Literature DB >> 3149149

Juvenile galactosialidosis in a white male: a new variant.

D Chitayat1, D A Applegarth, J Lewis, J E Dimmick, A Q McCormick, J G Hall.   

Abstract

We describe a 19-year-old white male with juvenile galactosialidosis. He presented with hip arthralgia and was found to have facial "coarseness," corneal clouding, mitral and aortic insufficiency, and hepatosplenomegaly. Ultrastructural studies of skin biopsy and peripheral blood lymphocytes showed membrane-bound inclusions containing sparse fibrillogranular material. Biochemical analysis showed elevated urinary sialyloligosaccharides and no free sialic acid. Fibroblast enzyme analysis showed low activities of both alpha-neuraminidase and beta-galactosidase. To date, most patients with juvenile galactosialidosis have been Japanese. However, unlike those patients, our patient did not have macular cherry-red spots, neurologic abnormalities, or mental retardation. We speculate that this young man represents a new subtype of juvenile galactosialidosis with a potentially different molecular defect from that of the Japanese variant.

Entities:  

Mesh:

Substances:

Year:  1988        PMID: 3149149     DOI: 10.1002/ajmg.1320310423

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  4 in total

1.  Ultrastructural change of ligamentum flavum in galactosialidosis.

Authors:  Sueo Nakama; Suguru Hagiwara; Masaki Kato; Yuji Kanaya; Hideaki Watanabe
Journal:  Eur Spine J       Date:  2013-08-30       Impact factor: 3.134

2.  Characteristics of the beta-galactosidase-carboxypeptidase complex in GM1-gangliosidosis and beta-galactosialidosis fibroblasts.

Authors:  R M D'Agrosa; M Hubbes; S Zhang; R Shankaran; J W Callahan
Journal:  Biochem J       Date:  1992-08-01       Impact factor: 3.857

3.  Left ventricular "diverticulum" in a patient affected by galactosialidosis.

Authors:  Alessandro Durante; Mariaemilia Traini; Roberto Spoladore
Journal:  Case Rep Med       Date:  2011-06-26

4.  A mutation in a mild form of galactosialidosis impairs dimerization of the protective protein and renders it unstable.

Authors:  X Y Zhou; N J Galjart; R Willemsen; N Gillemans; H Galjaard; A d'Azzo
Journal:  EMBO J       Date:  1991-12       Impact factor: 11.598

  4 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.