005; 9: 818-821
9. Okabayashi T, Sun ZL, Montgomey RA, Hanazaki K. Surgical outcome of carcinosarcoma of gall bladder: A review. World J Gastroenterol. 2009; 15(39): 4877-82
10. Nimura Y. Extended surgery in bilio-pancreatic cancer: The Japanese experience. Semin Oncol. 2002; 29:17–22

Teratomas are the benign tumours, which may occur anywhere in the body. Development of these lesions in the oral cavity is extremely rare. In the oral cavity, they usually arise in the midline, in the floor of mouth. Infrequently, they may be seen in the tongue proper. We hereby, present a case of swelling tongue in 56 years female diagnosed as teratoma.

INTRODUCTION

Teratomas are benign neoplasms composed of all the three germinal layers. Around 80% are located in the ovaries and sacral lesions, 7% are seen in head and neck region, only approximate 1.6% of these tumours are found in oral region. Pure oral presentation in the tongue is extremely rare. Only a few number of cases have been reported in the literature so  far (1).

CASE REPORT

A 56 years old female presented with swelling on the dorsal surface of tongue. Examination revealed a firm, rubbery, non-tender 3×2x2cm sessile swelling with no induration or ulceration (fig 1).

Fig 1: Swelling on surface of tongue

She noticed this swelling 6 months previously, which more recently increased in size. Lately, she experienced difficulty in moving tongue, resulting in globbus sensation and dysphagia. General examination of the patient revealed average built, pulse rate 80 beats/min with regular rhythm, BP 130/90 mm Hg, RR 24 breaths/min. There was no history of fever, night sweats and weight loss. Jaundice, cyanosis and oedema were absent. Computed tomography revealed a 3×2 cm cystic anterior lingual structure, the wall of which was thin and regular with a content consisting of homogenous fluid. There was no bone involvement. With a clinical diagnosis of dermoid cyst an excision biopsy was performed. Histopathological findings consisted of cyst wall lining of stratified squamous epithelium with sebaceous glands, blood vessels, muscle and cartilage in the underlying connective tissue, and a diagnosis of teratoma was made (fig 2). No evidence of malignant transformation was noted. One year after surgical removal of the lesion, there was no sign of recurrence.

Fig 2: Histological specimen

DISCUSSION

The tongue is derived from two separate embryologic origins. The anterior two thirds is derived from ectoderm and posterior one third is from endoderm. The anterior two third s originate from paired lateral lingual swellings, which are contributed by first branchial arch. These swellings fuse in the midline to form the tuberculum impar. The posterior one third of the tongue arises from the hypobranchial eminence, which is made up of mesoderm of the second, third and a portion of the fourth pharyngeal arches. Congenital dermoid cysts arise from epithelial rests trapped during midline fusion of these branchial arches whereas acquired dermoid cyst arise from epithelium implanted during trauma and they occur at the sites away from midline. The terms teratoma, teratoid cyst and dermoid cyst have been used interchangeably to describe a wide variety of lesions by some authors. Meyer has classified these cyst as epidermoid, dermoid and teratoid. Epidermoid cysts are lined with simple squamous epithelium and surrounding connective tissue. Dermoid type cysts contain skin appendages whereas teratoids contain epithelium lined mesodermal or endodermal elements such as bone, teeth, muscle, mucous membrane (2).

Teratomas of the oral cavity are divided anatomically depending on their location. They can be sublingual, geniohyoid and lateral (3). The other differentials which are encountered at these sites are ranula, lymphangioma, angioma and lipoma (1). Teratoma is a tumour, which contains disorderly arranged tissues and organs. There is an epithelial lined cavity containing mesodermal as well as endodermal derivatives like muscle, intestinal mucosa, respiratory mucosa, fibres, bone and blood vessels etc. Teratoma of tongue may exhibit skin, hair, bone, cartilage or mucous membrane on the surface (7). The rarity of teratoma has been stemmed from the fact that it is not located along embryonic fusion line and it does not involve the floor of the mouth (4). Teratoma in the head and neck region are rare, comprising 1-10% of cases. Very few numbers of cases have been reported so far. They probably arise from totiopotent embryonic tissue that has been displaced during ontogeny (8).

A good patho-radiological correlation is required to confirm the diagnosis. Ultrasonography establishes the presence of solid and cystic components and can differentiate cyst from surrounding tissue. By far MRI has been proven to be superior among imaging modalities, as it can locate exact position, extention and demarcations of the lesion (1). Because of their avascular character, teratoma do not enhance with administration of contrast material and thus can cause diagnostic confusion with choriostoma, endodermal sinus tumours and granular cell tumours. Because oral teratomas are well defined, complete excision is usually possible. Recurrences are very rarely seen in head and neck teratomas. Most of the times, these tumours are benign but may result in high degree of mortality and morbidity due to variations in their size and location. If large enough, they may cause airway obstruction, respiratory distress, dysphagia, difficult in eating and pain due mostly to infection in the lesion. In malignant teratoma radio-chemotherapy is used after surgical removal of the tumour.(5) Alphafetoprotein( AFP) has been shown to be reliable indicator of disease activity and some authors advocate investigating teratoma recurrence by doing serial serum AFP levels. They have been shown to increase in teratocarcinoma (6). Though teratoma has been reported in infants so far it is unusual with the site not being the midline. Malignant change was not seen. Patient responded better after complete surgical excision of the lesion. Though rare, teratoma should be considered in the differential diagnosis of tongue masses.

REFERENCES

1. Fuchshuber S, Grevers G, Issing W I. Dermoid cyst of the floor of the mouth- a case report. Eur Arch Otorhinolaryngol 2002;259:60-2
2. Myssiorek D, Lee J, Wasserman P, Lustrin E. Intralingualdermoid cysts: A report of two new cases. Ear Nose Throat Journal 2000;79:380-3
3. Gold BD, Sheinkopf DE, Levy B. Dermoid, epidermoid, and teratomatous cysts of the tongue and the floor of the mouth. J Oral Surgery 1974;32:107-11
4. Goldberg AF. Dermoid cyst of the tongue: report of case. J Oral Surg, Oral Med, Oral Pathol 1980:50;217-18
5. Gupta M, Chaudhary N, Rai AK. Teratoma tongue: case report and review of literature. Indian J. Otolaryngol, Head Neck Surg 2007:59;160-2
6. Azizkhan RG, Haase GM, Applebaum H, Dillon PW, Coran AG, King PA etal. Diagnosis, management, and outcome of cervicofacial teratoma in neonates: a childrens cancer group study. J Paed Surg 1995:30;312-16
7. Miller AP, Owens JB. Teratoma of the tongue. Cancer 1996:19;1583
8. Batsakis JG, Pathology consultation. Nomenclature of developmental tumours. Ann OtolRhinolLaryngol 1984;93:98-99

Gastro-colic fistulas as a complication of benign gastric ulcers are relatively uncommon. The authors report a 59 year old caucasian female with a known hiatus hernia and gastro-oesophageal reflux disease presenting with diarrhoea and weight loss. Barium enema is the investigation of choice although it is important to exclude malignancy. Management is by en-bloc resection of the fistula with primary gastro-intestinal reconstruction.

INTRODUCTION

Gastro-colic fistula is a rare cause of vomiting, diarrhoea and weight loss and one that must be considered if the more common differential diagnoses such as gastroenteritis are excluded. There are numerous causes of gastro-colic fistulas but those caused by benign gastric ulceration are a rarity. The investigation of choice is a barium enema. Other investigations to exclude malignancy must be carried out. The optimal intervention is surgical with en-bloc resection of the fistula with primary gastro-intestinal reconstruction.

