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Blood, Vol. 92 No. 10 (November 15), 1998:
pp. 3879-3886
Are Complicated Forms of Celiac Disease Cryptic T-Cell Lymphomas?
Franck Carbonnel,
Laurence Grollet-Bioul,
Jean Claude Brouet,
Marie Françoise Teilhac,
Jacques Cosnes,
Régis Angonin,
Marie Deschaseaux,
François-Patrick Châtelet,
Jean Pierre Gendre, and
François Sigaux
From the Service de Gastroentérologie et Nutrition and the
Laboratoire d'Anatomopathologie, Hôpital Rothschild, Paris,
France; INSERM U462, Paris, France; the Service
d'immunohématologie, Hôpital Saint Louis, Paris, France;
the Laboratoire d'Anatomopathologie, CHU de Besançon,
Besançon, France; and the Etablissement de transfusion sanguine
de Franche Comté, Besançon, France.
We assessed the clonality of duodenal mucosal T cells in patients
with celiac disease and controls. Fifteen adult patients were studied.
Four patients had a complicated celiac disease, 3 did not respond to a
gluten-free diet, and 2 had an ulcerative jejunitis (including 1 patient with nonresponsive celiac disease). Seven patients had an
untreated celiac disease responsive to a gluten-free diet. Histological
examination of duodenal biopsies of these 11 patients showed
benign-appearing celiac disease without evidence of lymphoma. Four
patients with nonulcer dyspepsia and normal duodenal biopsies served as
controls. TCR gene rearrangements were analyzed by multiplex
polymerase chain reaction on DNA extracted from duodenal biopsies.
Major clonal rearrangements of the T-cell receptor were found in 4 cases, all with complicated celiac disease. Monoclonality was confirmed
by DNA sequencing of the junctional region in 3 cases and by
hybridization with clone-specific oligoprobes. Patients with celiac
disease responsive to gluten-free diet had mainly a polyclonal pattern,
with 1 of them having an oligoclonal rearrangement. An oligoclonal
pattern was also observed in 2 control patients. Three patients with
complicated celiac disease evolved to T-cell lymphoma with liver (n = 2) or bone marrow (n = 1) invasion. Identical clones were found in
the enteropathic duodenojejunum and peripheral blood in the patient
with large-cell lymphoma with bone marrow invasion. This study suggests
that complicated celiac disease is a cryptic T-cell lymphoma.
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