Alsahlawi Zahra, Isa Hasan M, Alresias Sulaiman, Hasan Sayed Mohamed, Malalla Husain A, Ebrahim Ayman K, Ali Khadija
Department of Pediatrics, Arabian Gulf University, Manama, BHR.
Department of Pediatrics, Salmaniya Medical Complex, Manama, BHR.
Cureus. 2024 Nov 14;16(11):e73667. doi: 10.7759/cureus.73667. eCollection 2024 Nov.
Vanishing white matter (VWM) disease is an autosomal recessive disorder caused by mutations in the gene EIF2B encoding the subunits 1-5 of eukaryotic initiation factor 2B. Although rare, with a reported prevalence of 1:80,000 (0.001%), it was considered as one of the most common leukodystrophies. However, the worldwide incidence and prevalence of this disease are not clear. In Bahrain, of 21 patients who were diagnosed with leukodystrophy, two patients were found to have VWM disease accounting for 9.5%. Vaccinations and infections were the trigger factors for this disease to manifest. Rapid neurological deterioration, loss of developmental milestones, and seizure disorders are the main presentations in both patients. Magnetic resonance imaging (MRI) showed the classical radiological changes of demyelination and leukodystrophy. Patient 1 had associated ulcerative colitis, a finding that was not reported before. Patient 1's condition progressed to a vegetative stage, while patient 2 passed away, reflecting the poor disease outcome. In patient 2, a novel homozygous missense mutation was found in the EIF2B3 gene (c.25G>A, p.Ala9Thr). In this report, we present in detail the prevalence of VWM disease among cases with leukodystrophy, patients' characteristics, clinical presentations, radiological findings, associated diseases, genetic results, and clinical outcomes in the main tertiary hospital in Bahrain between 1998 and 2024. Moreover, we conducted a thorough literature review on this rare condition.
消失性白质(VWM)病是一种常染色体隐性疾病,由编码真核起始因子2B亚基1-5的EIF2B基因突变引起。尽管罕见,报道的患病率为1:80,000(0.001%),但它曾被认为是最常见的脑白质营养不良症之一。然而,这种疾病在全球的发病率和患病率尚不清楚。在巴林,21例被诊断为脑白质营养不良症的患者中,有2例被发现患有VWM病,占9.5%。疫苗接种和感染是该疾病显现的触发因素。快速的神经功能恶化、发育里程碑丧失和癫痫障碍是这两名患者的主要表现。磁共振成像(MRI)显示了脱髓鞘和脑白质营养不良的典型影像学变化。患者1伴有溃疡性结肠炎,这一发现此前未见报道。患者1的病情进展至植物人状态,而患者2去世,反映出该疾病预后不佳。在患者2中,在EIF2B3基因中发现了一个新的纯合错义突变(c.25G>A,p.Ala9Thr)。在本报告中,我们详细介绍了1998年至2024年期间巴林主要三级医院中患有脑白质营养不良症的病例中VWM病的患病率、患者特征、临床表现、影像学检查结果、相关疾病、基因检测结果及临床结局。此外,我们对这种罕见疾病进行了全面的文献综述。