CASE PRESENTATION

A 59 year old female presented to the Emergency Department with a two week history of diarrhoea and weight loss. The diarrhoea occurred more after meals and the patient had lost 2-3 stone over the last 2 weeks.

She was known to have a hiatus hernia and a gastric ulcer that was diagnosed a month previously and was being actively treated for pyloric stenosis by oesophagogastroduodenoscopy and dilatation.

On examination she was apyrexial and looked cachectic. Systemic examination was non-specific and did not reveal and pathological findings.

Laboratory findings revealed marked dehydration but erect chest x-ray and abdominal x-ray was unremarkable. Stool culture failed to reveal any infective cause of this diarrhoea.

The patient underwent further investigation commencing with an oeasophagogastroduodenoscopy which demonstrated the hiatus hernia, gastritis and a gastric ulcer as well as pyloric scarring and deformity. A biopsy sample was taken showing features consistent with non-specific active chronic gastritis. No cause for the patient’s symptoms was established.

She then had a barium meal and follow through which clearly demonstrated a gastro-colic fistula. This was confirmed by colonoscopy which demonstrated the fistula. There were no signs of neoplasia and the no malignancy was witnessed on biopsy. These findings were confirmed on CT.

Fig 1: A series of radiographs from barium swallow demonstrating gastro-colic fistula

Fig 2: A series of radiographs from barium swallow demonstrating gastro-colic fistula

The patient underwent a laparotomy and resection of the gastro-colic fistula. The transverse colon was mobilised and a segment of the colon resected with the fistula. Partial gastrectomy was carried out en-bloc with the fistula. A Bilroth I gastro-duodenal anastamosis was undertaken with two layers of PDS. Colo-colic anastamosis was performed in a single layer of PDS. A feeding jejunostomy and a drain in the sub-hepatic space were placed before closure of the abdomen.

Fig 3: A series of radiographs from barium swallow demonstrating gastro-colic fistula

Fig 4: A series of radiographs from barium swallow demonstrating gastro-colic fistula

DISCUSSION

Gastro-colic fistulas are a direct communication between the stomach and the colon and are a relatively rare condition. More common causes are invasive carcinomas of the stomach or colon, post-operatively, secondary to Crohns disease, diverticular disease and intra-abdominal abscesses and due to infections such as tuberculosis or syphyllis (1). Gastro-colic fistula secondary to benign gastric ulcer disease is uncommon with only 111 reported cases in English literature (2). Most patients with benign gastro-colic fistula were middle-age women with a history of recent anti-inflammatory drug therapy (3).

The literature reports patients with gastro-colic fistula presenting with a syndrome of severe diarrhoea, weight loss, anaemia and feculent vomiting. This is a result of colonic contents regurgitating into the stomach and entering the small bowel, leading to a derangement of digestive and absorptive functions of the small bowel (4). Patients may give a history of peptic ulcer disease. Majority of patients will be malnourished and dehydrated at time of presentation. Occult or frank gastrointestinal bleeding occurs in 25% to 33% of those with benign disease (3).

The patient will often have derangement of their electrolytes. Biochemical test will be suggestive of vitamin deficiencies and decrease serum protein. Haematologically, there may be anaemia and a slight leucocytosis. If a nasogastric tube to passed, aspiration of feculent material may hint towards the diagnosis (1).

Barium studies are the investigation of choice in diagnosing gastro-colic fistula (1). Thoeny, Hodgson and Scudamore, in the 1960, reported a 95% success rate with barium enema. There have been reports of computer tomography to delineate the fistula and identify underlying pathology (5,6). Gastroscopy and colonoscopy are less sensitive in establishing diagnosis but are important in obtaining multiple biopsies to excluding malignant disease (7).

As majority of these patients are malnourished at the time of presentation, nutritional support via IV hyperalimentation is an important adjunct (1). There have been reports in the literature of effective treatment gastro-colic fistula secondary to benign gastric ulcer with antacid medication. In these cases, healing time varied between 2 to 16 weeks with an average of 9 weeks (3).

Surgical intervention is traditionally the treatment of choice by en bloc resection and primary reconstruction (1).

REFERENCES

1. Cody JH, DiVincenti FC, Cowick DR, Mahanes JR: Gastrocoli and gastrojejunocoli fistulae: report of twelve cases and review of the literature. Ann Surg 1975, 181(3):376-80
2. Shaik AS, Singh B, Haffejee AA: Gastrocolic fistula as a complication of benign gastric ulcer. SAMJ 1999, 89(9):1011-1014
3. Soybel DI, Kestenberg A, Brunt EM, Becker JM: Gastrocolic fistula as a complication of benign gastric ulcer: report of four cases and update of the literature. J Am Osteopath Assoc. 1978, 77:684-8
4. Lee WJ, Horton KM, Fishman EK: Gastro-colic fistula due to adenocarcinoma of the colon: simulation of primary gastric leiomyosarcoma on CT. Clin Imaging 1999, 23:295-297
5. Kiskaddon, RM, Templeton FE, Renshaw RJF: Gastro-colic Fistula: A New concept of Pathologic Physiology; Mechanism of Production of the Syndrome. Cleveland Clin. Quart. 1947, 14:94
6. Matsuo S, Eto T, Ohara O, Miyazaki J, Tsunoda T, Kanematsu T: Gastrocolic fistula originating from transverse colon cancer: report of a case and review of the Japanese literature. Surg Today 1994, 241085-1089
7. Thoeny RH, Hodgson JR, Scudamore HH: The Roentgenologic Diagnosis of Gastrocolic and Gastrojejunocolic Fistulas. Am. J. Roentgenol. 1960, 83:876

A 71 year old lady was treated for a squamous cell carcinoma of the oesophagus with neo-adjuvant chemotherapy followed by a two phase Ivor-Lewis oesophagectomy with two field lymphadenectomy. She presented four years later with life threatening bleeding from a fistula between the thoracic aorta and the gastric conduit, which was treated successfully with a thoracic aortic stent.

INTRODUCTION

Fistulation from aorta to gastric conduit after Ivor-Lewis oesophagectomy is a rare and life threatening condition. Reported cases have occurred early post operatively, with a mean post-operative day of presentation of 46 in a review of 23 cases. The majority of these patients presented with a herald bleed (93%) (1).

Various aetiologies have been hypothesised. Early post operative leakage from the suture line of the anastamosis, erosion of the gastric conduit by acidic gastric contents and reduced blood supply to the conduit have been suggested as potential causative factors (2,3). Thoracic lymphadenectomy, whereby the descending aorta is completely skeletonised, is believed to predispose to fistulation.

In this report we present a rare case of the successful treatment of an aorto enteric fistula developing four years after oesophagectomy.

CASE REPORT

A fit and well 71 year old woman presented with a history of reduced appetite and weight loss and was found to have a squamous cell carcinoma of the oesophagus at 30cm. Staging investigations suggested a T₂N₁ cancer with no haematogenous spread. The patient was offered neo-adjuvant chemotherapy followed by oesophagectomy. She received two cycles of Cisplatin and 5-Flurouracil. Following restaging an Ivor-Lewis oesophagectomy was performed with meticulous two field thoraco-abdominal lymphadenectomy. Histopathology confirmed a complete pathological response (Mandard regression grade 1) to neo-adjuvant therapy (T₀N₀ with 0/37 nodes positive). There were no immediate post operative complications and she was discharged day 16 post op. Over the subsequent three years she was troubled by recurrent dysphagia related to benign anastamotic strictures which required multiple dilatations.

