Literature DB >> 24768072

[GACI syndrome: a case report with a neonatal beginning].

C Freychet1, C Gay2, M-P Lavocat2, G Teyssier2, H Patural2, J Bacchetta3, J Cottalorda4, B Bader Meunier5, A Linglart6, G Baujat7, J-L Stephan2.   

Abstract

GACI (generalized arterial calcification of infancy) is a rare autosomal recessive disorder characterized by arterial and periarticular calcifications. Most children die in the first months of life of cardiovascular complications. Hypophosphatemic rickets (HR) resistant to medical treatment may complete the phenotype and is associated with a milder phenotype. This report discusses the case of a girl who presented neonatal ectopic periarticular calcifications with spontaneous regression, and then at the age of 3 years developed HR. There was no clinical improvement after treatment with calcitriol and phosphate, and correction of alkaline phosphatase induced the recurrence of periarticular and tissular calcifications : the treatment was reduced and the bone distortion treated by surgery. GACI diagnosis was confirmed by genetic analysis. At the age of 4.5 years, she developed a retinal abnormality and decreased radial pulse: these clinical signs are usually observed in pseudoxanthoma elasticum (PXE). It is now established that GACI and PXE belong to the same entity characterized by arterial and tissular calcifications of which this original case report is an illustration.
Copyright © 2014 Elsevier Masson SAS. All rights reserved.

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Year:  2014        PMID: 24768072     DOI: 10.1016/j.arcped.2014.03.004

Source DB:  PubMed          Journal:  Arch Pediatr        ISSN: 0929-693X            Impact factor:   1.180


  5 in total

1.  Treatment of hypophosphatemic rickets in generalized arterial calcification of infancy (GACI) without worsening of vascular calcification.

Authors:  Carlos R Ferreira; Shira G Ziegler; Ashutosh Gupta; Catherine Groden; Kevin S Hsu; William A Gahl
Journal:  Am J Med Genet A       Date:  2016-02-09       Impact factor: 2.802

Review 2.  From variome to phenome: Pathogenesis, diagnosis and management of ectopic mineralization disorders.

Authors:  Eva Yg De Vilder; Olivier M Vanakker
Journal:  World J Clin Cases       Date:  2015-07-16       Impact factor: 1.337

3.  Response of the ENPP1-Deficient Skeletal Phenotype to Oral Phosphate Supplementation and/or Enzyme Replacement Therapy: Comparative Studies in Humans and Mice.

Authors:  Carlos R Ferreira; Dillon Kavanagh; Ralf Oheim; Kristin Zimmerman; Julian Stürznickel; Xiaofeng Li; Paul Stabach; R Luke Rettig; Logan Calderone; Colin MacKichan; Aaron Wang; Hunter A Hutchinson; Tracy Nelson; Steven M Tommasini; Simon von Kroge; Imke Ak Fiedler; Ethan R Lester; Gilbert W Moeckel; Björn Busse; Thorsten Schinke; Thomas O Carpenter; Michael A Levine; Mark C Horowitz; Demetrios T Braddock
Journal:  J Bone Miner Res       Date:  2021-02-18       Impact factor: 6.741

4.  Magnesium and Anti-phosphate Treatment with Bisphosphonates for Generalised Arterial Calcification of Infancy: A Case Report

Authors:  Fatma Dursun; Tülay Atasoy Öztürk; Serçin Güven; Heves Kırmızıbekmez; Gülcan Seymen Karabulut; Sevinç Kalın; Betül Sözeri
Journal:  J Clin Res Pediatr Endocrinol       Date:  2018-12-11

Review 5.  Generalized Arterial Calcification of Infancy: New Insights, Controversies, and Approach to Management.

Authors:  Alison M Boyce; Rachel I Gafni; Carlos R Ferreira
Journal:  Curr Osteoporos Rep       Date:  2020-06       Impact factor: 5.163

  5 in total

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