St John's Institute of Dermatology, London, UK.
Australas J Dermatol. 2012 May;53(2):151-4. doi: 10.1111/j.1440-0960.2011.00738.x. Epub 2011 Mar 10.
Birt-Hogg-Dubé syndrome (BHDS) is an autosomal dominant condition caused by mutations in the gene which codes for folliculin (FLCN). It is characterised clinically by fibrofolliculomas, trichodiscomas, pulmonary cysts, spontaneous pneumothoraces and renal cancers. This case illustrates a patient with BHDS and a renal angiomyolipoma. Angiomyolipomas are not described as a feature of BHDS, but rather they can occur sporadically or in tuberous sclerosis complex (TSC). Recent studies suggest that clinical similarities between BHDS and TSC may be explained by FLCN and TSC proteins functioning on a common pathway, mammalian target of rapamycin. This case adds to the literature of cases with clinical similarities.
Birt-Hogg-Dubé 综合征(BHDS)是一种常染色体显性遗传疾病,由编码滤泡素(FLCN)的基因突变引起。其临床特征为纤维毛囊瘤、毳毛囊肿、肺囊肿、自发性气胸和肾癌。本病例说明了一位 BHDS 合并肾血管平滑肌脂肪瘤患者。血管平滑肌脂肪瘤并非 BHDS 的特征性表现,而是可散发性或在结节性硬化症复合征(TSC)中发生。最近的研究表明,BHDS 和 TSC 之间的临床相似性可能是由 FLCN 和 TSC 蛋白在共同途径——哺乳动物雷帕霉素靶蛋白上发挥作用所解释。本病例增加了具有临床相似性的病例文献。