History and findings: A 45-year-old man with type I diabetes mellitus
was admitted to hospital because of colicky abdominal pain and 5-6 wat
ery stools daily. Upper gastrointestinal endoscopy showed nearly total
atrophy of the villi in the duodenum and jejunum suggesting coeliac d
isease. However, gluten-free diet for 2 weeks brought no improvement.
Another examination of the biopsy 6 weeks after the first endoscopy re
vealed extensive collagen deposition in the lamina propria of the smal
l intestine, giving the diagnosis of collagenous sprue. Treatment and
course: Parenteral nutrition, lactulose, cisaprid, colestyramine, doxy
cyclin, paromomycin, vancomycin and octreotide failed to affect the lo
ss of fluid from the gut which 12 weeks after admission had increased
to 221 daily. However it was stopped after prednisolone was administer
ed (100 mg daily). 7 months after starting the steroid treatment the c
ollagen layer had disappeared and the villous atrophy had partially re
gressed. Over the next 6 months the prednisolone dosage was decreased
to 10 mg daily. Shortly thereafter a perimembranous glomerulonephritis
occurred, with proteinuria (up to 60 g/d) and oedema. It regressed to
6 g/d when the steroid dose was increased and cyclosparin, 0.5 g/d, h
ad been added. On maintenance dosage of cyclosporin the histological a
nd clinical remission of the collagenous sprue has now lasted for over
2 years. Conclusions: This case suggests that steroid administration
is an effective treatment of collagenous sprue. The presence of diabet
es and other immune-related diseases in this case also suggests that a
n immunological mechanism may play a causative role in collagenous spr
ue.