Isolated Colonic Leukocytoclastic Vasculitis Causing Segmental Megacolon: Report of a Rare Case
Harokopion University of Athens, Athínai, Attica, GreeceDiseases of the Colon & Rectum (Impact Factor: 3.75). 02/2005; 48(1):167-71. DOI: 10.1007/s10350-004-0758-x
We report the case of a 44-year-old white man who presented with progressively worsening crampy abdominal pain and distention. Deterioration of his clinical picture along with leukocytosis and radiographic evidence of severe colonic dilation rendered exploratory laparotomy necessary. Greatly distended and inflamed transverse and descending colon were evident and an extended left colectomy was performed. Characteristic changes of leukocytoclastic vasculitis in the serosal and muscular layers of the resected colon were demonstrated at histopathologic examination. Systemic leukocytoclastic vasculitis, usually coexisting with Henoch-Schonlein purpura, commonly affects the small bowel with clinical evidence of ischemia or bleeding. Colon involvement is infrequently reported in the context of systemic disease. Isolated colonic leukocytoclastic vasculitis without extraintestinal manifestations is rare. A previously unreported case of localized leukocytoclastic vasculitis of the left colon resulting in the impressive presentation of megacolon, without the presence of any precipitating factor or associated systemic disease is presented here, with an overview of the related literature.
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ABSTRACT: Isolated vasculitis of the gastrointestinal (GI) tract is a rare entity. Endoscopic biopsies have low sensitivity to diagnose intestinal vasculitis, even though the endoscopic findings may be suggestive of this condition. Our aims were to describe a case of biopsy-proven colonic leukocytoclastic vasculitis and review the literature. A patient with biopsy-proven colonic leukocytoclastic vasculitis is described. A Medline database search of cases with localized GI vasculitis between January 1985 and September 2005 was conducted. A 32-year-old man was admitted to the hospital because of abdominal pain and diarrhea. A colonic biopsy showed leukocytoclastic vasculitis. There are very few articles on leukocytoclastic GI vasculitis as a separate disease, and most of them emphasize the difficulty in classification. Unlike our case, in former cases of localized vasculitis a diagnosis was made after surgery. Although our patient had steroid-refractory biopsy-proven isolated intestinal vasculitis, treatment with intravenous cyclophosphamide resulted in rapid resolution of symptoms and surgery was not required. In patients with abdominal pain a diagnosis of intestinal vasculitis should be considered. Immunosuppressive therapy allowed our patient to avoid surgery and may be similarly beneficial in other similar cases.
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ABSTRACT: Persistent megacolon that results from frequent episodes of fecal impaction without adequate treatment is a rare and seldom reported condition in the elderly. This report discusses a 72-year-old white woman presenting with a large abdominal mass, who had at least 4 episodes of radiographically demonstrated fecal impaction over the previous year without adequate treatment. The patient required hospitalization for a bleeding rectal ulcer during the second episode of fecal impaction. Computed tomography (CT) scans on this admission revealed a dilated colon up to 16 x 14 cm in maximal dimensions extending over 30 cm, filled with massive fecal material. Several follow-up abdominal radiographs revealed a persistent megacolon after 10 months despite the absence of significant fecal material in the rectosigmoid colon. While multiple contributing factors were likely involved in her frequent fecal impactions, the clinical course of this case suggests that frequent fecal impactions without adequate treatment can lead to megacolon in high-risk patients. Clinicians should aggressively treat fecal impaction and monitor the adequacy of treatment with abdominal radiography in order to avoid significant complications. Complications and management of fecal impaction and the pathophysiology of megacolon in the literature are reviewed and discussed.
Article: Single-organ vasculitis[Show abstract] [Hide abstract]
ABSTRACT: To provide a critical analysis of a rare disorder, single-organ vasculitis, emphasizing those organs in which the excision of the vasculitic lesion can be curative. To recommend a rational approach to diagnosis, longitudinal follow-up and treatment. Patients with focal single-organ vasculitis affecting abdominal and genitourinary organs, breast and aorta have been reported as individual cases and small series. Single-organ vasculitis differs from systemic forms of vasculitis in disease expression and prognosis. Occasionally, what appears to be a localized process evolves into a systemic disease. Depending on the organ affected, some clinical, serological and histopathologic features may be helpful in predicting the extent of the vasculitic process. With the exception of severe ischemic or hemorrhagic complications affecting the abdominal organs and dissection or rupture of the aortic arch, the prognosis of focal single-organ vasculitis tends to be excellent. Resection of the inflammatory lesion may be curative. The diagnosis of focal single-organ vasculitis is always presumptive and requires exclusion of systemic illness at the time of diagnosis as well as throughout the period of continued care. Clues from clinical symptoms, laboratory tests and histopathologic features at the time of diagnosis may assist in devising surveillance strategies.
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