Medical Molecular Genetics Department, Human Genetics and Genome Research Institute, National Research Centre, Cairo, Egypt.
Biochemistry and Molecular Biology Department, Faculty of Pharmacy (Girls), Al-Azhar University, Cairo, Egypt.
J Mol Neurosci. 2023 Aug;73(7-8):598-607. doi: 10.1007/s12031-023-02139-6. Epub 2023 Jul 20.
Lysosomal acid lipase (LAL) is a necessary enzyme for the hydrolysis of both triglycerides (TGs) and cholesteryl esters (CEs) in the lysosome. Deficiency of this enzyme encoded by the lipase A (LIPA) gene leads to LAL deficiency (LAL-D). A severe disease subtype of LAL-D is known as Wolman disease (WD), present with diarrhea, hepatosplenomegaly, and adrenal calcification. Untreated patients do not survive more than a year. The aim of this study was to assess the clinical and molecular characterizations of WD patients in Egypt. A total of seven patients (from five unrelated Egyptian families) were screened by targeted next-generation sequencing (NGS), and the co-segregation of causative variants was analyzed using Sanger sequencing. Furthermore, multiple in silico analyses were performed to assess the pathogenicity of the candidate variants. Overall, we identified three diseases causing variants harbored in the LIPA gene. One of these variants is a novel missense variant (NM_000235.4: c.1122 T > G; p. His374Gln), which was classified as a likely pathogenic variant. All variants were predicted to be disease causing using in silico analyses. Our findings expand the spectrum of variants involved in WD which may help to investigate phenotype-genotype correlation and assist genetic counseling. To the best of our knowledge, this is the first clinico-genetic study carried out on Egyptian patients affected with WD.
溶酶体酸性脂肪酶 (LAL) 是溶酶体中甘油三酯 (TGs) 和胆固醇酯 (CEs) 水解所必需的酶。该酶由脂肪酶 A (LIPA) 基因编码,如果该基因发生缺陷,就会导致 LAL 缺乏症 (LAL-D)。LAL-D 的一种严重疾病亚型被称为沃曼病 (WD),其特征是腹泻、肝脾肿大和肾上腺钙化。未经治疗的患者活不过一年。本研究旨在评估埃及 WD 患者的临床和分子特征。对 7 名患者(来自五个不相关的埃及家庭)进行了靶向下一代测序 (NGS) 筛查,并通过 Sanger 测序分析了致病变异的共分离情况。此外,还进行了多种计算机分析来评估候选变异的致病性。总的来说,我们在 LIPA 基因中发现了三个导致疾病的变异。其中一个变异是一种新的错义变异(NM_000235.4: c.1122 T>G; p. His374Gln),被归类为可能的致病变异。所有变异都通过计算机分析预测为致病变异。我们的发现扩展了 WD 中涉及的变异谱,这可能有助于研究表型-基因型相关性并协助遗传咨询。据我们所知,这是对埃及 WD 患者进行的首次临床遗传学研究。