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异染性脑白质营养不良患者的酸碱平衡异常及代谢性酸中毒倾向。

Abnormalities of acid-base balance and predisposition to metabolic acidosis in Metachromatic Leukodystrophy patients.

作者信息

Lorioli L, Cicalese M P, Silvani P, Assanelli A, Salvo I, Mandelli A, Fumagalli F, Fiori R, Ciceri F, Aiuti A, Sessa M, Roncarolo M G, Lanzani C, Biffi A

机构信息

San Raffaele Telethon Institute for Gene Therapy (HSR-TIGET), San Raffaele Hospital, Milano, Italy; Pediatric Immunohematology Unit, San Raffaele Hospital, Milano, Italy; Stem Cell Transplantation Program, San Raffaele Hospital, Milano, Italy; Vita-Salute San Raffaele University, Milano, Italy.

San Raffaele Telethon Institute for Gene Therapy (HSR-TIGET), San Raffaele Hospital, Milano, Italy; Pediatric Immunohematology Unit, San Raffaele Hospital, Milano, Italy.

出版信息

Mol Genet Metab. 2015 May;115(1):48-52. doi: 10.1016/j.ymgme.2015.02.009. Epub 2015 Mar 6.

DOI:10.1016/j.ymgme.2015.02.009
PMID:25796965
Abstract

Metachromatic Leukodystrophy (MLD; MIM# 250100) is a rare inherited lysosomal storage disorder caused by the deficiency of Arylsulfatase A (ARSA). The enzymatic defect results in the accumulation of the ARSA substrate that is particularly relevant in myelin forming cells and leads to progressive dysmyelination and dysfunction of the central and peripheral nervous system. Sulfatide accumulation has also been reported in various visceral organs, although little is known about the potential clinical consequences of such accumulation. Different forms of MLD-associated gallbladder disease have been described, and there is one reported case of an MLD patient presenting with functional consequences of sulfatide accumulation in the kidney. Here we describe a wide cohort of MLD patients in whom a tendency to sub-clinical metabolic acidosis was observed. Furthermore in some of them we report episodes of metabolic acidosis of different grades of severity developed in acute clinical conditions of various origin. Importantly, we finally show how a careful acid-base balance monitoring and prompt correction of imbalances might prevent severe consequences of acidosis.

摘要

异染性脑白质营养不良(MLD;MIM# 250100)是一种罕见的遗传性溶酶体贮积症,由芳基硫酸酯酶A(ARSA)缺乏引起。酶缺陷导致ARSA底物蓄积,这在髓鞘形成细胞中尤为重要,并导致中枢和周围神经系统进行性脱髓鞘和功能障碍。尽管关于这种蓄积的潜在临床后果知之甚少,但也有报道称在各种内脏器官中存在硫脂蓄积。已经描述了不同形式的与MLD相关的胆囊疾病,并且有一例MLD患者报告了硫脂在肾脏中蓄积的功能后果。在这里,我们描述了一大群MLD患者,观察到他们有亚临床代谢性酸中毒的倾向。此外,在其中一些患者中,我们报告了在各种不同病因的急性临床情况下发生的不同严重程度的代谢性酸中毒发作。重要的是,我们最终展示了仔细监测酸碱平衡并及时纠正失衡如何可能预防酸中毒的严重后果。

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