Cetiner Metin, Bergmann Carsten, Bettendorf Markus, Faust Johanna, Gäckler Anja, Gillissen Bernarda, Hansen Matthias, Kerber Maximilian, Klaus Günter, König Jens, Kühlewein Laura, Oh Jun, Richter-Unruh Annette, von Schnurbein Julia, Wabitsch Martin, Weihrauch-Blüher Susann, Pape Lars
Department of Pediatrics II, University Hospital Essen, Essen, Germany.
Human genetic diagnostics, Medical Genetics Mainz, Mainz, Germany.
Klin Padiatr. 2024 Sep;236(5):269-279. doi: 10.1055/a-2251-5382. Epub 2024 Mar 8.
Bardet-Biedl syndrome (BBS) is a rare, autosomal recessive multisystem disease. The pathophysiological origin is a dysfunction of the primary cilium. Clinical symptoms are heterogeneous and variable: retinal dystrophy, obesity, polydactyly, kidney abnormalities, hypogenitalism and developmental delays are the most common features. By the approval of the melanocortin 4 receptor agonist setmelanotide, a drug therapy for BBS-associated hyperphagia and obesity can be offered for the first time. Hyperphagia and severe obesity represent a considerable burden and are associated with comorbidity and increased mortality risk. Due to the limited experience with setmelanotide in BBS, a viable comprehensive therapy concept is to be presented. Therapy decision and management should be conducted in expert centers. For best therapeutic effects with setmelanotide adequate information of the patient about the modalities of the therapy (daily subcutaneous injection) and possible adverse drug events are necessary. Furthermore, the involvement of psychologists, nutritionists and nursing services (support for the application) should be considered together with the patient. The assessment of therapy response should be carried out with suitable outcome measurements and centrally reported to an adequate register.
巴德-比德尔综合征(BBS)是一种罕见的常染色体隐性多系统疾病。其病理生理起源是初级纤毛功能障碍。临床症状具有异质性且多变:视网膜营养不良、肥胖、多指(趾)畸形、肾脏异常、生殖器发育不全和发育迟缓是最常见的特征。随着促黑素4受体激动剂setmelanotide获批,首次有了针对BBS相关的食欲亢进和肥胖的药物治疗方法。食欲亢进和重度肥胖带来了相当大的负担,且与合并症及死亡风险增加相关。鉴于在BBS患者中使用setmelanotide的经验有限,需要提出一个可行的综合治疗方案。治疗决策和管理应在专家中心进行。为了使setmelanotide达到最佳治疗效果,有必要让患者充分了解治疗方式(每日皮下注射)及可能的药物不良事件。此外,应与患者共同考虑让心理学家、营养师和护理服务人员参与进来(协助用药)。应采用合适的疗效评估指标对治疗反应进行评估,并集中上报至一个合适的登记处。