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. 1996 Apr;38(4):619–622. doi: 10.1136/gut.38.4.619

Colitis complicating chronic granulomatous disease. A clinicopathological case report.

J M Sloan 1, C H Cameron 1, R J Maxwell 1, D R McCluskey 1, J S Collins 1
PMCID: PMC1383126  PMID: 8707099

Abstract

BACKGROUND--This report concerns a female patient now aged 24 years, diagnosed at the age of 7 years as suffering from chronic granulomatous disease. At age 20 she developed diarrhoea accompanied by rectal bleeding. Endoscopy showed extensive colitis. She failed to respond to medical treatment and underwent total colectomy two years later. AIMS--To discuss the histological changes in the colon in chronic granulomatous disease. RESULTS--There was extensive mucosal inflammation throughout colon and rectum resembling ulcerative colitis. In addition characteristic large pigmented macrophages were distributed in the basal mucosa and superficial submucosa. Similar cells accompanied by granulomata were present in mesenteric lymph nodes. CONCLUSIONS--Colitis is an unusual clinical manifestation of chronic granulomatous disease but the presence and characteristic distribution of such pigmented macrophages in colonic biopsy in children and young adults may suggest the diagnosis.

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Selected References

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  1. Ament M. E., Ochs H. D. Gastrointestinal manifestations of chronic granulomatous disease. N Engl J Med. 1973 Feb 22;288(8):382–387. doi: 10.1056/NEJM197302222880802. [DOI] [PubMed] [Google Scholar]
  2. Babior B. M., Woodman R. C. Chronic granulomatous disease. Semin Hematol. 1990 Jul;27(3):247–259. [PubMed] [Google Scholar]
  3. Baehner R. L. Chronic granulomatous disease of childhood: clinical, pathological, biochemical, molecular, and genetic aspects of the disease. Pediatr Pathol. 1990;10(1-2):143–153. doi: 10.3109/15513819009067103. [DOI] [PubMed] [Google Scholar]
  4. Dinauer M. C., Orkin S. H. Chronic granulomatous disease. Annu Rev Med. 1992;43:117–124. doi: 10.1146/annurev.me.43.020192.001001. [DOI] [PubMed] [Google Scholar]
  5. Ectors N., Geboes K., De Vos R., Heidbuchel H., Rutgeerts P., Desmet V., Vantrappen G. Whipple's disease: a histological, immunocytochemical and electronmicroscopic study of the immune response in the small intestinal mucosa. Histopathology. 1992 Jul;21(1):1–12. doi: 10.1111/j.1365-2559.1992.tb00337.x. [DOI] [PubMed] [Google Scholar]
  6. Fisher J. E., Khan A. R., Heitlinger L., Allen J. E., Afshani E. Chronic granulomatous disease of childhood with acute ulcerative colitis: a unique association. Pediatr Pathol. 1987;7(1):91–96. doi: 10.1080/15513818709177120. [DOI] [PubMed] [Google Scholar]
  7. Gallin J. I., Buescher E. S., Seligmann B. E., Nath J., Gaither T., Katz P. NIH conference. Recent advances in chronic granulomatous disease. Ann Intern Med. 1983 Nov;99(5):657–674. doi: 10.7326/0003-4819-99-5-657. [DOI] [PubMed] [Google Scholar]
  8. Harris B. H., Boles E. T., Jr Intestinal lesions in chronic granulomatous disease of childhood. J Pediatr Surg. 1973 Dec;8(6):955–956. doi: 10.1016/0022-3468(73)90022-5. [DOI] [PubMed] [Google Scholar]
  9. Hopkins P. J., Bemiller L. S., Curnutte J. T. Chronic granulomatous disease: diagnosis and classification at the molecular level. Clin Lab Med. 1992 Jun;12(2):277–304. [PubMed] [Google Scholar]
  10. Park S. H., Chi J. G. Chronic granulomatous disease: an ultrastructural study of the pigment laden histiocytes. Pediatr Pathol. 1992 Nov-Dec;12(6):839–847. doi: 10.3109/15513819209024241. [DOI] [PubMed] [Google Scholar]
  11. Quie P. G., White J. G., Holmes B., Good R. A. In vitro bactericidal capacity of human polymorphonuclear leukocytes: diminished activity in chronic granulomatous disease of childhood. J Clin Invest. 1967 Apr;46(4):668–679. doi: 10.1172/JCI105568. [DOI] [PMC free article] [PubMed] [Google Scholar]
  12. Tauber A. I., Borregaard N., Simons E., Wright J. Chronic granulomatous disease: a syndrome of phagocyte oxidase deficiencies. Medicine (Baltimore) 1983 Sep;62(5):286–309. [PubMed] [Google Scholar]
  13. Warren B. F., Shepherd N. A. Diversion proctocolitis. Histopathology. 1992 Jul;21(1):91–93. doi: 10.1111/j.1365-2559.1992.tb00353.x. [DOI] [PubMed] [Google Scholar]
  14. Werlin S. L., Chusid M. J., Caya J., Oechler H. W. Colitis in chronic granulomatous disease. Gastroenterology. 1982 Feb;82(2):328–331. [PubMed] [Google Scholar]
  15. Yogman M. W., Touloukian R. J., Gallagher R. Letter: Intestinal granulomatosis in chronic granulomatous disease and in Crohn's disease. N Engl J Med. 1974 Jan 24;290(4):228–228. doi: 10.1056/NEJM197401242900414. [DOI] [PubMed] [Google Scholar]

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