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Diffuse Cavernous Haemangioma of the Rectum: Case Report

 

Authors: Abdihakim M, MBChB1, Athar A. MD2, 1-Department of Surgery, AgaKhan University hospital, Nairobi, Kenya., 2-Department of Surgery,, AgaKhan Hospital, Dar es Salaam, Tanzania.

Correspondence: abdi.mohammed@aku.edu, Aga Khan University Hospital, P.O. Box 30270-00100 Nairobi

 

Summary

Diffuse carvenous haemangioma of the rectum, can be misdiagnosed as other common anorectal conditions due to the overlapping clinical presenta-tion and can be caused of massive lower gastrointestinal bleeding leading to significant morbidity. The classic colonoscopic and CT scan findings should be sought and early referral to centres with these investigative modalities should be considered. Treatment includes a complete resection by pull through transection and coloanal anastomosis.

Introduction

Diffuse cavernous haemangiomas of the rectum (DCHR) are rare. Usually confused with anorectal conditions that present with bright red bleeding, its correct treatment is commonly delayed(1). Up to 80% of patients with the DCHR have undergone a minimum of one inappropri-ate surgical procedure due to misdiagnosis (2). Proper understanding of the condition and careful use of imag-ing modalities is essential in its early treatment (3). With the paucity of resources in the developing world, the di-agnostic challenge is even more daunting. We present a case of DCHR in a 24 year old managed at our hospital. To our knowledge, this is the first reported case of DCHR in the East African literature.

 

Case Report

A 24 year old female presented with a five month history of recurrent fresh and painless rectal bleeding, occasional passage of clots and had a sensation of incomplete rectal emptying. She denied melaena, haematemesis or muco-sal bleeding. There was no history of abdominal pain, altered bowel habits (no constipation or diarrhoea) or weight loss. Her past surgical history was significant for several transfusions for severe anemia, hemorrhoidecto-my for rectal bleeding, left hemicolectomy with primary anastomosis one year earlier for massive lower gastroin-testinal bleeding requiring seven units of blood transfu-sion. She had no family history of malignancy. She was a college student with no history of smoking and only took alcohol occasionally.

 

 

On first encounter at our institution, she was very pale, not dyspnoeic or jaundiced, the patient was well hydrat-ed, had no generalized lymphadenopathy or lower limb oedema. The blood pressure of 100/70 mmHg, a pulse rate of 84 beats per minute and temperature of 36.8oC. She was fully oriented and had a normal respiratory and cardiovascular examination. Abdominal examination revealed a non-tender abdomen, a well-healed laparoto-my scar and no palpable masses. Digital rectal examina-tion revealed a normal anal tone, smooth anorectal mu-cosa, no masses, and soft stools which were non-bloody. Anoscopy was essentially non-revealing.

 

Her haemoglobin level was 3.6mg/dl and she received four units of packed cells at admission. The coagulation screen was normal. She was prepared for colonoscopy and the findings are shown on Fig. 1. Bleeding contin-ued after colonoscopy necessitating tranfusion of one more unit of packed red blood cells. A CT scan abdo-men done revealed the typical thickening of the rectal wall with multiple serpentine vessels and phleboliths (Fig 2a-c).She was counselled for low anterior resec-tion and coloanal anastomosis. After adequate colonic preparation, her pre-operative haemoglobin was 10.8g/ dl and three more units of packed cells were available for surgery. Through a midline abdominal incision, the pelvis was explored, rectum mobilised and an arterio-venous malformation (AVM) extending from the recto-sigmoid junction to the anorectal junction was found. The AVM was carefully excised and hand-sewn coloanal anastomosis done with a diverting loop transverse colostomy fashioned.

 

Click to view figure 1

 

 

Total operative time was four hours and total blood loss was 250mls. She had an unevent-ful post-operative period and was discharged on the 9th postoperative day.

 

On follow up in the surgical outpatient clinic, a gastro-graffin enema revealed stenosis at the anastomosis and rectovaginal fistula 1.5cm from the anal verge. The latter has subsequently closed spontaneously and she is due for colostomy closure. The stenosis responded to serial anal dilatations.

 

The surgical specimen (Fig 3) was sent for histology and confirmed the diagnosis of Diffuse Cavernous Haeman-gioma of the Rectum.

 

Discussion

Diffuse Cavernous Haemangioma of the Rectum (DCHR) is a rare benign lesion described in all age groups and may present with severe bleeding per rec-tum. It was first described by Philips in 1839 since when about 100 cases of DCHR have been reported (4). It may mimic internal haemorrhoids, ulcerat