Literature DB >> 26450566

The Frequencies of Different Inborn Errors of Metabolism in Adult Metabolic Centres: Report from the SSIEM Adult Metabolic Physicians Group.

S Sirrs1, C Hollak2, M Merkel3, A Sechi4, E Glamuzina5, M C Janssen6, R Lachmann7, J Langendonk8, M Scarpelli9, T Ben Omran10, F Mochel11, M C Tchan12.   

Abstract

BACKGROUND: There are few centres which specialise in the care of adults with inborn errors of metabolism (IEM). To anticipate facilities and staffing needed at these centres, it is of interest to know the distribution of the different disorders.
METHODS: A survey was distributed through the list-serve of the SSIEM Adult Metabolic Physicians group asking clinicians for number of patients with confirmed diagnoses, types of diagnoses and age at diagnosis.
RESULTS: Twenty-four adult centres responded to our survey with information on 6,692 patients. Of those 6,692 patients, 510 were excluded for diagnoses not within the IEM spectrum (e.g. bone dysplasias, hemochromatosis) or for age less than 16 years, leaving 6,182 patients for final analysis. The most common diseases followed by the adult centres were phenylketonuria (20.6%), mitochondrial disorders (14%) and lysosomal storage disorders (Fabry disease (8.8%), Gaucher disease (4.2%)). Amongst the disorders that can present with acute metabolic decompensation, the urea cycle disorders, specifically ornithine transcarbamylase deficiency, were most common (2.2%), followed by glycogen storage disease type I (1.5%) and maple syrup urine disease (1.1%). Patients were frequently diagnosed as adults, particularly those with mitochondrial disease and lysosomal storage disorders.
CONCLUSIONS: A wide spectrum of IEM are followed at adult centres. Specific knowledge of these disorders is needed to provide optimal care including up-to-date knowledge of treatments and ability to manage acute decompensation.

Entities:  

Year:  2015        PMID: 26450566      PMCID: PMC5580735          DOI: 10.1007/8904_2015_435

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  15 in total

1.  Neonatal screening for medium chain acyl-CoA deficiency: high incidence in Lower Saxony (northern Germany).

Authors:  S Sander; N Janzen; B Janetzky; S Scholl; U Steuerwald; J Schäfer; J Sander
Journal:  Eur J Pediatr       Date:  2001-05       Impact factor: 3.183

Review 2.  Adult presentations of medium-chain acyl-CoA dehydrogenase deficiency (MCADD).

Authors:  T F Lang
Journal:  J Inherit Metab Dis       Date:  2009-10-11       Impact factor: 4.982

3.  Prevalence of Fabry disease in patients with cryptogenic stroke: a prospective study.

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Journal:  Lancet       Date:  2005-11-19       Impact factor: 79.321

4.  Evaluation of newborn screening for medium chain acyl-CoA dehydrogenase deficiency in 275 000 babies.

Authors:  K Carpenter; V Wiley; K G Sim; D Heath; B Wilcken
Journal:  Arch Dis Child Fetal Neonatal Ed       Date:  2001-09       Impact factor: 5.747

5.  Carnitine transporter defect: diagnosis in asymptomatic adult women following analysis of acylcarnitines in their newborn infants.

Authors:  S Vijay; A Patterson; S Olpin; M J Henderson; S Clark; C Day; G Savill; J H Walter
Journal:  J Inherit Metab Dis       Date:  2006-07-23       Impact factor: 4.982

6.  Frequency of Fabry disease in male and female haemodialysis patients in Spain.

Authors:  Paulo Gaspar; Julio Herrera; Daniel Rodrigues; Sebastián Cerezo; Rodrigo Delgado; Carlos F Andrade; Ramón Forascepi; Juan Macias; Maria D del Pino; Maria D Prados; Pilar R de Alegria; Gerardo Torres; Pedro Vidau; Maria C Sá-Miranda
Journal:  BMC Med Genet       Date:  2010-02-01       Impact factor: 2.103

7.  Diagnoses of newborns and mothers with carnitine uptake defects through newborn screening.

Authors:  Ni-Chung Lee; Nelson Leung-Sang Tang; Yin-Hsiu Chien; Chun-An Chen; Sho-Juan Lin; Pao-Chin Chiu; Ai-Chu Huang; Wuh-Liang Hwu
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8.  Structure-function relationships in alpha-galactosidase A.

Authors:  Scott C Garman
Journal:  Acta Paediatr       Date:  2007-04       Impact factor: 2.299

Review 9.  A systematic review on screening for Fabry disease: prevalence of individuals with genetic variants of unknown significance.

Authors:  L van der Tol; B E Smid; B J H M Poorthuis; M Biegstraaten; R H Lekanne Deprez; G E Linthorst; C E M Hollak
Journal:  J Med Genet       Date:  2013-08-06       Impact factor: 6.318

Review 10.  Fatty acid oxidation disorders: outcome and long-term prognosis.

Authors:  Bridget Wilcken
Journal:  J Inherit Metab Dis       Date:  2010-01-05       Impact factor: 4.982

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  16 in total

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Authors:  Michel Tchan
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Review 2.  Unmet Cardiac Clinical Needs in Adult Mucopolysaccharidoses.

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Journal:  Front Cardiovasc Med       Date:  2022-06-10

3.  Identification of an Allosteric Binding Site on Human Lysosomal Alpha-Galactosidase Opens the Way to New Pharmacological Chaperones for Fabry Disease.

Authors:  Valentina Citro; Jorge Peña-García; Helena den-Haan; Horacio Pérez-Sánchez; Rosita Del Prete; Ludovica Liguori; Chiara Cimmaruta; Jan Lukas; Maria Vittoria Cubellis; Giuseppina Andreotti
Journal:  PLoS One       Date:  2016-10-27       Impact factor: 3.240

Review 4.  The Large Phenotypic Spectrum of Fabry Disease Requires Graduated Diagnosis and Personalized Therapy: A Meta-Analysis Can Help to Differentiate Missense Mutations.

Authors:  Valentina Citro; Marco Cammisa; Ludovica Liguori; Chiara Cimmaruta; Jan Lukas; Maria Vittoria Cubellis; Giuseppina Andreotti
Journal:  Int J Mol Sci       Date:  2016-12-01       Impact factor: 5.923

5.  Clinical characteristics of adult patients with inborn errors of metabolism in Spain: A review of 500 cases from university hospitals.

Authors:  J Pérez-López; L Ceberio-Hualde; J S García-Morillo; J M Grau-Junyent; A Hermida Ameijeiras; M López-Rodríguez; J C Milisenda; M Moltó Abad; M Morales-Conejo; J J Nava Mateos
Journal:  Mol Genet Metab Rep       Date:  2017-02-03

Review 6.  Second-Generation Pharmacological Chaperones: Beyond Inhibitors.

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Review 7.  Impact of pregnancy on inborn errors of metabolism.

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Journal:  Metabolites       Date:  2019-10-21

Review 9.  Neurometabolic disorders: Five new things.

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10.  Education and training in adult metabolic medicine: Results of an international survey.

Authors:  Annalisa Sechi; Elisa Fabbro; Mirjam Langeveld; Annarita Tullio; Robin Lachmann; Fanny Mochel
Journal:  JIMD Rep       Date:  2019-06-21
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