Galea Michela, Cocker Miriana, Soler Doriette, Bezzina Esther
Paediatrics, Mater Dei Hospital, Msida, MLT.
Radiology, Mater Dei Hospital, Msida, MLT.
Cureus. 2025 Apr 15;17(4):e82308. doi: 10.7759/cureus.82308. eCollection 2025 Apr.
Pelizaeus-Merzbacher disease (PMD) is a rare X-linked hypomyelinating leukodystrophy disorder caused by a mutation in the proteolipid protein 1 (PLP1) gene which is responsible for myelin formation in the central nervous system. We report a case of PMD in a male patient who initially presented with developmental delay at three months and was ultimately diagnosed at 10 years and 7 months of age. We aim to describe the initial presentation, clinical course of PMD and the investigations that aid in diagnosis so that future cases may be identified earlier. During the investigative workup of our patient, a deletion of exon 16 - an extremely rare heterozygous nucleotide variation, c.330C>T (p.D110D) - was identified, the pathogenicity of which has not been previously documented in the literature. In our report, we also aim to highlight the potential use of biotin in improving symptoms in such patients as there is currently no curative treatment available.
佩利措伊斯-梅茨巴赫病(PMD)是一种罕见的X连锁髓鞘形成不良性脑白质营养不良症,由蛋白脂蛋白1(PLP1)基因的突变引起,该基因负责中枢神经系统的髓鞘形成。我们报告了一例PMD男性患者,该患者最初在三个月大时出现发育迟缓,最终在10岁7个月时被确诊。我们旨在描述PMD的初始表现、临床病程以及有助于诊断的检查,以便未来能更早地识别病例。在对我们的患者进行调查评估期间,发现了外显子16的缺失——一种极其罕见的杂合核苷酸变异,即c.330C>T(p.D110D),其致病性此前在文献中尚未有记载。在我们的报告中,我们还旨在强调生物素在改善此类患者症状方面的潜在用途,因为目前尚无治愈性治疗方法。