Authors
Maguire, Aoife AGreenson, Joel K
Lauwers, Greg Y
Ginsburg, Richard E
Williams, Geraint T
Brown, Ian S
Riddell, Robert H
O'Donoghue, Diarmuid
Sheahan, Kieran D
Affiliation
Department of Histopathology, St Vincent's University Hospital, Dublin, Ireland. , Aoife.maguire@ucd.ieIssue Date
2012-02-01T10:33:27ZMeSH
AdultAged
Aged, 80 and over
Celiac Disease/*immunology/*pathology/therapy
Clone Cells
*Collagen
Diet Therapy
Female
Humans
Immunohistochemistry
Immunosuppressive Agents/therapeutic use
Male
Middle Aged
Polymerase Chain Reaction
T-Lymphocyte Subsets/immunology
T-Lymphocytes/immunology
Metadata
Show full item recordCitation
Am J Surg Pathol. 2009 Oct;33(10):1440-9.Journal
The American journal of surgical pathologyDOI
10.1097/PAS.0b013e3181ae2545PubMed ID
19641452Abstract
Collagenous sprue is a rare form of small bowel enteropathy characterized by chronic diarrhea and progressive malabsorption with little data available on its natural history. The pathologic lesion consists of subepithelial collagen deposition associated with variable alterations in villous architecture. The small bowel biopsies of 12 cases were reviewed. Clinical details, celiac serology, and T-cell receptor gene rearrangement study results, when available, were collated. There were 8 females and 4 males (age ranged from 41 to 84 y) who presented with chronic diarrhea and weight loss. Small intestinal biopsies showed subepithelial collagen deposition with varying degrees of villous atrophy and varying numbers of intraepithelial lymphocytes. Four patients had previous biopsies showing enteropathic changes without collagen deposition. Seven cases were associated with collagenous colitis and 1 also had features of lymphocytic colitis. Three patients also had collagen deposition in gastric biopsies. One case was associated with lymphocytic gastritis. Celiac disease (CD, gluten-sensitive enteropathy) was documented in 4 patients. Five patients made a clinical improvement with combinations of a gluten-free diet and immunosuppressive therapy. Two patients died of complications of malnutrition and 1 of another illness. Clonal T-cell populations were identified in 5 of 6 cases tested. Four of these patients improved clinically after treatment but 1 has died. Collagenous sprue evolved on a background of CD in 4 cases. There was no history of CD in others and these cases may be the result of a biologic insult other than gluten sensitivity. None has developed clinical evidence of lymphoma to date.Language
engISSN
1532-0979 (Electronic)0147-5185 (Linking)
ae974a485f413a2113503eed53cd6c53
10.1097/PAS.0b013e3181ae2545
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