Fig 1: Acute gastric ulcer seen on endoscopy

Fig 2: Acute gastric ulcer post treatment with gold probe

An endoscopy performed four years post-operatively demonstrated two atypical acute gastric ulcers in the distal stomach (figure 1), which were treated with a heat probe (figure 2). Two days later she presented as an emergency in hypovolaemic shock following a large volume haematemesis (haemoglobin of 40g/l). An urgent upper gastrointestinal endoscopy revealed a visible vessel at 35 cm forming part of an expansive mass within the gastric conduit. EUS demonstrated a heterogeneous mass indenting the gastric conduit, containing a strong Doppler signal (figure 3). An urgent computerised tomography (CT) angiogram revealed a Type-A thoracic aortic aneurysm with an enteric fistula (figure 4). The saccular aneurysm of the thoracic aorta was embedded in the gastric conduit. A 28×16cm Valiant® (Medtronic Ltd, Minneapolis, USA) stent was placed in the descending thoracic aorta with good occlusion of the aneurysm. The patient made an excellent recovery and was discharged on the seventh day post stenting.

Fig 3: EUS examination showing thoracic aneurysm compressing esophagus

Fig 4: CT scan showing thoracic aortic aneurysm

Post operatively the patient developed a low grade stent infection requiring a prolonged course of antibiotics as an outpatient. Two years after insertion of the stent she remains well with no further gastrointestinal bleeding and follow up endoscopy has shown intact mucosa at the site of fistula. Computerised tomography scanning has demonstrated involution of the aneurysm sac and no endo-leak (figure 5).

Fig 5: Stent in thoracic aortic aneurysm

DISCUSSION

Aorta-conduit or gastric fistulae are an uncommon complication following oesophagectomy. In this case report we have discussed a patient who developed an aorto-conduit fistula more than four years after an Ivor Lewis oesophagectomy.

In the few cases that have been reported in the literature oesophago-aortic fistulae have presented on average two to three weeks post operatively. Eighty two percent (82%) of cases reported presented between the second and sixth weeks postoperatively (1). The longest duration between oesophagectomy and presentation of bleed previously reported was fourteen months (1).

Aorta-conduit fistulae tend to present with a triad of symptoms, first described by Chiari-Strassburgh in 1914, of chest pain, sentinel haemorrhage and then finally exsanguination following a massive gastro-intestinal haemorrhage (5). In a review of 23 reported cases of aorto-conduit fistula 21 patients presented with a herald bleed and only one patient survived operative management of this complication. This presenting symptom may be the only sign prior to massive gastro-intestinal haemorrhage and death. Signs of gastro-intestinal bleeding in oesophagectomy patients should raise the suspicion of an aorto-conduit fistula. The latent period between herald bleed and exsanguination has an average duration of ten hours (1).

Proposed aetiology for this complication includes early post operative leakage from the suture line of the anastamosis causing an oesophago-pleural fistula and then an aorto-oesophageal fistula (2). This would seem an unlikely cause in the case we have described as four years had passed between the occurrence of the fistula and the original oesophagectomy.

Erosion of the gastric conduit by highly acidic gastric contents may also predispose to fistula formation (3). Gastric erosion and ulceration into both the aorta and pericardium from the intra-thoracic portion of the stomach has been reported post oesophagectomy.  A reduced blood supply to the gastric conduit may predispose to gastric erosion. It is further proposed that chronic infection of these gastric ulcers may be a contributory factor. Pathological sections of aorta-oesophageal fistula have shown a granulomatous type inflammation suggestive of mycotic infection complicating ulceration (3). Indeed this may well have been the cause of the fistula in the case report we have described. An endoscopy performed two days prior to the presentation of the aorto-oesophageal fistula showed two acute atypical looking gastric ulcers.

REFERENCES

1. Molina-Navarro C, Hosking SW, Hayward SJ, Flowerdew ADS. Gastroaortic fistula as an early complication of esophagectomy. Ann Thorac Surg. 2001:72:1783-1788
2. Maguire WC, Mitchell N. Perforation of the aorta by acid gastric contents at the site of gastroesophagostomy. Surgery. 1947;22:842-4
3. Brookes VS, Stafford JL. Peptic ulceration and perforation of the stomach after oesophagectomy. Thorax. 1952;7:167-9
4. Ullmann AS, Shier KJ, Horn RC. Aortoesophageal fistula: an unusual complication of esophago-gastrostomy following resection for carcinoma of the esophagus. Can Med Assoc J. 1961;85:27-31
5. Chiari-Strassburgh H. Injury of the esophagus with perforation of the aorta produced by a foreign body. Berl Klin Wochenschr. 1914:51:7-9

Management of upper gastrointestinal anastomotic leaks is an inter-disciplinary challenge.  We present a case of late pyloroplasty leak following 3-stage oesophagectomy.  We describe a novel, combined endoscopic and fluroscopic procedure to introduce a T-tube into the anastomotic leak proving an ideal plug at the site of leak which enabled the patient to consume a normal diet and return home safely.

INTRODUCTION

Data synthesized from existing randomised controlled trials show that pyloric drainage procedures reduce the occurrence of early post-operative gastric outlet obstruction after oesophagectomy with gastric reconstruction (1,2).  Pyloromyotomy and pyloroplasty are generally considered simple procedures, however, they may be complicated by stricture, leak, and even death (3).  Because of these complications some advocate not performing an emptying procedure during oesophagectomy (3).  We report a novel approach to the management of pyloroplasty leak following 3-stage oesophagectomy.

CASE REPORT

A 61-year-old gentleman presented in January 2010 with dysphagia for solids and regurgitation.

An urgent endoscopy was performed.  It revealed a mid to distal oesophageal cancer (25-32cm).  This arose from a long segment of multifocal dysplastic Barrett’s metaplasia (22-39cm).  Full pre-operative staging investigations diagnosed a poorly differentiated mid to distal oesophageal adenocarcinoma (T3, N1, M0).

The patient was recruited to the OEO5 trial in April 2010 and underwent neo-adjuvant chemotherapy with 5-fluorouracil and cisplatin.  In May 2010 he underwent 3-stage McKeown oesophagectomy with a formal Heineke-Mikulicz pyloroplasty (2-layer, interrupted closure with omental patch).  A covering tube drain and feeding jejunostomy were placed.

Final histology of the resected specimen revealed T3, N1 (2/22), M0, poorly differentiated adenocarcinoma with microscopically involved margins (circumferential) with vascular and lymphatic invasion.

On post-operative day 7 a routine post-operative contrast swallow was performed. This identified a spill of contrast due to a 5mm anterior defect at the level of the oesophago-gastric anastomosis suggestive of focal ischaemia.  This was managed with washout and drainage and a covered oesophageal stent.  A further contrast swallow revealed contrast aggregating around the drain (Figure 1) and that the covered oesophageal stent was appropriately positioned with no evidence of on-going leak (Figure 2).

Figure 1: A contrast swallow demonstrating contrast aggregating around the drain & Figure 2: A covered oesophageal stent appropriately positioned with no evidence of leak

On post-operative day 11, at routine dressing change on the ward, a full thickness abdominal wall dehiscence was identified.  The patient proceeded to laparotomy where turbid intra-peritoneal fluid was identified.  There was no evidence of intra-peritoneal bile.  The patient was managed with washout, drainage and re-suturing of the abdominal wall over a vicryl mesh.  The original intra-abdominal tube drain was left undisturbed.

On post-operative day 16 a late pyloroplasty leak was diagnosed on contrast swallow (Figure 3).  This presented as a persistent leak of bile stained discharge from the abdominal drain.  After failure of initial conservative treatment an attempt to control the leak was made.

The defect at the pyloroplasty suture line was now visible endoscopically and a covered pyloric stent was located appropriately across the defect.  Due to proximal stent migration this was unsuccessful in controlling the leak (Figure 4).  The patient still had a controlled fistula from the pyloroplasty suture line.

