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    Collagenous sprue: a clinicopathologic study of 12 cases.

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    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
    Issue Date
    2012-02-01T10:33:27Z
    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
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    Citation
    Am J Surg Pathol. 2009 Oct;33(10):1440-9.
    Journal
    The American journal of surgical pathology
    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
    ISSN
    1532-0979 (Electronic)
    0147-5185 (Linking)
    ae974a485f413a2113503eed53cd6c53
    10.1097/PAS.0b013e3181ae2545
    Scopus Count
    Collections
    St. Vincent's University Hospital

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