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ALG8-CDG: Molecular and phenotypic expansion suggests clinical management guidelines.
Albokhari, Daniah; Ng, Bobby G; Guberinic, Alis; Daniel, Earnest James Paul; Engelhardt, Nicole M; Barone, Rita; Fiumara, Agata; Garavelli, Livia; Trimarchi, Gabriele; Wolfe, Lynne; Raymond, Kimiyo M; Morava, Eva; He, Miao; Freeze, Hudson H; Lam, Christina; Edmondson, Andrew C.
Afiliación
  • Albokhari D; Department of Pediatrics, Division of Human Genetics, Section of Metabolism, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
  • Ng BG; Department of Pediatrics, Taibah University College of Medicine, Medina, Saudi Arabia.
  • Guberinic A; Human Genetics Program, Sanford Burnham Prebys, La Jolla, California, USA.
  • Daniel EJP; Department of Clinical Genomics, Mayo Clinic, Rochester, Minnesota, USA.
  • Engelhardt NM; Department of Pathology and Laboratory Medicine, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
  • Barone R; Department of Pediatrics, Division of Human Genetics, Section of Metabolism, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
  • Fiumara A; Department of Clinical and Experimental Medicine, Division of Child Neurology and Psychiatry, University of Catania, Catania, Italy.
  • Garavelli L; Department of Clinical and Experimental Medicine, Pediatric Clinic, University of Catania, Catania, Italy.
  • Trimarchi G; Medical Genetics Unit, Mother and Child Department, Local Health Authority (AUSL) of Reggio Emilia Research Unit (IRCCS), Arcispedale Santa Maria Nuova, Reggio Emilia, Italy.
  • Wolfe L; Medical Genetics Unit, Mother and Child Department, Local Health Authority (AUSL) of Reggio Emilia Research Unit (IRCCS), Arcispedale Santa Maria Nuova, Reggio Emilia, Italy.
  • Raymond KM; NIH Undiagnosed Diseases Program, Common Fund, Office of the Director, National Institutes of Health, Bethesda, Maryland, USA.
  • Morava E; Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, Minnesota, USA.
  • He M; Department of Clinical Genomics, Mayo Clinic, Rochester, Minnesota, USA.
  • Freeze HH; Department of Pathology and Laboratory Medicine, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
  • Lam C; Human Genetics Program, Sanford Burnham Prebys, La Jolla, California, USA.
  • Edmondson AC; Division of Genetic Medicine, Department of Pediatrics, University of Washington School of Medicine, Seattle, Washington, USA.
J Inherit Metab Dis ; 45(5): 969-980, 2022 09.
Article en En | MEDLINE | ID: mdl-35716054
Congenital disorders of glycosylation are a continuously expanding group of monogenic disorders of glycoprotein and glycolipid glycan biosynthesis. These disorders mostly manifest with multisystem involvement. Individuals with ALG8-CDG commonly present with hypotonia, protein-losing enteropathy, and hepatic involvement. Here, we describe seven unreported individuals diagnosed with ALG8-CDG based on biochemical and molecular testing and we identify nine novel variants in ALG8, bringing the total to 26 individuals with ALG8-CDG in the medical literature. In addition to the typical multisystem involvement documented in ALG8-CDG, our cohort includes the two oldest patients reported and further expands the phenotype of ALG8-CDG to include stable intellectual disability, autism spectrum disorder and other neuropsychiatric symptoms. We further expand the clinical features in a variety of organ systems including ocular, musculoskeletal, dermatologic, endocrine, and cardiac abnormalities and suggest a comprehensive evaluation and monitoring strategy to improve clinical management.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Trastornos Congénitos de Glicosilación / Trastorno del Espectro Autista Tipo de estudio: Diagnostic_studies / Guideline / Prognostic_studies Límite: Humans Idioma: En Revista: J Inherit Metab Dis Año: 2022 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Trastornos Congénitos de Glicosilación / Trastorno del Espectro Autista Tipo de estudio: Diagnostic_studies / Guideline / Prognostic_studies Límite: Humans Idioma: En Revista: J Inherit Metab Dis Año: 2022 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos