Volume 69 - 2006 - Fasc.2 - Case reports
Intra-abdominal lymphatic malformation
Intra-abdominal lymphangioma is a rare congenital lymphatic vascular malformation. It is found, most commonly, in adult patients who present with abdominal pain, and whom medical imaging (ultrasound, endoscopy and tomography) reveals a cystic intra-abdominal mass. Such masses may be on the mesentery, in contact with abdominal viscera or may be part of the visceral wall. Diagnosis of intra-abdominal lymphangioma is difficult because of its rarity, and because other pathologies may have the same radi- ological signs. The diagnosis is confirmed by anatomopathological examination with immunohistochemistry. The treatment is complete surgical excision, in order to prevent recurrence. (Acta gastroenterol. belg., 2006, 69, 209-212).
Complicated granulomatous colitis in a patient with Hermansky-Pudlak syndrome, successfully treated with infliximab
Hermansky-Pudlak syndrome (HPS) is a rare autosomal reces- sive disorder which is characterised by the triad of oculocutaneous albinism, platelet dysfunction and accumulation of ceroidlike pigment in tissues.
Complications of the syndrome, such as fatal pulmonary fibro- sis, renal failure and cardiomyopathy have been described. Granulomatous colitis has been documented in several families with the HPS. The bowel disease of the HPS is a unique type of inflammatory bowel disease with clinical features suggestive of idiopathic ulcerative colitis (UC) and pathologic features sugges- tive of Crohn's disease.
We report a patient with HPS which was complicated by granulomatous colitis with perineal and rectovaginal fistulas refractory to antibiotics and azathioprine but dramatically responded to repeated infusions of infliximab. (Acta gastroenterol. belg., 2006, 69, 213-216).
Miliary tuberculosis following infliximab therapy for Crohn disease : A case report and review of the literature
We present a case of miliary tuberculosis diagnosed 15 months after infliximab treatment despite negative screening for previous exposure to Mycobacteria on skin PPD and chest X-ray. This case shows that - although screening for TB with a skin PPD and a chest X-ray should be performed in all patients - this is not 100% effective and may be a problem in patients on concomitant immunosuppression. The clinical course of this patient further shows that in a patient treated with anti-TNF antibodies who's condition does not improve one should always be aware of the pos- sibility of a tuberculosis infection. Even though tuberculosis is usu- ally not rapidly fatal, the disease may show a fulminant course in immunocompromised patients. (Acta gastroenterol. belg., 2006, 69, 217-220).
An unusual cause of acute lower gastrointestinal bleeding
A 45 year old male patient was referred to hospital after syn- cope during defaecation. Rapid peranal blood loss occurred shortly after admission. Gastroscopy and coloscopy performed as first line diagnostic measures failed to detect the source of haemorrhage. Ultrasound (US) revealed a hypoechoic and hyper- vascularized tumor mass in the right lower abdomen. A gastro- intestinal stromal tumor (GIST) of the jejunum was diagnosed after laparatomy. Collectively US should be among the first line diagnostic procedures in younger patients presenting with lower gastrointestinal haemorrhage. (Acta gastroenterol. belg., 2006, 69, 221-223).