Figure 3: A late pyloroplasty leak diagnosed on contrast swallow & Figure 4: A contrast swallow demonstrating proximal stent migration and evidence of on-going leak

The patient was now stable and he requested to eat and drink.  A surgical approach to the management of fistula after pyloroplasty was deemed inappropriate because of the recent surgery for intra-abdominal sepsis and the presence of an abdominal wall mesh.

A novel approach to the management of a fistula after pyloroplasty was employed.  A combined endoscopic and fluoroscopic procedure was performed where a radiologically placed snare was inserted through the abdominal drain and manipulated into the gastric tube through the defect in the pyloroplasty.  A gastroscope was used to introduce a T-tube into the gastric tube and into the snare.  The abdominal drain and snare were withdrawn and the T-tube placed appropriately, plugging the hole in the pyloroplasty, under direct vision.  A further guide wire was left in place along the tract of the tube drain and was used to deploy a pigtail drain alongside the pyloroplasty (Figures 5 and 6).

Figure 5: An Intra-operative image demonstrating the combined endoscopic and fluoroscopic procedure

Figure 6: A pictorial representation of the result of the procedure

Follow up CT, with contrast both orally and through the T-tube, showed no evidence of ongoing leak from the pyloroplasty.  The flanged T–tube satisfactorily plugged the hole through the anterior wall of the pyloroplasty, best demonstrated in the lateral decubitus position (Figures 7 and 8).

The T-tube was then capped off and the pigtail drain subsequently removed.  The patient was commenced on oral fluids and progressed to oral diet.  The patient was discharged with a capped off T-tube in situ and made a good recovery.

Figure 7: A follow-up CT demonstrating no evidence of on-going leak from the pyloroplasty & Figure 8: A follow-up CT demonstrating the flanged T-tube satisfactorily plugging the hole through the anterior wall of the pyloroplasty

DISCUSSION

Management of anastomotic leaks is an inter-disciplinary challenge (1).  Anastomotic leaks are serious complications after upper gastrointestinal surgery.  Treatment of leaks by direct suture repair, revision, patching, and application of fibrin glue to leaks have failed to gain widespread acceptance owing to a high failure rate (4).  Alternative methods of controlling leaks such as the placement of covered stents are feasible but can be complicated by stent migration (4) and inadequate drainage of associated collections.

To our knowledge this is the only reported case of management of late pyloroplasty leak following oesophagectomy with a combined endoscopic and radiological placement of a T-tube.  The T-tube provided an ideal plug at the site of leak which enabled the patient to consume a normal diet and return home safely.

REFERENCES

1. Wedemyer J, Schneider A, Manns MP, Jackobs S. Endoscopic vacuum-assisted closure of upper intestinal anastomotic leaks. Gastrointest Endoscop 2008;67(4):708-711
2. Urschel JD, Blewett CJ, Young JE, Miller JD, Bennett WF. Pyloric drainage (pyloroplasty) or no drainage in gastric reconstruction after esophagectomy: a meta-analysis of randomized controlled trials. Dig Surg 2002;19:160-164
3. Wang LS, Huang MH, Huang BS, Chien KY. Gastric substitution for resectable carcinoma of the esophagus: an analysis of 368 cases. Ann Thorac Surg 1992;53:289-294
4. Babor R, Talbot M, Tyndal A. Treatment of upper gastrointestinal leaks with a removable, covered, self-expanding metallic stent. Surg Laparosc Endosc Percutan Tech 2009;19(1):e1-4

Intraperitoneal perforation can occur as a rare but life threatening complication of indwelling urethral catheters. Computed tomography (CT) revealed the bladder perforation by the Foley catheter. The injury could not be managed conservatively due to the presence of systemic infection. We present the case of a successful laparoscopic repair of the bladder due to indwelling urethral catheter.

INTRODUCTION

Intraperitoneal perforation is a rare and serious complication of indwelling urethral catheters. Bladder perforation should be included in the differential diagnosis, especially if patients are known to have an indwelling catheter. The laparoscopic approach is the best short recovery and less traumatic treatment for simple and accessible bladder injuries.

CASE PRESENTATION

A 73-year-old woman with a medical history of Parkinson’s disease and chronic restrictive respiratory failure due to kyphoscoliosis was hospitalized for acute coronary syndrome. During hospitalisation, a bladder Foley catheter of size 16 French was placed for drainage and haemodynamic monitoring.

Forty-eight hours later, she presented with acute respiratory distress and septic shock requiring ventilatory assistance. She had no abdominal pain and no signs of peritoneal irritation. The daily diuresis was around 900 mL. The urinalysis was positive for nitrites, leukocytes and hematuria. Laboratory tests showed serum urea 10 mM/L and serum creatinine 232 µM/L. Plain radiographs of the abdomen did not reveal any signs of perforation or intestinal obstruction.

Together with vasoactive medication, intravenous antibiotics (Tazocilline and Linezolid) for nosocomial-acquired pneumonia were administrated. The patient showed no sign of haemodynamic improvement regardless of the antibiotic therapy.

Thoraco-abdomino-pelvic CT scan was then performed. The pelvic sagittal scan showed the Foley catheter perforated the urinary bladder and liquid effusion likely to be urine (Figure 1). The Foley catheter was unfortunately withdrawn to be well replaced.

Fig 1: Sagittal CT image showing urethral catheter in the abdominal cavity.

Due to systemic infection with no sign of improvement, a laparoscopic surgical exploration was decided. The patient underwent laparoscopic surgery with three trocars, a 10 mm umbilical trocar and two 5 mm trocars, one in each iliac fossa. Abdominal exploration found bowel loops of the ileum clustered at the bladder and liquid in the peritoneal cavity. Despite adhesiolysis, the perforation was not visualized. Methylene blue dye was injected through the Foley catheter and it highlighted a 1 cm perforation in the bladder dome (Figure 2) with no other macroscopic lesions. The perforated part was sutured with interrupted vicryl in one layer. A methylene blue dye test was performed and excluded a leak. Abdominal cavity lavage with 0.9 % saline was performed. A suction drain was placed into the pouch of Douglas.

Fig 2: Methylene blue dye highlighting the bladder perforation

The patient’s condition improved 24 hours after surgery allowing cessation of catecholamine and extubation. Renal function returned to normal with a good urine output from the Foley catheter. The Foley catheter was removed on day 15. The postoperative course was uncomplicated.

DISCUSSION

Laparoscopic repair of intraperitoneal bladder perforation was first described in 1994 and since then, a few cases of such approach have been reported to treat traumatic, spontaneous and iatrogenic bladder rupture, avoiding laparotomy (1). The laparoscopic approach is the best short recovery and less traumatic treatment allowing visualization of the entire peritoneal cavity to exclude others lesions.

Laparoscopy is a minimally invasive technique to treat abdominal trauma. It can avoid laparotomy in 63% of the cases, decreasing its associated morbidity (2). In the presence of simple and accessible injuries the therapeutic laparoscopy is performed.

Abdomino-pelvic CT scan is frequently performed in hospitalised patients and the bladder is included in the examination. In many cases the urinary bladder is catheterised for various indications. Familiarity with the different bladder pathologies and a routine meticulous search for them are indicated in every case. (3)

Early diagnosis and laparoscopic treatment might decrease the high mortality rate associated with this uncommon but treatable condition (4).

Aged patients with an acute abdomen might not complain of abdominal pain, and abdominal tenderness might be relatively mild (5). However, bladder perforation should be included in the differential diagnosis, especially if patients are known to have an indwelling catheter.

