Division of Thoracic Surgery, Department of Surgery, National Taiwan University College of Medicine, Taipei, Taiwan.
J Formos Med Assoc. 2010 May;109(5):397-400. doi: 10.1016/S0929-6646(10)60069-1.
Proteus syndrome is a very rare, sporadic and congenital condition that is characterized by postnatal mosaic overgrowth. This disorder is thought to be caused by a somatic gene mutation, but the exact etiology is unknown. Commonly involved tissues include connective tissue, bone, skin and the central nervous system. Another less common symptom involves pulmonary emphysematous changes. This report documents a 25-year-old man with Proteus syndrome who presented with progressive exertional dyspnea and asymmetric overgrowth of his extremities. He underwent left pneumonectomy and his postoperative course was uneventful. Lung tissue showed emphysematous changes with multiple bulla formation and scattered calcification. We also review recent literature related to pulmonary manifestations and management of Proteus syndrome.
先天性红细胞生成性卟啉病是一种非常罕见的、散发性的、先天性疾病,其特征为出生后嵌合体过度生长。这种疾病被认为是由体细胞基因突变引起的,但确切的病因尚不清楚。常见受累组织包括结缔组织、骨骼、皮肤和中枢神经系统。另一个不太常见的症状涉及肺气肿改变。本报告记录了一例 25 岁男性患有 Proteus 综合征,表现为进行性运动性呼吸困难和四肢不对称过度生长。他接受了左肺切除术,术后过程顺利。肺组织显示出肺气肿改变,伴有多个大疱形成和散在钙化。我们还回顾了与 Proteus 综合征的肺部表现和治疗相关的最新文献。