Chin A C, Stich D, White F V, Radhakrishnan J, Holterman M J
Division of Pediatric Surgery, Department of Surgery, The University of Illinois College of Medicine at Chicago, Chicago IL, USA.
J Pediatr Surg. 2001 Dec;36(12):E22. doi: 10.1053/jpsu.2001.28877.
Castleman's disease is an infrequent and usually benign lymphoproliferative disorder. Resection of the tumor usually is curative. The immunostimulatory nature of the tumor can, in rare instances, result in paraneoplastic manifestations. The authors present a case of a 14 year old with mucocutaneous ulcerations and progressive dyspnea that was found to have a large mediastinal mass and circulating autoantibodies that were responsible for his paraneoplastic pemphigus and bronchiolitis obliterans. In spite of aggressive immunotherapy to control the autoimmune mucocutaneous lesions, the pulmonary fibrosis was irreversible and progressed to pulmonary failure necessitating lung transplantation. J Pediatr Surg 36:E22.
卡斯尔曼病是一种罕见且通常为良性的淋巴增生性疾病。肿瘤切除通常可治愈。肿瘤的免疫刺激特性在极少数情况下可导致副肿瘤表现。作者报告了一例14岁患者,该患者有皮肤黏膜溃疡和进行性呼吸困难,发现有一个巨大的纵隔肿块以及循环自身抗体,这些自身抗体导致了他的副肿瘤性天疱疮和闭塞性细支气管炎。尽管积极进行免疫治疗以控制自身免疫性皮肤黏膜病变,但肺纤维化是不可逆的,并进展为呼吸衰竭,需要进行肺移植。《小儿外科杂志》36卷:E22。