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肝移植恢复肝 N-糖基化,伴有持续的 IgG 糖基化异常:一例磷酸甘露糖变位酶-2 先天性糖基化障碍患者的 3 年随访。

Liver transplantation recovers hepatic N-glycosylation with persistent IgG glycosylation abnormalities: Three-year follow-up in a patient with phosphomannomutase-2-congenital disorder of glycosylation.

机构信息

Department of Clinical Genomics, Mayo Clinic, Rochester, MN, United States of America; Division of Medical Genetics, Stanford University, CA, United States of America.

Division of Pediatric Gastroenterology and Hepatology, Mayo Clinic, Rochester, MN, United States of America.

出版信息

Mol Genet Metab. 2023 Apr;138(4):107559. doi: 10.1016/j.ymgme.2023.107559. Epub 2023 Mar 17.

Abstract

Phosphomannomutase-2-congenital disorder of glycosylation (PMM2-CDG) is the most common CDG and presents with highly variable features ranging from isolated neurologic involvement to severe multi-organ dysfunction. Liver abnormalities occur in in almost all patients and frequently include hepatomegaly and elevated aminotransferases, although only a minority of patients develop progressive hepatic fibrosis and liver failure. No curative therapies are currently available for PMM2-CDG, although investigation into several novel therapies is ongoing. We report the first successful liver transplantation in a 4-year-old patient with PMM2-CDG. Over a 3-year follow-up period, she demonstrated improved growth and neurocognitive development and complete normalization of liver enzymes, coagulation parameters, and carbohydrate-deficient transferrin profile, but persistently abnormal IgG glycosylation and recurrent upper airway infections that did not require hospitalization. Liver transplant should be considered as a treatment option for PMM2-CDG patients with end-stage liver disease, however these patients may be at increased risk for recurrent bacterial infections post-transplant.

摘要

磷酸甘露糖变位酶-2 型先天性糖基化障碍(PMM2-CDG)是最常见的 CDG 类型,表现出高度可变的特征,从孤立的神经系统受累到严重的多器官功能障碍不等。几乎所有患者均存在肝脏异常,常包括肝肿大和转氨酶升高,尽管只有少数患者发生进行性肝纤维化和肝功能衰竭。目前尚无针对 PMM2-CDG 的治愈疗法,尽管正在对几种新疗法进行研究。我们报告了首例成功的 PMM2-CDG 4 岁患者肝移植。在 3 年的随访期间,她的生长和神经认知发育得到改善,肝酶、凝血参数和糖基化转移蛋白谱完全正常化,但 IgG 糖基化持续异常和反复上呼吸道感染,无需住院治疗。对于终末期肝病的 PMM2-CDG 患者,肝移植应被视为一种治疗选择,然而这些患者在移植后可能有更高的复发性细菌感染风险。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9b62/10164344/afe0c3c99da1/nihms-1885675-f0001.jpg

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