Holmes-Adie syndrome, autoimmune hepatitis and celiac disease: a case report.
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.
1st Department of Medicine, Semmelweis University, Koranyi St. 2A, H-1083 Budapest, Hungary.
Timea
T
Csak
Aniko
A
Folhoffer
Andrea
A
Horvath
Judit
J
Halász
Csaba
C
Diczházi
Zsuzsa
Z
Schaff
Ferenc
F
Szalay
D000270:Adie Syndrome / Q000150:complications* / Q000175:diagnosis / Q000276:immunology; D000328:Adult; D002446:Celiac Disease / Q000150:complications* / Q000175:diagnosis / Q000276:immunology; D005260:Female; D019693:Hepatitis, Autoimmune / Q000150:complications* / Q000175:diagnosis / Q000276:immunology; D006801:Humans; D018191:Lymphangioma, Cystic / Q000150:complications / Q000175:diagnosis; D010534:Peritoneal Neoplasms / Q000150:complications / Q000175:diagnosis