| Literature DB >> 32681750 |
Ruqaiah Altassan1,2, Silvia Radenkovic3,4,5,6, Andrew C Edmondson7, Rita Barone8, Sandra Brasil9,10,11, Anna Cechova12, David Coman13, Sarah Donoghue14, Kristina Falkenstein15, Vanessa Ferreira9, Carlos Ferreira16, Agata Fiumara8, Rita Francisco9,10,11, Hudson Freeze17, Stephanie Grunewald18, Tomas Honzik12, Jaak Jaeken19, Donna Krasnewich16, Christina Lam20,21, Joy Lee14, Dirk Lefeber22, Dorinda Marques-da-Silva9,10,11, Carlota Pascoal9,10,11, Dulce Quelhas23, Kimiyo M Raymond24, Daisy Rymen25, Malgorzata Seroczynska26, Mercedes Serrano27, Jolanta Sykut-Cegielska26, Christian Thiel15, Frederic Tort28, Mari-Anne Vals29, Paula Videira10,11, Nicol Voermans30, Peter Witters25,31, Eva Morava6.
Abstract
Phosphoglucomutase 1 (PGM1) deficiency is a rare genetic disorder that affects glycogen metabolism, glycolysis, and protein glycosylation. Previously known as GSD XIV, it was recently reclassified as a congenital disorder of glycosylation, PGM1-CDG. PGM1-CDG usually manifests as a multisystem disease. Most patients present as infants with cleft palate, liver function abnormalities and hypoglycemia, but some patients present in adulthood with isolated muscle involvement. Some patients develop life-threatening cardiomyopathy. Unlike most other CDG, PGM1-CDG has an effective treatment option, d-galactose, which has been shown to improve many of the patients' symptoms. Therefore, early diagnosis and initiation of treatment for PGM1-CDG patients are crucial decisions. In this article, our group of international experts suggests diagnostic, follow-up, and management guidelines for PGM1-CDG. These guidelines are based on the best available evidence-based data and experts' opinions aiming to provide a practical resource for health care providers to facilitate successful diagnosis and optimal management of PGM1-CDG patients.Entities:
Keywords: zzm321990d-galactose; PGM1-CDG; congenital disorder of glycosylation; management guidelines; phosphoglucomutase 1 deficiency
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Year: 2020 PMID: 32681750 PMCID: PMC7855268 DOI: 10.1002/jimd.12286
Source DB: PubMed Journal: J Inherit Metab Dis ISSN: 0141-8955 Impact factor: 4.982