REFERENCES

1. Matsui Y, Ohara H, Ichioka K, Terada N, Yoshimura K, Terai A: Traumatic bladder rupture managed successfully by laparoscopic surgery. Int J Urol. 2003; 10:278-80
2. Gorecki PJ, Cottam D, Angus LD, Shaftan GW: Diagnostic and therapeutic laparoscopy for trauma: a technique of safe and systematic exploration. Surg Laparosc Endosc Percutan Tech. 2002; 12:195-8
3. Abadi S, Brook OR, Solomonov E, Fischer D. Misleading positioning of a Foley catheter balloon. British Journal of Radiology. 2006; 79:175-176
4. Figuerdo A. Laparoscopic treatment of traumatic intraperitoneal bladder rupture. Int Braz J Urol. 2007; 33:380-2
5. Bender JF. Approach to the acute abdomen. Med Clin North Am 1989; 73:1413-22

A traumatic right diaphragmatic rupture is an uncommon condition, as it occurs in 0.8-3.6% after blunt trauma. It is challenging to find the diagnosis immediately and is illustrated by the incidence of 12-66% initially missed diagnosis (1).

Most blunt traumatic diaphragm ruptures are an indication for early aggressive surgical intervention by way of thoracotomy, laparotomy, or both if it is necessary. Delayed diagnosis and treatment of diaphragm rupture is associated with increased rates of morbidity and mortality. Therefore, diagnosis of diaphragmatic injury requires a high index of suspicion. We report a case of a 37-year-old man with right diaphragmatic rupture after blunt injury 31 years prior to admission.

INTRODUCTION

A traumatic right diaphragmatic rupture as the result of blunt trauma is a rare condition. It is generally considered to be a marker of severe trauma. The overall incidence of diaphragmatic rupture after blunt trauma is 0.8-3.6% (1). Most injuries are due to vehicular-related incidents (79.5%) and falling from a height (15.9%) (2). Right-sided tears are significantly less likely than left-sided tears, 27.3% and 68.2% respectively (3). The correct diagnosis of diaphragmatic injury is initially missed in 12%-66% (1). Therefore the diagnosis of a diaphragmatic injury requires a high index of suspicion. Delayed diagnosis and treatment of diaphragm rupture is associated with increased rates of morbidity and mortality. In our case we treated a patient 31 years after a blunt trauma.

CASE REPORT

On our outpatient clinic, a 37-year-old man was presented with 31 years before a fall from a wall of 3 meters on his right side. This fall was complicated by multiple rib fractures. Next to this major event the man was involved in a moped -car accident with only his face wounded, 18 years ago. During his life he never had clinical symptoms of dyspnoea or pain until one year ago. The medical history showed nephrolithiasis in 2006.

The patient presented to us with primary complaints of progressive, stabbing pain in the upper right abdomen and right lower thorax with simultaneous periods of dyspnoea. The symptoms worsened over a year, eventually leading to shortness of breath during rest. Beside this presentation the patient had right upper abdominal pain while eating spicy food.

On admission, his vital signs were stable and colour was fair. The dyspnoea was not severe, as the patient could talk in full sentences. Pulmonary examination revealed diminished breath sounds at the basal right hemithorax and the diaphragm was determined to be higher on the right by percussion.

The diagnosis was confirmed by thoracic-abdominal X-rays and computerised tomography (CT) imaging, which revealed intra-thoracic displacement of the liver and also the gallbladder (figure 1).

Fig. 1: Thoracic-abdominal CT in lung setting. Intra-thoracic displacement of the liver with gallbladder.

An elective thoracotomy in the sixth intercostal space was performed with repairing the diaphragmatic hernia (figure 2). The liver with gallbladder was replaced in the abdominal cavity, after adhesiolysis of the lung from pleura and diaphragm, whereupon the diaphragm was closed tensionless by running technique with non-absorbable sutures. Two drains were left behind, one near the diaphragm and one in the right upper thorax. The patient recovered postoperatively well and left the hospital in 5 days.

Fig 2: Preoperative picture closing of the diaphragm

DISCUSSION

Right sided diaphragmatic rupture with consequently hepatothorax is a rare condition. Blunt traumatic diaphragm rupture is an absolute indication for early surgical intervention (4). A delay could result in increased rates of morbidity and mortality (5). In case of hepatothorax, hypovolemic shock can occur, because of possible kinking of the inferior vena cava with obstruction of the blood.

In this case there was a lateral impact on the chest, which distorted the chest wall and sheared the diaphragm. There remains doubt about the cause of delayed diagnosis and treatment of this diaphragm injury, since it could occur as the result of either delayed rupture or delayed detection. Even if the herniation did not take place initially it would result in herniation eventually, because of the significant discrepancy between the intra-thoracic pressures and higher intra-abdominal pressure. Most of the diaphragmatic ruptures occur on the left side, because the right diaphragm is congenitally stronger than the left. Moreover the liver partially protects the right side as the impact can be spread over a large area.

Pain in the upper abdomen and lower thorax, dyspnoea, cyanosis and hypotension are typical symptoms of diaphragmatic injury, but if the rupture is small the patient can remain symptom free for a long time.

The true incidence of traumatic diaphragm rupture is unknown because in 12-66% (1) of major trauma victims, the diagnosis is missed. This is particularly true for ruptures of the right hemidiaphragm. Right sided ruptures are often associated with severe injuries and greater haemodynamic instability. There is no consensus yet, on the gold standard of imaging technologies to diagnose diaphragm rupture. Based on the literature, diagnosis of diaphragmatic injury requires a high index of suspicion regardless whether you make an X-Ray or CT.

The choice of surgical approach includes thoracotomy, laparotomy, or both if it is necessary. The decision to repair the diaphragm either way is dictated by the stability of the patient and the presence of other associated organ injuries.

REFERENCES

1. Chughtai T, Ali S, Sharkey P, Lins M, Rizzoli S. Update on managing diaphragmatic rupture in blunt trauma: a review of 208 consecutive cases. Cam J Surg 2009;52:177-81
2. Boulanger BR, Milzman DP, Rosati C, et al. A comparison of right and left blunt traumatic diaphragmatic rupture. J Trauma. 1993;35:255–60
3. Gwely NN. Outcome of blunt diaphragmatic rupture. Analysis of 44 cases. Asian cardiocasc thorac ann 2010;18:240-3
4. Meyers BF, McCabe CJ. Traumatic diaphragmatic hernia:occult marker of serious injury. Ann Surg 1993;218:783-90
5. Bozla U. Late presentation of blunt right diaphragmatic rupture. American J Emerg Med. 2008;26:638

Colorectal cancer is a common type of cancer in developed countries and is an important public health problem. Patients with colorectal cancer presents in a variety of ways in different settings. Most commonly, they present in the outpatient settings with changes in bowel habits, rectal bleeding and iron deficiency anaemia. This case reports an atypical presentation of colorectal cancer and how the cancer was diagnosed and treated.

INTRODUCTION

Colorectal cancer is the third most common cancer in the UK and approximately 37500 new cases are diagnosed each year (1). Patients with colorectal cancer commonly present to the outpatient setting with changes in bowel habit, rectal bleeding or iron deficiency anaemia. Cases are also identified via the national bowel cancer screening programme and another group of patients present acutely with bowel obstruction or perforation.

Outside these common modes of presentations, there are rare manifestations and we present such a case which demonstrates the value of modern imaging modalities and careful analysis of results.

CASE REPORT

A 78-year-old woman was admitted to our hospital with a week-long history of lethargy and lower back pain. She was found to be tender over her lumbar paraspinal muscles and was pyrexial. Laboratory investigations revealed iron deficiency anaemia (Hb – 9.8, Ferritin  8.0) and a raised C-reactive protein (324). An MRI scan of her spine shower L1/L2 discitis (figure 1) and she was commenced on intravenous antibiotics.

