Lederer David J, Kawut Steven M, Sonett Joshua R, Vakiani Efsevia, Seward Samuel L, White James G, Wilt Jessie S, Marboe Charles C, Gahl William A, Arcasoy Selim M
Division of Pulmonary, Allergy and Critical Care Medicine, Department of Medicine, Columbia University College of Physicians and Surgeons, New York, New York 10032, USA.
J Heart Lung Transplant. 2005 Oct;24(10):1697-9. doi: 10.1016/j.healun.2004.11.015.
Hermansky-Pudlak syndrome (HPS) is a genetic disorder characterized by oculocutaneous albinism, a bleeding diathesis, and in a subset of patients, pulmonary fibrosis. Lung transplantation, the only curative therapy for pulmonary fibrosis, has not been previously reported as a successful treatment strategy for patients with HPS because the bleeding diathesis was thought to contraindicate major thoracic surgery. We successfully performed bilateral sequential lung transplantation in a patient with pulmonary fibrosis and HPS after transfusion of 6 units of platelets. Lung transplantation is a viable therapeutic option in patients with pulmonary fibrosis and only a mild bleeding diathesis associated with HPS.
赫尔曼斯基-普德拉克综合征(HPS)是一种遗传性疾病,其特征为眼皮肤白化病、出血素质,部分患者还伴有肺纤维化。肺移植是治疗肺纤维化的唯一根治性疗法,此前尚未有报道称其对HPS患者是一种成功的治疗策略,因为出血素质被认为是胸外科大手术的禁忌证。我们在为一名患有肺纤维化和HPS的患者输注6单位血小板后,成功实施了双侧序贯肺移植。对于患有肺纤维化且仅伴有与HPS相关的轻度出血素质的患者,肺移植是一种可行的治疗选择。