A rare complication of gluten enteropathy diagnosed with surgery: Enteropathy associated T-cell lymphoma

Year 2012, Volume: 20 Issue: 1, - , 01.04.2012

İlhan Dolaşık Hakan Uzunoğlu Selman Çınar Selçuk Yusuf Şener Mehmet Zeki Aydın Koray Çelebi Ömer Şentürk Sadettin Hülagu

https://doi.org/10.17940/endoskopi.74844

Abstract

One of the well-known complications of gluten enteropathy in the advanced stage is lymphoma-associated enteropathy. In the treatment of the disease and prevention of lymphoma development, gluten-containing foods must be removed from the diet on a lifelong basis. In the majority of patients, clinical improvement is seen and intestinal mucosal architecture normalizes after a gluten-free diet. Refractory disease is considered in a small number of patients without recovery; underlying jejunoileitis (ulcerative jejunitis) or lymphoma may exist. Accordingly, we want to report our case with gluten enteropathy who initially responded to the diet but later become refractory. In this case, we detected a jejunal mass with double balloon enteroscopy, and enteropathyassociated T-cell lymphoma was diagnosed after surgical excision. This case showed that neoplastic cells may not be defined in biopsies taken under the impact of inflammatory changes, especially in the presence of ulcer and erosions, even though a malignant-looking mass lesion was detected in endoscopic examinations. As in this case, both diagnosis and treatment can be achieved with surgery in patients with strongly suspected malignancy.

Keywords

Gluten enteropathy, T-cell lymphoma, endoscopy

Gluten enteropatisinin nadir ve cerrahi ile tanı konan komplikasyonu: Enteropati ilişkili T-hücreli lenfoma

Abstract

Gluten enteropatisinin ileri dönemdeki iyi bilinen komplikasyonlarından biri enteropati ilişkili lenfomadır. Hastalığın tedavisinde ve lenfoma gelişiminden korumada, gluten içeren gıdaların, ömür boyunca diyetten uzaklaştırılmaları gerekir. Glutensiz diyet sonrası, hastaların çoğunda klinik düzelme görülür ve intestinal mukozal yapı normale döner. Düzelmeyen az sayıdaki hastada, refrakter hastalık düşünülür; altta yatan bir jejunoileitis (ülseratif jejunit) veya lenfoma olabilir. Bu nedenle biz de gluten enteropatisi tanısı konan ve diyete önce cevap verip sonrasında refrakter olan, çift balon enteroskopide jejunal kitle olduğunu saptadığımız ve cerrahi rezeksiyon sonrası enteropati ilişkili T hücreli lenfoma tespit ettiğimiz olgumuzu sunmak istedik. Bu vaka bize endoskopik tetkiklerle malign görünümlü kitle lezyonu tespit edilse dahi özellikle ülser ve erozyonların varlığında inflamatuvar değişikliklerin etkisi ile alınan biyopsilerde neoplastik hücrelerin tanımlanamayabileceğini göstermiştir. Bu vakada olduğu gibi şiddetle malignite düşünülen vakalarda cerrahi ile hem tanı konabilir hem de tedavi sağlanabilir.

Keywords

Gluten enteropatisi, T hücreli lenfoma, endoskopi

References

• American Gastroenterological Association medical position statement: Coeliac sprue. Gastroenterology 2001;120:1522-5.
• Leeds JS, Hopper AD, Sanders DS. Coeliac disease. Br Med Bull 2008;88: 157-70.
• Solaymani-Dodaran M, West J, Logan RF. Long-term mortality in people with celiac disease diagnosed in childhood compared with adulthood: a population- based cohort study. Am J Gastroenterol 2007;102:864-70.
• Peters U, Askling J, Gridley G, et al. Causes of death in patients with coeliac disease in a population-based Swedish cohort. Arch Intern Med 2003;163:1566-72.
• Freeman HJ. Neoplastic disorders in 100 patients with adult celiac disease. Can J Gastroenterol 1996;10:163-6.
• Daum S, Cellier C, Mulder CJ. Refractory coeliac disease. Best Pract Res Clin Gastroenterol 2005;19:413-24.
• Cellier C, Delabesse E, Helmer C, et al. Refractory sprue, coeliac disease, and enteropathyassociated T-cell lymphoma. French Coeliac Disease Study Group. Lancet 2000;356:203-8.
• Patey-Mariaud de Serre N, Cellier C, Jabri B, et all. Distinction between coeliac disease and refractory sprue: a simple immunohistochemical method. Histopathology 2000;37:70-7.
• Rodrigo L. Celiac disease. World J Gastroenterol 2007;12:6585-93.
• Brousse N, Meijer JW. Malignant complications of coeliac disease. Best Pract Res Clin Gastroenterol 2005;19:401-12.
• Catassi C, Bearzi I, Holmes GK. Association of celiac disease and intestinal lymphomas and other cancers. Gastroenterology 2005;128(4 Suppl 1):S79-86.
• Freeman HJ. Fulminant liver failure with necrotizing foci in the liver, spleen and lymph nodes in celiac disease due to malignant lymphoma. Can J Gastroenterol 1996;10:225-9.
• Holmes GK. Non-malignant complications of coeliac disease. Acta Paediatr Suppl 1996;412:68-75.
• Van Weyenberg SJB, Jarbandhan SVA, Mulder CJJ, et al. Double Balloon Endoscopy in Celiac Disease. Tech Gastrointest Endosc 2008;10:87-93.
• Freeman HJ, Weinstein WM, Shnitka TK, et al. Primary abdominal lymphoma. Presenting manifestation of celiac sprue or complicating dermatitis herpetiformis. Am J Med 1977;63:585-94.
• Egan LJ, Walsh SV, Stevens FM, et al. Celiac-associated lymphoma. A single institution experience of 30 cases in the combination chemotherapy era. J Clin Gastroenterol 1995;21:123-9.