Fig 1: Sagittal view of MRI scans of spine confirming discitis of L1/L2 (indicated by arrow).

Urine microscopy was normal but blood cultures taken on the day of admission grew Escherichia coli (E.coli) and this, coupled with iron deficiency anaemia prompted a search for other pathology and a CT scan of chest, abdomen and pelvis showed thickening of the ascending colon (figures 2 and 3). A colonoscopy confirmed a fungating lesion in the mid-ascending colon and biopsies showed adenocarcinoma of the colon. Our patient then had a laparoscopic right hemicolectomy followed by adjuvant chemotherapy for a moderately differentiated adenocarcinoma (pT4 N2 V1 R0 M0). Her discitis settled with antibiotic treatment.

Fig 2: Axial view of the CT abdomen showing thickening of the ascending colon (indicated by arrow).

Fig 3: Coronal view of the CT abdomen showing thickening of the ascending colon (indicated by arrow).

DISCUSSION

Bacterial translocation is defined as the phenomenon by which organisms or their products escape from the intestinal tract to extra-luminal sites such as mesenteric lymph nodes, liver, spleen and the bloodstream (2). There are several factors that influence bacterial translocation including permeability of the gut mucosal barrier, host defence mechanism, immune status and type and load of gastrointestinal flora. These factors play a key role in determining the ease of bacterial translocation. It is known that bowel obstruction promotes bacterial translocation (3), but the pathophysiological significance of this process remains controversial in colorectal cancer. Some authors have suggested that although translocation takes place in bowel cancer patients, this is not of any clinical significance (4). It is thought that bacterial translocation occurs by intracellular passage but in colorectal cancer patients, the intestinal epithelium can also be penetrated by the extracellular route (5). The translocating bacteria can then spread to other sites in the body via the lymphatic and/or vascular route and cause infection at the distant sites. Bacterial endocarditis is well described in colorectal cancer (6,7) and septic arthritis has been also reported (8,9). We are aware of a case report of Streptococcus bovis discitis originating from colorectal malignancy (10) but we think this is the first reported case of E. coli discitis associated with colonic cancer.

With common diseases, it remains important to remember that atypical presentations do occur. It this case, the combination of iron deficiency anaemia and E. coli discitis alerted us to a possible gastrointestinal pathology and this was easily proven with cross-sectional imaging and subsequent colonoscopy.

This case lends support to the hypothesis of bacterial translocation during periods of intestinal disease and the locally advanced nature of the cancer would seem to have allowed route of bacterial passage via the lymphatic or vascular route.

REFERENCES

1. References Cancer Research UK. Cancer Stats: http://info.cancerresearhuk.org/cancerstats/types/bowel/ [Accessed 10th June 2011]
2. Deitch EA, Winterton J, Li M. The gut as a portal of entry of bacteraemia: role of protein malnutrition. Ann Surg 1981; 205:681–92
3. Sagar PM, Macfie J, Sedman P et al. Intestinal obstruction promotes gut translocation of bacteria. Dis Colon Rectum 1995; 38:640–644
4. Taksue Y, Kakehashi M, Ohge H et al. Bacterial translocation: Not a clinically relevant phenomenon in colorectal caner. World J Surg 2005; 29:198 –202
5. Berg R. Bacterial translocation from the gastrointestinal tract. Trends in Microbiology 1995; Vol. 3(4):149–154
6. Roses D, Richman H and Localio S. Bacterial endocarditis associated with colorectal carcinoma. Ann Surg. 1974; 179 (2):190–191
7. Trajber I, Solomon A, Michowitz et al. Streptococcus bovis subacute bacterial endocarditis as a presenting symptom of occult double carcinoma of the colon. Journal of surgical oncology 1984; 27(3):186–188
8. Garcia-Porrua C, Gonzalez-gay A, Monterroso R et al. Septic arthritis due to Streptoccus bovis as presenting sign of ‘silent’ colon carcinoma. Rheumatology 2000; 39 (3):338–339
9. Marsal S, Castro Guardiola A, Clemente C et al. Streptococcus bovis endocarditis presenting as acute spondylodiscitis. Br J Rheumatol 1994; 33:403–408
10. Rodrigues Genta P, Carneiro L and Norbert Genta E. Streptococcus bovis bacteremia : Unusual complications. Southern Medical Journal 1998; 91 (12):1167–1169

We report on a case of oesophageal leiomyoma presenting as a potential cause of sudden airway obstruction. The patient presented with a large mass in the oral cavity after an episode of forceful vomiting. The operative findings and histology of which confirmed this to be an oesophageal leiomyoma emanating from the upper oesophagus, a rare finding. The mass was excised and the patient’s subsequent post-operative recovery and follow-up has been unremarkable.

This diagnosis should be borne in mind when one is referred the odd–sounding case of a patient who appears to have ‘coughed up their tonsil’.

INTRODUCTION

Benign tumours, such as leiomyomas account for less than 1% of all hypopharyngeal and oesophageal tumours (1,2). Leiomyoma is the most common benign oesophageal neoplasm, the reported post-mortem incidences range from 0.005% to 5.1%. However in comparison to oesophageal carcinoma they are relatively rare. Usually presenting in the 3rd-5th decade with a male:female ratio of 2:1 (3).

CASE REPORT

We report on a 48-year old female ex-smoker who presented to the Emergency department with a sizeable right-sided oral cavity mass associated with audible stertor, but no significant airway compromise in the upright position. The mass appeared after an episode of forceful vomiting having had a cough for two weeks. The remainder of the history was unremarkable apart from an alcohol intake of up to 27 units of wine a week.

Flexible nasoendoscopy suggested an epithelial attachment of the mass to the right tonsil. The patient subsequently underwent a general anaesthetic examination with possible tonsillectomy. This demonstrated that the large pedunculated necrotic mass was in fact, appearing to arise from the right piriform fossa and not the tonsil confirmed by rigid pharyngolaryngo-upper oesophagoscopy. The upper oesophagoscopy was limited to 20 centimetres (cm) due to mucosal oedema. The mass was ligated at its base (silk suture) and sent for further analysis (figure 1). It is likely that the mass had acutely twisted and protruded into the upper oesophagus causing obstructive symptoms leading to the history of sudden-onset vomiting. The patient’s post-operative recovery was uneventful and an outpatient review was arranged.

Fig 1: Macroscopic excised leiomyoma specimen (5 x 3.5 x 3 cms).

Histology confirmed a benign leiomyoma macroscopically measuring 5 x 3.5 x 3cm, which had been shelled out of a squamous covering with no evidence of dysplastic or malignant change. Further imaging [computerised tomography (CT) of neck] demonstrated a remnant of the base of the lesion low in the piriform fossa possibly going up to the upper oesophagus and a barium swallow revealed some oesophageal mucosal irregularity at approximately 21cm.

Further flexible nasoendoscopy assessment in clinic was concerning and unhelpful in assessing the piriform fossa (right side – erythematous and oedematous), prompting a further general anaesthetic rigid laryngopharyngo-upper oesophagoscopy procedure to exclude a synchronous tumour. This revealed a normal piriform fossae and a stump in the left posterior upper oesophagus at approximately 19cm, with the original silk suture attached to it suggesting that the mass was in fact an oesophageal leiomyoma. Biopsies of the stump confirmed oesophageal mucosa only.

The patient was kept under close review jointly with the general surgeons for flexible oesophageal endoscopy and eventually underwent a flexible endoscopy whereby the stump was removed, confirming the same histology (oesophageal mucosa). The patient is now asymptomatic and has seen been discharged from follow-up.

