Division of Neurology, Department of Pediatrics, The Hospital for Sick Children, University of Toronto, 555 University Avenue, Toronto, ON M5G 1X8, Canada; Department of Pediatrics, King Faisal Specialist Hospital & Research Centre, Jeddah, Saudi Arabia.
Division of Clinical and Metabolic Genetics, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada; Fred A Litwin Family Centre in Genetic Medicine, University Health Network and Mount Sinai Hospital, Toronto, Ontario, Canada.
Neuromuscul Disord. 2022 Oct;32(10):842-844. doi: 10.1016/j.nmd.2022.09.007. Epub 2022 Sep 22.
PURA syndrome is caused by heterozygous de novo pathogenic variants in PURA. It is characterized by moderate to severe neurodevelopmental disability with a wide clinical spectrum and an evolving phenotype. We present two individuals with genetically confirmed PURA syndrome who had severe neonatal signs and symptoms and a novel phenotype suggestive of neuromuscular junction pathology. We demonstrate that PURA syndrome shares features consistent with a congenital myasthenic syndrome; we thus recommend electrodiagnostic study in neonates and infants with PURA syndrome, and consideration of salbutamol as a therapeutic option.
PURA 综合征是由 PURA 中的杂合新生致病性变异引起的。其特征是具有广泛临床表现谱和不断发展的表型的中度至重度神经发育障碍。我们介绍了两名经基因证实患有 PURA 综合征的患者,他们具有严重的新生儿体征和症状,以及新的表型提示神经肌肉接头病理学。我们证明 PURA 综合征具有与先天性肌无力综合征一致的特征;因此,我们建议对 PURA 综合征的新生儿和婴儿进行电诊断研究,并考虑沙丁胺醇作为一种治疗选择。