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Holmes-Adie syndrome, autoimmune hepatitis and celiac disease: A case report

Holmes-Adie syndrome, autoimmune hepatitis and celiac disease: A case report

作     者:Timea Csak Aniko Folhoffer Andrea Horvath Judit Halász Csaba Diczházi Zsuzsa Schaff Ferenc Szalay 

作者机构:Department of Medicine Semmelweis UniversityBudapestHungary Department of Pathology Semmelweis UniversityBudapestHungary 

出 版 物:《World Journal of Gastroenterology》 (世界胃肠病学杂志(英文版))

年 卷 期:2006年第12卷第9期

页      面:1485-1487页

核心收录:

学科分类:1002[医学-临床医学] 100201[医学-内科学(含:心血管病、血液病、呼吸系病、消化系病、内分泌与代谢病、肾病、风湿病、传染病)] 10[医学] 

主  题:Holmes-Adie syndrome Autoimmune hepatitis Celiac disease Mesenteric lymphangioma Autonomic neuropathy 

摘      要:A 35-year-old female patient presented with the following symptoms of Holmes-Adie syndrome: photophobia, enlargement of the left pupil unresponsive to light, Achilles areflexia. The pilocarpine test was positive. No tumor or other neurological abnormality was found. She had a 19-year history of autoimmune hepatitis. Flares up were observed following each 3 deliveries. At age of 31 she presented with diarrhea and weight loss. Abdominal tumor was detected by ultrasound. The surgically removed tumor was histologically a benign mesenteric multicystic lymphangioma. Simultaneously, celiac disease was diagnosed. Gluten-free diet resulted in a significant improvement of celiac disease, but not of autoimmune hepatitis. Autonomic neuropathy was proven by standard cardiovascular tests. The patient was a homozygous carrier for HLA DQ2 antigen characteristic for celiac disease and heterozygous for HLA DR3 B8 frequent in autoimmune liver diseases. Our novel observation on association of Holmes-Adie syndrome with autoimmune hepatitis and celiac disease is suggestive for a common immunological background for all three entities present in a patient with mesenteric multicystic lymphangioma.

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