Dao-Ping Sun1,2, Wen-Ming Chen3, Li Wang1,4, Zhen Wang5, Jin-Hua Liang1,4, Hua-Yuan Zhu1,4, Lei Fan1,4, Yu-Jie Wu1,4, Wei Xu6,7, Jian-Yong Li8,9. 1. Department of Hematology, The First Affiliated Hospital of Nanjing Medical University, Jiangsu Province Hospital, Nanjing, 210029, China. 2. Department of Hematology, Jining No. 1 People's Hospital, Jining, 272011, China. 3. Department of Oncology, Jining No. 1 People's Hospital, Jining, 272011, China. 4. Pukou CLL Center, Nanjing, 210000, China. 5. Department of Pathology, The First Affiliated Hospital of Nanjing Medical University, Jiangsu Province Hospital, Nanjing, 210029, China. 6. Department of Hematology, The First Affiliated Hospital of Nanjing Medical University, Jiangsu Province Hospital, Nanjing, 210029, China. xuwei10000@hotmail.com. 7. Pukou CLL Center, Nanjing, 210000, China. xuwei10000@hotmail.com. 8. Department of Hematology, The First Affiliated Hospital of Nanjing Medical University, Jiangsu Province Hospital, Nanjing, 210029, China. lijianyonglm@126.com. 9. Pukou CLL Center, Nanjing, 210000, China. lijianyonglm@126.com.
Abstract
PURPOSE: To explore the clinical features and immunological mechanisms of Castleman disease (CD) complicated with autoimmune diseases (AID). METHODS: We explored the prevalence and clinical manifestations of CD complicated with AID by reviewing clinical, pathological, and laboratory data of 40 CD patients retrospectively, and then explored abnormal immune mechanisms in the co-existence of the two entities by monitoring lymphocyte subsets in peripheral blood. RESULTS: Paraneoplastic pemphigus, autoimmune hemolytic anemia, Sjogren's syndrome, myasthenia gravis, and psoriasis were found to be coexisted with CD in 9/40 (22.5%) patients with different sequence of onset. No bias in the clinical and histological type of CD was observed for the occurrence of AID. CD patients with AID were more likely to have skin and/or mucous membrane damage and pulmonary complications, and presented elevated erythrocyte sedimentation rate, hypergammaglobulinemia, and positive autoantibodies than those without AID (p < 0.05). Deregulated cellular and innate immune responses as indicated by decreased CD3+ T cells and increased natural killer cells were observed in peripheral blood of CD patients with AID (p < 0.05). UCD patients with AID were successfully treated with surgery and immunosuppressive therapy. MCD complicated by AID relieved with immunosuppressors, cytotoxic chemotherapy, and rituximab. CONCLUSION: Systemic inflammation/immunological abnormalities and organ dysfunction were associated with the occurrence of AID in CD. Impairment of cellular and innate immunity may be a candidate etiology for the coexistence of the two entities.
PURPOSE: To explore the clinical features and immunological mechanisms of Castleman disease (CD) complicated with autoimmune diseases (AID). METHODS: We explored the prevalence and clinical manifestations of CD complicated with AID by reviewing clinical, pathological, and laboratory data of 40 CD patients retrospectively, and then explored abnormal immune mechanisms in the co-existence of the two entities by monitoring lymphocyte subsets in peripheral blood. RESULTS:Paraneoplastic pemphigus, autoimmune hemolytic anemia, Sjogren's syndrome, myasthenia gravis, and psoriasis were found to be coexisted with CD in 9/40 (22.5%) patients with different sequence of onset. No bias in the clinical and histological type of CD was observed for the occurrence of AID. CD patients with AID were more likely to have skin and/or mucous membrane damage and pulmonary complications, and presented elevated erythrocyte sedimentation rate, hypergammaglobulinemia, and positive autoantibodies than those without AID (p < 0.05). Deregulated cellular and innate immune responses as indicated by decreased CD3+ T cells and increased natural killer cells were observed in peripheral blood of CD patients with AID (p < 0.05). UCD patients with AID were successfully treated with surgery and immunosuppressive therapy. MCD complicated by AID relieved with immunosuppressors, cytotoxic chemotherapy, and rituximab. CONCLUSION: Systemic inflammation/immunological abnormalities and organ dysfunction were associated with the occurrence of AID in CD. Impairment of cellular and innate immunity may be a candidate etiology for the coexistence of the two entities.
Authors: Daniel Aletaha; Tuhina Neogi; Alan J Silman; Julia Funovits; David T Felson; Clifton O Bingham; Neal S Birnbaum; Gerd R Burmester; Vivian P Bykerk; Marc D Cohen; Bernard Combe; Karen H Costenbader; Maxime Dougados; Paul Emery; Gianfranco Ferraccioli; Johanna M W Hazes; Kathryn Hobbs; Tom W J Huizinga; Arthur Kavanaugh; Jonathan Kay; Tore K Kvien; Timothy Laing; Philip Mease; Henri A Ménard; Larry W Moreland; Raymond L Naden; Theodore Pincus; Josef S Smolen; Ewa Stanislawska-Biernat; Deborah Symmons; Paul P Tak; Katherine S Upchurch; Jirí Vencovsky; Frederick Wolfe; Gillian Hawker Journal: Ann Rheum Dis Date: 2010-09 Impact factor: 19.103
Authors: David C Fajgenbaum; Thomas S Uldrick; Adam Bagg; Dale Frank; David Wu; Gordan Srkalovic; David Simpson; Amy Y Liu; David Menke; Shanmuganathan Chandrakasan; Mary Jo Lechowicz; Raymond S M Wong; Sheila Pierson; Michele Paessler; Jean-François Rossi; Makoto Ide; Jason Ruth; Michael Croglio; Alexander Suarez; Vera Krymskaya; Amy Chadburn; Gisele Colleoni; Sunita Nasta; Raj Jayanthan; Christopher S Nabel; Corey Casper; Angela Dispenzieri; Alexander Fosså; Dermot Kelleher; Razelle Kurzrock; Peter Voorhees; Ahmet Dogan; Kazuyuki Yoshizaki; Frits van Rhee; Eric Oksenhendler; Elaine S Jaffe; Kojo S J Elenitoba-Johnson; Megan S Lim Journal: Blood Date: 2017-01-13 Impact factor: 22.113