Mobeireek Abdullah, Saleemi Sarfraz, Khalid Mohammad, Imtiaz Faiqa, Almutairy Eid A
Department of Medicine, King Faisal Hospital & Research Centre, Riyadh, Saudia Arabia.
Am J Med Genet A. 2015 Feb;167A(2):434-7. doi: 10.1002/ajmg.a.36858. Epub 2014 Nov 26.
Schimke Immuno-osseous Dysplasia (SIOD) is a rare genetic disorder with multiple systemic manifestations. Pulmonary manifestations have been described but not well characterized. They are believed to be secondary to decreased elasticity, and include emphysema, pulmonary hypertension and bronchiectasis. We describe a 24-year-old female patient with SIOD with tracheobronchial anomalies not reported before, including tracheomalacia and a tracheal bronchus with Epstein-Barr virus (EBV) related leiomyoma causing endobronchial obstruction. Such anomalies, in addition to the difficult upper airway associated with SIOD present specific challenges during management. This case reports documents tracheobronchial abnormalities that have not been described before in SIOD.
施姆克免疫性骨发育不良(SIOD)是一种罕见的具有多种全身表现的遗传性疾病。肺部表现已有描述,但特征尚不明确。它们被认为是弹性降低的继发表现,包括肺气肿、肺动脉高压和支气管扩张。我们描述了一名24岁患有SIOD的女性患者,其存在以前未报道过的气管支气管异常,包括气管软化和伴有与爱泼斯坦-巴尔病毒(EBV)相关平滑肌瘤的气管支气管,导致支气管内阻塞。除了与SIOD相关的困难上气道外,这些异常在管理过程中带来了特殊挑战。本病例报告记录了SIOD中以前未描述过的气管支气管异常。