Division of Pediatric Endocrinology, Vanderbilt University Medical Center, Nashville, Tennessee, USA.
Diabetes Obes Metab. 2024 Apr;26 Suppl 2:25-33. doi: 10.1111/dom.15494. Epub 2024 Feb 21.
Bardet-Biedl syndrome (BBS) is a genetic disorder characterized by early-onset obesity, polydactyly, genital and kidney anomalies, developmental delay and vision loss due to rod-cone dystrophy. BBS is an autosomal recessive disorder with >20 implicated genes. The genotype-phenotype relationship in BBS is not clear, and there may be additional modifying factors. The underlying mechanism is dysfunction of primary cilia. In BBS, receptor trafficking in and out of the cilia is compromised, affecting multiple organ systems. Along with early-onset obesity, hyperphagia is a prominent symptom and contributes significantly to clinical morbidity and caregiver burden. While there is no cure for BBS, setmelanotide is a new pharmacotherapy approved for treatment of obesity in BBS. The differential diagnosis for BBS includes other ciliopathies, such as Alstrom syndrome, and other genetic obesity syndromes, such as Prader-Willi syndrome. Careful clinical history and genetic testing can help determine the diagnosis and a multidisciplinary team is necessary to guide clinical management.
Bardet-Biedl 综合征(BBS)是一种遗传疾病,其特征为发病早的肥胖、多指(趾)畸形、生殖器和肾脏异常、发育迟缓以及因 rods-cone 营养不良导致的视力丧失。BBS 是一种常染色体隐性遗传疾病,涉及 >20 个相关基因。BBS 的基因型-表型关系尚不清楚,可能存在其他修饰因素。其潜在机制是初级纤毛功能障碍。在 BBS 中,纤毛内外的受体运输受损,影响多个器官系统。除了发病早的肥胖外,食欲过盛也是一个突出的症状,并显著增加临床发病率和照护者负担。虽然目前尚无治愈 BBS 的方法,但 setmelanotide 是一种新的药物疗法,被批准用于治疗 BBS 引起的肥胖。BBS 的鉴别诊断包括其他纤毛病,如 Alstrom 综合征,以及其他遗传肥胖综合征,如 Prader-Willi 综合征。仔细的临床病史和基因检测有助于确定诊断,多学科团队的参与对于指导临床管理是必要的。