Toni Ledjona, Dušátková Petra, Novotná Dana, Zemková Daniela, Průhová Štěpánka, Lebl Jan
Department of Paediatrics, 2nd Faculty of Medicine, Charles University and University Hospital Motol, Praha, Czech Republic.
Department of Paediatrics, Faculty of Medicine, Masaryk University and University Hospital Brno, Brno, Czech Republic.
J Pediatr Endocrinol Metab. 2019 Jul 26;32(7):775-779. doi: 10.1515/jpem-2019-0107.
Background Hutchinson-Gilford progeria syndrome (HGPS) is a rare disease caused by pathogenic variants in the LMNA gene, which leads to premature aging. The median life expectancy is shortened to 13 years due to cardiovascular complications. Case report We present a boy born with a pathogenic LMNA variant c.433G > A, which causes atypical progeria syndrome (APS) and was previously described in one single patient. When investigated for poor growth prior to the diagnosis of APS, his laboratory tests revealed growth hormone (GH) deficiency and magnetic resonance imaging (MRI) of the midbrain showed partial empty sella. GH treatment had only a limited and transient effect. His first ischemic complication manifested at age 4.2 years; at the age of 7 years, he had a fatal haemorrhagic stroke. Conclusion To the best of our knowledge, this is the first patient with APS showing partial empty sella and GH deficiency that might have contributed to his poor growth. GH failed to improve long-term outcome.
哈钦森-吉尔福德早衰综合征(HGPS)是一种由LMNA基因突变导致的罕见疾病,会引发早衰。由于心血管并发症,患者的中位预期寿命缩短至13岁。病例报告:我们报告了一名出生时携带致病性LMNA变异c.433G > A的男孩,该变异导致非典型早衰综合征(APS),此前仅在一名患者中被描述过。在诊断APS之前,因其生长发育迟缓进行检查时,他的实验室检查显示生长激素(GH)缺乏,中脑磁共振成像(MRI)显示部分空蝶鞍。GH治疗仅产生了有限的短暂效果。他的首次缺血性并发症出现在4.2岁;7岁时,他死于出血性中风。结论:据我们所知,这是首例患有APS且表现出部分空蝶鞍和GH缺乏的患者,这可能是导致他生长发育迟缓的原因。GH未能改善长期预后。