DISCUSSION

Our case presented with leiomyoma emanating from the upper oesophagus, which is rare as they are most frequently found in the mid to lower oesophagus correlating with the predominant muscular composition found in each of the regions; smooth muscle in the lower third and mixed (smooth/skeletal) muscle in the middle third. They usually appear intramurally our case presented intraluminally as a pedunculated mass with sudden onset symptoms of nausea and vomiting as opposed to the usual non-specific, longer duration symptoms of dysphagia, pain and weight loss (3).

Macroscopically leiomyomas are characterised as well circumscribed, smooth or nodular masses with a whorled cut surface (figure 2) and microscopically with bundles of smooth muscle cells intermingled with collagenous stroma (4).

Fig 2: Cut surface of specimen demonstrating whorled surface.

Imaging modalities may include CT scanning to assess extra-oesophageal extent, barium swallow (filling defect) and endoscopic ultrasonography to assess which oesophageal layer the mass is in and its size, extent and nature (sold or cystic). Management options are non-operative (asymptomatic) or operative from enucleation via endoscopic, open or thorascopic methods to oesophageal resection and reconstruction for select tumours (greater than 8cm or adherent to the mucosa etc.) (3).

Anecdotally, a similar case was described by Simpson, 1934 of a lady coughing up a mass, which was subsequently swallowed. The patient was erroneously diagnosed as having globus hystericus and presented ten months later with a similar picture associated with sudden onset dysphagia. Furthermore, direct laryngoscopy confirmed a mass suspended by a pedicle from the area of the right piriform fossa hanging into the oesophageal entrance. This appeared to have twisted causing a haemorrhagic necrosis and the acute symptoms (5).

Our case describes the rare occurrence of an upper oesophageal leiomyoma presenting acutely as a large mass in the oral cavity. This diagnosis should be entertained as a differential in the circumstance of an odd-sounding referral of a patient who is reported to have ‘coughed up their tonsil’.

REFERENCES

1. Jones A. Tumours of the hypopharynx and oesophagus. In: Gleeson M, et al., editors. Scott-Brown’s Otorhinolaryngology, Head and Neck Surgery. Great Britain: Hodder Arnold Publishers; 2008. P. 2635
2. Seremetis MG, Lyons WS, deGuzman VC, Peabody JW Jr. Leiomyomata of the esophagus. An analysis of 838 cases. Cancer. 1976. 38(5): 2166-77
3. Lee LS, Singhal S, Brinster CJ, Marshall B, Kochman ML, Kaiser LR, et al. Current management of esophageal leiomyoma. J. Amer. Coll. Surg. 2004. 198(1):136-46
4. Miettinen M, Sarlomo-Rikala M, Sobin LH, Lasota J. Esophageal stromal tumors: a clinicopathologic, immunohistochemical, and molecular genetic study of 17 cases and comparison with esophageal leiomyomas and leiomyosarcomas. Amer. J Surg. Pathol. 2000. 24(2): 211-22
5. Simpson, F. Specimen: Torsion of Fibroid Polypus of Pyriform Fossa, Proc. of the Roy. Soc. Med. 1934. 27(3): 216

We present the case of a transverse fracture of the scapula resulting from the use of electronic muscle stimulation (EMS): highlighting the dangers of these devices that are commonly used for massage and body-building purposes.

INTRODUCTION

Isolated fractures of the scapular are relatively rare and often the result of direct high-energy trauma (1,2). Occasionally they result from indirect injury with reports of cases following electrocution, but this is rare (3,4). The common orthopaedic injury to the upper limb following electric shock is a posterior fracture-dislocation of the proximal humerus. This is due to powerful contraction of the muscles around the shoulder forcing the humeral head superiorly and posteriorly against the acromion, and medially against the glenoid before it slips behind the glenoid rim into posterior dislocation. We present the unusual case of a transverse scapula fracture following the use of EMS which we propose occurred due to a similar mechanism as those suffering an electric shock.

CASE REPORT

A 51 year old, right-handed martial arts teacher, presented to accident & emergency with a painful and swollen right shoulder. He gave a history of using an EMS machine whilst lying in bed with electrode pads positioned over the posterior, superior aspect of the shoulder and over Latissimus dorsi, both pads on the right side. Accidentally the machine was turned up to full power and this resulted in two powerful muscular contractions, causing full adduction and internal rotation of the right arm for over five seconds. There was no history of direct trauma to the shoulder or arm.

Figure 1 Radiograph showing fracture to blade of scapula

On examination there was restriction of the range of movement of the right shoulder with tenderness and swelling over the scapular. The skin was intact. There was no neurovascular deficit and the patient was otherwise physically well. X-rays revealed a fracture of the body of the scapula with posterior displacement of the inferior fragment. The shoulder joint was not dislocated and there were no associated fractures of the proximal humerus. The patient was admitted for analgesia and computed tomography was performed to assess the fracture.  The scan showed involvement of the glenoid, with the fracture extending through the posterior lip. This was deemed inappropriate for surgical fixation. The patient was managed in a broad arm sling and discharged with simple range of movement exercises.

Figure 2 Computed tomographic scan, showing fracture extension into glenoid

Review in clinic at 4 weeks showed an improvement in range of movements and improved pain and the patient was referred for physiotherapy. Upon review at 10 weeks, the scapular was clinically united with no residual tenderness. Range of movement was full in abduction and flexion, but both internal and external rotation was still limited and the patient continued with physiotherapy for active range of movement exercises.

Figure 3 Computed tomographic scan with 3D reconstruction showing the scapula fracture

DISCUSSION

The unusual injury that we report probably occurred due to the same mechanisms as those suffered during electric shock. We hypothesise that the electrical stimulation caused forceful and uncontrollable contraction of Latissimus dorsi and the upper portion of Trapezius in opposing directions. This created forces great enough to fracture the scapula.

We present this case for two reasons: firstly this is an unexpected and unreported cause of scapular fracture; secondly, as this type of injury has not previously been recognised, there are safety implications regarding the use of the electronic muscle stimulation apparatus. Searching through the literature supporting the use of EMS, there is no mention of the possibility of causing bony injury in the warnings or precautions section. Users and manufacturers of the EMS systems must be aware of this rare but serious risk.

REFERENCES

1. Walter MC, Evans EB. Fractures of scapula. An analysis of forty cases and a review of literature. J Bone Joint Surg Am.1977. Apr 59(3):358-62
2. McAtee SJ. Low-energy scapular body fracture: A case report. Am J Orthop. 1999.
   Aug 28(8): 468-72
3. Rana M, Banerjee R. Scapular fracture after electric shock. Ann R Coll Surg Engl. 2006.
   Mar 88(2):3-4
4. Kotak B, Haddo O, Iqbal M, Chissell H. Bilateral scapular fractures after electrocution. J R Soc Med. 2000. Mar 93:143-144
]]> http://jscr.co.uk/2012/01/trauma-orthopaedic-surgery/scapular-fracture-following-electronic-muscle-stimulation/feed/ 0 http://jscr.co.uk/2012/01/hepatobiliary-surgery/clostridium-perfringens-sepsis-and-liver-abscess-following-laparoscopic-cholecystectomy/ http://jscr.co.uk/2012/01/hepatobiliary-surgery/clostridium-perfringens-sepsis-and-liver-abscess-following-laparoscopic-cholecystectomy/#comments Sat, 07 Jan 2012 02:12:10 +0000 JSCR http://jscr.co.uk/?p=17640 ABSTRACT

Clostridium perfringens sepsis with intravascular haemolysis is a catastrophic process with a reported mortality of between 90 to 100%. We successfully treated a case of severe clostridial infection with a liver abscess following laparoscopic cholecystectomy, the first to our knowledge. A 59-year-old man presented one week after an uneventful laparoscopic cholecystectomy with jaundice, peritonism, sepsis and acute renal failure. He was found to have a haemolytic anaemia, unconjugated hyperbilirubinemia and blood cultures grew Clostridium perfringens. A CT revealed a large gas forming abscess in the gallbladder fossa and right lobe of liver. He was treated with directed antibiotic therapy and underwent emergency laparotomy, drainage of the abscess and peritoneal washout. He required intensive care support, parenteral nutrition and inotropic support for a limited period. CT liver angiogram post op was normal. Continued renal dysfunction necessitated protracted haemofiltration. This resolved and the patient was discharged home at 2 months.

