Effect of a Gluten-free Diet on the Risk of Enteropathy-associated T-cell Lymphoma in Celiac Disease

2007 ◽  
Vol 53 (4) ◽  
pp. 972-976 ◽  
Author(s):  
Marco Silano ◽  
Umberto Volta ◽  
Alessandro De Vincenzi ◽  
Mariarita Dessì ◽  
Massimo De Vincenzi ◽  
...  
Keyword(s):  
Celiac Disease ◽  
T Cell ◽  
Cell Lymphoma ◽  
T Cell Lymphoma ◽  
Gluten Free Diet ◽  
Gluten Free

Are Complicated Forms of Celiac Disease Cryptic T-Cell Lymphomas?

Blood ◽  
1998 ◽  
Vol 92 (10) ◽  
pp. 3879-3886 ◽  
Author(s):  
Franck Carbonnel ◽  
Laurence Grollet-Bioul ◽  
Jean Claude Brouet ◽  
Marie Françoise Teilhac ◽  
Jacques Cosnes ◽  
...  
Keyword(s):  
Bone Marrow ◽  
Celiac Disease ◽  
T Cell ◽  
Cell Lymphoma ◽  
Large Cell ◽  
Cell Receptor ◽  
T Cell Lymphoma ◽  
Gluten Free Diet ◽  
Gluten Free ◽  
Duodenal Biopsies

Abstract 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. TCRγ 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.

Are Complicated Forms of Celiac Disease Cryptic T-Cell Lymphomas?

Blood ◽  
1998 ◽  
Vol 92 (10) ◽  
pp. 3879-3886
Author(s):  
Franck Carbonnel ◽  
Laurence Grollet-Bioul ◽  
Jean Claude Brouet ◽  
Marie Françoise Teilhac ◽  
Jacques Cosnes ◽  
...  
Keyword(s):  
Bone Marrow ◽  
Celiac Disease ◽  
T Cell ◽  
Cell Lymphoma ◽  
Large Cell ◽  
Cell Receptor ◽  
T Cell Lymphoma ◽  
Gluten Free Diet ◽  
Gluten Free ◽  
Duodenal Biopsies

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. TCRγ 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.

Increased IgA Glycoprotein-2 Specic Antibody Titres in Refractory Celiac Disease

2014 ◽  
Vol 23 (2) ◽  
pp. 127-133 ◽  
Author(s):  
Sascha Gross ◽  
Sjoerd F. Bakker ◽  
Adriaan A. Van Bodegraven ◽  
Ingrid M.W. Van Hoogstraten ◽  
Kyra A. Gelderman ◽  
...  
Keyword(s):  
Ulcerative Colitis ◽  
Crohn’S Disease ◽  
Celiac Disease ◽  
T Cell ◽  
Cell Lymphoma ◽  
T Cell Lymphoma ◽  
Gluten Free Diet ◽  
Gluten Free ◽  
Refractory Celiac Disease

Background & Aims: In most cases celiac disease (CD) is successfully treated with a gluten-free diet (GFD). However, some patients become refractory to the GFD. Refractory CD (RCD) patients have an increased risk for developing enteropathy associated T-cell lymphoma and early diagnosis is therefore of importance. Currently, RCD diagnosis relies on endoscopy and adequate serological markers are lacking. Antibodies against glycoprotein-2 (GP2A) were described in Crohn's disease (CrD) and active CD but not in ulcerative colitis (UC), suggesting this is a specific marker for small intestinal lesions.Methods: Sera obtained from patients visiting our outpatient clinic for routine serological tests for diagnosis and/or follow-up of inflammatory bowel disease (n=78), active CD (n=45), GFD (n=34) and RCD (n=15) were analysed for GP2A titres.Results: Increased GP2A-IgA levels in CrD and active CD as compared to controls (p<0.001) and lack thereof in UC was confirmed. However, we could not confirm the association with small bowel localization within the CrD patient group. Within CD patients, we demonstrated a significant decrease of GP2A-IgA titres upon a GFD and increased levels in RCD patients as compared to patients on a GFD. Although GP2A-IgA was not associated with the degree of villous atrophy, GP2A-IgA levels were able to distinguish RCD patients from GFD patients (ROC AUC=0.79, p=0.002).Conclusion: Follow-up of GP2A-IgA titres in CD patients on a GFD may help to identify patients at risk for developing RCD.Abbreviations: ABSA: anti-bovine serum albumine; ASCA: anti- Saccharomyces cerevisiae antigen; AU:artificial units; AUC: area under the curve; CD: celiac disease; CrD: Crohn's disease; EATL: enteropathyassociated T-cell lymphoma; EmA: anti-endomysium antibodies; GFD: gluten free diet; GP2A: glycoprotein 2 antibodies; IEL: intraepithelial lymphocytes; RCD: refractory celiac disease; ROC: receiver operator curve; TG2A: transglutaminase-2 antibodies; UC: ulcerative colitis.

