Chediak-Higashi syndrome with intestinal complication. Report of a case.
A 21-year-old woman with Chediak-Higashi syndrome developed intestinal lesions similar to those of Crohn's disease. Radiographic findings showed a stenotic lesion with multiple nodular changes in the ascending colon and cecum, and a longitudinal ulceration in the terminal ileum. In addition, multiple oval-shaped ulcerations were seen between the terminal ileum and descending colon. However, serial histological examinations revealed only an infiltration of lymphoid cells, which was a nonspecific finding. Though there are few reports of gastrointestinal involvement in Chediak-Higashi syndrome, such an intestinal complication may develop in patients long surviving with this disease.
Ishii
E
E
Department of Pediatrics, Faculty of Medicine, Kyushu University, Fukuoka, Japan.
Matui
T
T
Iida
M
M
Inamitu
T
T
Ueda
K
K
D000328:Adult; D002609:Chediak-Higashi Syndrome / Q000473:pathology*; D003106:Colon / Q000473:pathology; D003424:Crohn Disease / Q000473:pathology*; D005260:Female; D006801:Humans; D007413:Intestinal Mucosa / Q000473:pathology