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西班牙真实世界环境中慢性神经病变型戈谢病患者的临床结局:一项回顾性研究。

Clinical Outcomes of Patients with Chronic Neuropathic Form of Gaucher Disease in the Spanish Real-World Setting: A Retrospective Study.

作者信息

Stanescu Sinziana, Correcher Medina Patricia, Del Castillo Francisco J, Alonso Luengo Olga, Arto Millan Luis Maria, Belanger Quintana Amaya, Camprodon Gomez Maria, Diez Langhetée Lydia, Garcia Campos Oscar, Matas Garcia Ana, Perez-Moreno Jimena, Rubio Gribble Barbara, Visa-Reñé Nuria, Giraldo-Castellano Pilar, O'Callaghan Gordo Mar

机构信息

Pediatric Metabolic Unit, Hospital Universitario Ramón y Cajal, European Reference Center (MetabERN), 28034 Madrid, Spain.

Pediatric Nutrition and Metabolic Unit, Hospital Universitario y Politécnico La Fe, 46026 Valencia, Spain.

出版信息

Biomedicines. 2023 Oct 22;11(10):2861. doi: 10.3390/biomedicines11102861.

Abstract

This was a retrospective, multicenter study that aimed to report the characteristics of type 3 Gaucher disease (GD3) patients in Spain, including the genotype, phenotype, therapeutic options, and treatment responses. A total of 19 patients with GD3 from 10 Spanish hospitals were enrolled in the study (14 men, 5 women). The median age at disease onset and diagnosis was 1 and 1.2 years, respectively, and the mean age at follow-up completion was 12.37 years (range: 1-25 years). Most patients exhibited splenomegaly (18/19) and hepatomegaly (17/19) at the time of diagnosis. The most frequent neurological abnormalities at onset were psychomotor retardation (14/19) and extrinsic muscle disorders (11/19), including oculomotor apraxia, supranuclear palsy, and strabismus. The L444P (c.1448T>C) allele was predominant, with the L444P (c.1448T>C) homozygous genotype mainly associated with visceral manifestations like hepatosplenomegaly, anemia, and thrombocytopenia. All patients received enzyme replacement therapy (ERT); other treatments included miglustat and the chaperone (ambroxol). Visceral manifestations, including hepatosplenomegaly and hematological and bone manifestations, were mostly controlled with ERT, except for kyphosis. The data from this study may help to increase the evidence base on this rare disease and contribute to improving the clinical management of GD3 patients.

摘要

这是一项回顾性多中心研究,旨在报告西班牙3型戈谢病(GD3)患者的特征,包括基因型、表型、治疗选择和治疗反应。来自西班牙10家医院的19例GD3患者纳入本研究(14例男性,5例女性)。疾病发病和诊断的中位年龄分别为1岁和1.2岁,随访结束时的平均年龄为12.37岁(范围:1 - 25岁)。大多数患者在诊断时表现为脾肿大(18/19)和肝肿大(17/19)。发病时最常见的神经学异常是精神运动发育迟缓(14/19)和外部肌肉疾病(11/19),包括动眼失用、核上性麻痹和斜视。L444P(c.1448T>C)等位基因占主导,L444P(c.1448T>C)纯合基因型主要与肝脾肿大、贫血和血小板减少等内脏表现相关。所有患者均接受了酶替代疗法(ERT);其他治疗包括米格列醇和伴侣分子(氨溴索)。除脊柱后凸外,包括肝脾肿大以及血液和骨骼表现在内的内脏表现大多通过ERT得到控制。本研究的数据可能有助于增加关于这种罕见疾病的证据基础,并有助于改善GD3患者的临床管理。

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[Clinical phenotype and genotype of Gaucher disease in 14 children].14例儿童戈谢病的临床表型与基因型
Zhonghua Er Ke Za Zhi. 2022 Jun 2;60(6):527-532. doi: 10.3760/cma.j.cn112140-20220228-00159.

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