How I treat enteropathy-associated T-cell lymphoma

Blood ◽  
2012 ◽  
Vol 119 (11) ◽  
pp. 2458-2468 ◽  
Author(s):  
Antonio Di Sabatino ◽  
Federico Biagi ◽  
Paolo G. Gobbi ◽  
Gino R. Corazza
Keyword(s):  
T Cell ◽  
Cell Lymphoma ◽  
High Dose Chemotherapy ◽  
T Cell Lymphoma ◽  
Gluten Free Diet ◽  
High Dose ◽  
Gluten Free ◽  
Strict Adherence ◽  
Positron Emission ◽  
Magnetic Resonance Enteroclysis

Abstract Enteropathy-associated T-cell lymphoma (EATL) is a complication of celiac disease (CD). This tumor derives from the neoplastic transformation of aberrant intraepithelial T lymphocytes emerging in celiac patients unresponsive to a gluten-free diet. Poor adherence to a gluten-free diet, HLA-DQ2 homozygosity, and late diagnosis of CD are recognized as risk factors for malignant evolution of CD. Recurrence of diarrhea, unexplained weight loss, abdominal pain, fever, and night sweating should alert physicians to this complication. The suspicion of EATL should lead to an extensive diagnostic workup in which magnetic resonance enteroclysis, positron emission tomography scan, and histologic identification of lesions represent the best options. Treatment includes high-dose chemotherapy preceded by surgical resection and followed by autologous stem cell transplantation, although biologic therapies seem to be promising. Strict adherence to a gluten-free diet remains the only way to prevent EATL.

Serendipity in Refractory Celiac Disease: Full Recovery of Duodenal Villi and Clinical Symptoms after Fecal Microbiota Transfer

2016 ◽  
Vol 25 (3) ◽  
pp. 385-388 ◽  
Author(s):  
Yvette H. Van Beurden ◽  
Tom Van Gils ◽  
Nienke A. Van Gils ◽  
Zain Kassam ◽  
Chris J.J. Mulder ◽  
...  
Keyword(s):  
Celiac Disease ◽  
T Cell ◽  
Clostridium Difficile ◽  
Clostridium Difficile Infection ◽  
Cell Lymphoma ◽  
Fecal Microbiota ◽  
Disease Type ◽  
T Cell Lymphoma ◽  
Type Ii ◽  
Refractory Celiac Disease

Treatment of refractory celiac disease type II (RCD II) and preventing the development of an enteropathy associated T-cell lymphoma in these patients is still difficult. In this case report, we describe a patient with RCD II who received fecal microbiota transfer as treatment for a recurrent Clostridium difficile infection, and remarkably showed a full recovery of duodenal villi and disappearance of celiac symptoms. This case suggests that altering the gut microbiota may hold promise in improving the clinical and histological consequences of celiac disease and/or RCD II. Abbreviations: CDI: Clostridium difficile infection; EATL : enteropathy associated T-cell lymphoma; FMT: fecal microbiota transfer; IEL: intraepithelial lymphocytes; RCD II: refractory celiac disease type II; TPN: total parenteral nutrition.

Enteropathy-associated T-cell lymphoma in a patient without a prior diagnosis of celiac disease: A diagnostic conundrum

2011 ◽  
Vol 48 (1) ◽  
pp. 124
Author(s):  
S Jacob ◽  
D Mohapatra ◽  
ML Nordberg ◽  
JD Cotelingam ◽  
S Upadhyay ◽  
...  
Keyword(s):  
Celiac Disease ◽  
T Cell ◽  
Cell Lymphoma ◽  
T Cell Lymphoma

scholarly journals Hematologic manifestations of celiac disease

Blood ◽  
2006 ◽  
Vol 109 (2) ◽  
pp. 412-421 ◽  
Author(s):  
Thorvardur R. Halfdanarson ◽  
Mark R. Litzow ◽  
Joseph A. Murray
Keyword(s):  
Celiac Disease ◽  
T Cell ◽  
Cell Lymphoma ◽  
B Cell Lymphoma ◽  
Iga Deficiency ◽  
Gluten Free ◽  
Strict Adherence ◽  
Increased Risk ◽  
Systemic Disorder ◽  
Iron Folic Acid

AbstractCeliac disease is a common systemic disorder that can have multiple hematologic manifestations. Patients with celiac disease may present to hematologists for evaluation of various hematologic problems prior to receiving a diagnosis of celiac disease. Anemia secondary to malabsorption of iron, folic acid, and/or vitamin B12 is a common complication of celiac disease and many patients have anemia at the time of diagnosis. Celiac disease may also be associated with thrombocytosis, thrombocytopenia, leukopenia, venous thromboembolism, hyposplenism, and IgA deficiency. Patients with celiac disease are at increased risk of being diagnosed with lymphoma, especially of the T-cell type. The risk is highest for enteropathy-type T-cell lymphoma (ETL) and B-cell lymphoma of the gut, but extraintestinal lymphomas can also be seen. ETL is an aggressive disease with poor prognosis, but strict adherence to a gluten-free diet may prevent its occurrence.

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