Geramizadeh Bita, Ezgu Fatih, Beyzaei Zahra
Department of Pathology, Medical School of Shiraz University, Shiraz University of Medical Sciences, Shiraz, Iran.
Department of Pediatric Metabolism and Genetics, Faculty of Medicine, Gazi University, Ankara, Turkey.
Orphanet J Rare Dis. 2024 Dec 20;19(1):475. doi: 10.1186/s13023-024-03488-0.
Glycogen storage disorders (GSD) GSD-IX are characterized by deficiencies in muscular and/or hepatic phosphorylase enzymes. GSD type IX za is an X-linked disorder, while IXb and IXc are autosomal recessive disorders resulting from pathogenic variants in the genes encoding the Phosphorylase b Kinase regulatory subunit alpha (PHKA), beta (PHKB), and gamma (PHKG), respectively. Despite progress in understanding these diseases, there are still unclear questions regarding their clinical manifestations, genetic variations, and the relationship between genotype and phenotype. Therefore, this review focuses on variants of GSD IX subtypes and all clinical findings to establish a genotype-phenotype relationship as well as highlighting the wide spectrum of disease-causing variants. Such information is beneficial for the establishment of a privileged mutation screening process in a specific region or ethnic group. Diagnosis is based on clinical manifestations and laboratory test results, but molecular analysis is often necessary to distinguish the various forms with similar presentations.
糖原贮积病(GSD)-IX的特征是肌肉和/或肝脏磷酸化酶缺乏。GSD-IXa是一种X连锁疾病,而IXb和IXc是常染色体隐性疾病,分别由编码磷酸化酶b激酶调节亚基α(PHKA)、β(PHKB)和γ(PHKG)的基因中的致病变异引起。尽管在了解这些疾病方面取得了进展,但关于它们的临床表现、基因变异以及基因型与表型之间的关系仍存在不清楚的问题。因此,本综述重点关注GSD-IX亚型的变异和所有临床发现,以建立基因型-表型关系,并突出致病变异的广泛谱系。此类信息有助于在特定地区或种族群体中建立优先的突变筛查流程。诊断基于临床表现和实验室检查结果,但通常需要进行分子分析以区分表现相似的各种形式。