A case of Adult-onset Cyclic Neutropenia Presenting with Arthritis
의약학 > 내과학
( Myung Jin Lee ) , ( Jong Jin Yoo ) , ( Eun Ha Kang ) , ( Eun Young Lee ) , ( Kichul Shin ) , ( Eun Bong Lee ) , ( Yeong Wook Song ) , ( Yun Jong Lee )
대한내과학회 추계학술발표논문집 2011년, 제2011권 제1호, 337(총1페이지)
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    Cyclic neutropenia is a rare disorder characterized by repetitive episodes of neutropenia every 14 to 28 days. During the neutropenic periods, patients may have fever, recurrent aphthous stomatitis, and occasionally infections. Usually, the disease begins in childhood and a genetic mutation in the elastase gene (ELANE) has been reported. However, adult-onset cyclic neutropenia is considered an autoimmune disorder in association with proliferation of cytotoxic T cells. We report here a case of 22-year old man with adult-onset cyclic neutropenia presenting with fever and polyarthritis. He had periodic neutropenia with intervals of about 21 days and his arthritis symptoms were aggravated simultaneously with the neutropenic episodes. Additionally, he had oral ulcer, abdominal pain, or fever in the neutropenic period. Bone marrow study, peripheral blood smear, peripheral lymphocyte subset analysis, and ELANE sequencing did not show any abnormal findings. Initial trial of cyclosporine had no effect on his cyclic neutropenia episodes. After regular administration of G-CSF subcutaneous injection, the duration of neutropenia was shortened and his arthritis was improved. To the best of our knowledge, this is the first case of adult-onset cyclic neutropenia in Korea.
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