INTRODUCTION

Clostridium perfringens sepsis with intravascular haemolysis is a catastrophic process with a reported mortality of between 70 to 100%. (1)

Cases of clostridial infections following conventional biliary surgery first appear in the literature in 1960’s (2). Graybeal summarised 3 cases of clostridial sepsis and highlighted a reported total of 24 in 1982 (3). It is around this time that laparoscopic surgery began to explode onto the scene. Despite the ubiquitous practice of this operation there have been fortunately few reports of clostridial sepsis as a sequelae.  The first report was in 1996 (4)and there have been few since (5) (4 in total).

The clinical pattern varies in the cases observed – sepsis, peritonitis and emphysematous myonecrosis being the variants to date. Rapid deterioration, haemolysis, septic shock and renal dysfunction are common to all (2-6). We observed a case of severe clostridial infection with a liver abscess following laparoscopic cholecystectomy, the first to our knowledge.

CASE REPORT

A 59 year old man with non-insulin dependent diabetes was initially admitted with gallstone pancreatitis. He underwent an Endoscopic retrograde cholangiopancreatography (ERCP) to clear a single stone from the common bile duct and a papillotomy was performed. After convalescing he proceeded to laparoscopic cholecystectomy at a 6 month interval. No cholangiogram was indicated given the recent papillotomy and he was discharged within 24hours without event.

One week later he re-presented with jaundice, peritonism, sepsis and acute renal failure. On arrival his Haemoglobin measured 9.1 but fell to 6.8g/dl within 12hours. Initial blood samples were haemolysed and a blood film revealed marked red cell spherocytes and occasional nucleated red cell – in keeping with haemolysis. Blood cultures grew Clostridium perfringens.

Initial treatment comprised intravenous fluid resuscitation, high flow oxygen and broad spectrum antibiotics (Tazocin, Gentamicin and Metronidazole). He proceeded to CT scan which demonstrated a gas filled cavity in and under the right lobe of liver. A laparotomy was performed, revealing a large abscess cavity in the right lobe of liver emanating from the gallbladder fossa. This contained pockets of gas. The bile duct was found intact and the cystic duct clips secure. The pus was drained and a generous peritoneal lavage performed. Large bore (32Fr) sub-hepatic and sub-phrenic drains were placed.

Post-operatively the patient was ventilated, continued on broad spectrum antibiotics, transfused packed red cells, fed parenterally initally, and haemofiltered for acute renal failure. Ventilatory and inotropic support were able to be weaned quickly and the jaundice improved over the following week. The abdominal drains were removed sequentially once dry.

There was a protracted recovery period thereafter with bilateral pleural effusions and persistent renal dysfunction. At 3 weeks post re-admission a repeat CT revealed a persistent small collection in the gallbladder fossa and CT guided drainage was attempted through the right 8^th intercostal space. This yielded minimal debris only. A massive haemothorax developed after the patient was heparinised for haemofiltration later that day. Intercostal chest drain yielded over 2 litres of blood and clot and a mini-thoracotomy was later required to clear the pleural cavity completely.



Fig 1: CT at re-admission 1 week post lap chole - Free intraperitoneal gas abscess in gallbladder fossa



Fig 2: Findings at laparotomy - Purulent slough and emphysema in the gallbladder fossa



Fig 3: Repeat CT post-laparotomy and lavage showing large bore peritoneal drains

The patient was transferred to the care of the renal physicians. The renal function improved, diuresis resumed and the patient eventually weaned from dialysis. He was discharged at 2 months.

DISCUSSION

Infectious complications of laparoscopic cholecystectomy most commonly consist of port site infection and intra-abdominal abscess. The risk of these complications has been noted to be reduced from an incidence of 2.8 to 0.5% by the administration of peri-operative antibiotics_.

In a 10-year study of pyogenic liver abscess, Clostridium perfringens was cultured in only one of 79 cases (7) making it a relative rarity as a cause of primary liver abscess. The organism is widely distributed in the intestinal flora of the majority of humans and a small percentage of normal and inflamed gallbladders. Positive cultures for Clostridium perfringens have been reported to be found in 1 to 19% of surgically removed gallbladders (8). The question of the pathogenesis of clostridial sepsis post cholecystectomy remains unanswered given that such a low incidence is reported in the face of a relatively high rate existence of the bacteria.

There are various theories as to the development of bactobilia; entero-hepatic, ascending & haematogenous routes.

The diagnosis of Clostridium perfringens sepsis following laparoscopic cholecystectomy requires a high index of suspicion. The number of reported cases is too few to allow analysis of the common presenting factors. The mode of presentation does however comprise anecdotally consistent findings; abdominal pain, jaundice, haemolysis, renal dysfunction, confusion and occasionally crepitus, if gas gangrene is present. Haemodynamic parameters may be falsely re-assuring initially. Significantly the patients are almost always diabetic.

The mode of presentation is often similar to necrotising fascitiis, which has been more widely reported following laparoscopic surgery and is most often the result of mixed bacteriological agents, commonly gram negative enteric bacilli, staphlococcal and streptococcal species.

It is thought that Clostridium perfringens sepsis presents in three different modes – simple contamination, clostridial crepitant cellulitis and finally as gas gangrene.

Our case is unique not only as Clostridium perfingens sepsis following laparoscopic cholecystectomy is rarely seen, but for being the first to report an associated gas forming liver abscess. That the patient survived is surprising and attributed to the rapid and aggressive intervention to remove the nidus of infection.

REFERENCES

1. Rogstad B, Ritland S Lunde S, Hagen AG. Clostridium Perfringens septicaemia with massive hemolysis. Infection. 1993. 21:60-2
2. Turner FP. Fatal Clostridium Welchii Septicemia Following Cholecystectomy. Am J Surg. 1964 Jul. 108:3-7
3. Graybeal GE. Clostridial sepsis following biliary surgery: a report of three patients and a review of the literature. Del Med J. 1982 Sep. 54(9):493-8
4. Bush GW, Clements RH, Phillips M, Kent RB Jr. Clostridium perfringens sepsis with intravascular hemolysis following laparoscopic cholecystectomy: a newly reported complication. Am Surg. 1996 Apr. 62(4):326-7
5. Samel S, Post S, Martell J, Becker H. Clostridial gas gangrene of the abdominal wall after laparoscopic cholecystectomy. J Laparoendosc Adv Surg Tech A. 1997 Aug. 7(4):245-7
6. Mittermair RP, Schobersberger W, Hasibeder W, Allerberger F, Peer R, Bonatti H. Necrotizing fasciitis with Clostridium perfringens after laparoscopic cholecystectomy. Surg Endosc. 2002 Apr. 6(4):716. Epub 2001 Nov 30
7. Hansen PS, SchØnheyder HC. Pyogenic hepatic abscess. A 10-year population-based retrospective study. APMIS. 1998 Jan. 106(1-6): 396–402
8. Magee HR. Clostridium Perfringens Infection of the Biliary Tract. ANZ Journal of Surgery. 1967 Nov. 37(2): 177–183
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