Tran Audrey M, Jnah Amy J, De Castro Pretelt Mauricio J
College of Nursing, East Carolina University, Greenville, NC, USA
College of Nursing, East Carolina University, Greenville, NC, USA.
Neonatal Netw. 2025 Jun 1;44(3):159-166. doi: 10.1891/NN-2024-0052.
Joubert syndrome (JS) is a rare neurodevelopmental and multisystem ciliopathy that affects 1 in 80,000-10,0000 infants globally per year. Classic defects include hypoplasia of the cerebellar vermis, thickened cerebellar peduncles, and deepened interpeduncular fossa, which is regarded as a "molar tooth" sign. More than 30 genes have been associated with JS. Gene mutations disrupt normal ciliary development and function during early embryogenesis and cause a vast array of symptoms and pathologies. The purpose of this article is to investigate this rare and complex medical condition. We present up-to-date information on the genetics, pathophysiology, clinical manifestations, diagnostics, and management of JS. We also include a valuable perspective from a pediatric geneticist who specializes in JS to enhance our understanding of challenges associated with making a molecular diagnosis and emphasize the importance of early recognition and diagnostics as a means to optimize the quality of care.
乔伯特综合征(JS)是一种罕见的神经发育和多系统纤毛病,全球每年每80000至100000名婴儿中就有1人受其影响。典型缺陷包括小脑蚓部发育不全、小脑脚增粗以及脚间窝加深,这被视为“磨牙”征。超过30个基因与JS相关。基因突变会破坏胚胎早期正常的纤毛发育和功能,并导致一系列症状和病理变化。本文旨在研究这种罕见且复杂的病症。我们提供了关于JS的遗传学、病理生理学、临床表现、诊断和管理的最新信息。我们还纳入了一位专门研究JS的儿科遗传学家的宝贵观点,以加深我们对分子诊断相关挑战的理解,并强调早期识别和诊断作为优化护理质量手段的重要性。