Collagenous sprue: a clinicopathologic study of 12 cases.

Hdl Handle:
http://hdl.handle.net/10147/207633
Title:
Collagenous sprue: a clinicopathologic study of 12 cases.
Authors:
Maguire, Aoife A; Greenson, 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.ie
Citation:
Am J Surg Pathol. 2009 Oct;33(10):1440-9.
Journal:
The American journal of surgical pathology
Issue Date:
1-Feb-2012
URI:
http://hdl.handle.net/10147/207633
DOI:
10.1097/PAS.0b013e3181ae2545
PubMed ID:
19641452
Abstract:
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:
eng
MeSH:
Adult; Aged; 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
ISSN:
1532-0979 (Electronic); 0147-5185 (Linking)

Full metadata record

DC FieldValue Language
dc.contributor.authorMaguire, Aoife Aen_GB
dc.contributor.authorGreenson, Joel Ken_GB
dc.contributor.authorLauwers, Greg Yen_GB
dc.contributor.authorGinsburg, Richard Een_GB
dc.contributor.authorWilliams, Geraint Ten_GB
dc.contributor.authorBrown, Ian Sen_GB
dc.contributor.authorRiddell, Robert Hen_GB
dc.contributor.authorO'Donoghue, Diarmuiden_GB
dc.contributor.authorSheahan, Kieran Den_GB
dc.date.accessioned2012-02-01T10:33:27Z-
dc.date.available2012-02-01T10:33:27Z-
dc.date.issued2012-02-01T10:33:27Z-
dc.identifier.citationAm J Surg Pathol. 2009 Oct;33(10):1440-9.en_GB
dc.identifier.issn1532-0979 (Electronic)en_GB
dc.identifier.issn0147-5185 (Linking)en_GB
dc.identifier.pmid19641452en_GB
dc.identifier.doi10.1097/PAS.0b013e3181ae2545en_GB
dc.identifier.urihttp://hdl.handle.net/10147/207633-
dc.description.abstractCollagenous 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.en_GB
dc.language.isoengen_GB
dc.subject.meshAdulten_GB
dc.subject.meshAgeden_GB
dc.subject.meshAged, 80 and overen_GB
dc.subject.meshCeliac Disease/*immunology/*pathology/therapyen_GB
dc.subject.meshClone Cellsen_GB
dc.subject.mesh*Collagenen_GB
dc.subject.meshDiet Therapyen_GB
dc.subject.meshFemaleen_GB
dc.subject.meshHumansen_GB
dc.subject.meshImmunohistochemistryen_GB
dc.subject.meshImmunosuppressive Agents/therapeutic useen_GB
dc.subject.meshMaleen_GB
dc.subject.meshMiddle Ageden_GB
dc.subject.meshPolymerase Chain Reactionen_GB
dc.subject.meshT-Lymphocyte Subsets/immunologyen_GB
dc.subject.meshT-Lymphocytes/immunologyen_GB
dc.titleCollagenous sprue: a clinicopathologic study of 12 cases.en_GB
dc.contributor.departmentDepartment of Histopathology, St Vincent's University Hospital, Dublin, Ireland. , Aoife.maguire@ucd.ieen_GB
dc.identifier.journalThe American journal of surgical pathologyen_GB
dc.description.provinceLeinster-

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