Literature DB >> 28555253

A Turkish case of galactosialidosis with a new homozygous mutation in CTSA gene.

Ayşe Kartal1, Kürşad Aydın2.   

Abstract

Galactosialidosis is an autosamal reressive lysosomal storage disease caused by a combined deficiency of lysosomal β-galactosidase and neuraminidase, due to a primary defect in protective protein/cathepsin A. Three subtypes are recognized: the early infantile type, the late infantile type, and the juvenile/adult type. We report here a female patient with early infantile galactosialidosis who was born at 35 weeks of gestation. After birth she remained at the neonatal intensive care unit. Physical examination revealed, coarse facial features, hepatomegaly, cardiac murmur and diffuse hypotonia. The patient's mother had a past history of fetal hydrops history. The diagnosis of galactosialidosis was confirmed by decreased activity of β-galactosidase and undetectable neuraminidase activity in fibroblasts. Genetic examination revealed a new homozygous mutation (c.1284delG) in the CTSA gene.

Entities:  

Keywords:  Fetal hydrops; Galactosialidosis; Homozygous mutation; Lysosomal storage disease

Mesh:

Substances:

Year:  2017        PMID: 28555253     DOI: 10.1007/s11011-017-0042-0

Source DB:  PubMed          Journal:  Metab Brain Dis        ISSN: 0885-7490            Impact factor:   3.584


  8 in total

1.  Structural and functional study of K453E mutant protective protein/cathepsin A causing the late infantile form of galactosialidosis.

Authors:  K Takiguchi; K Itoh; M Shimmoto; P T Ozand; H Doi; H Sakuraba
Journal:  J Hum Genet       Date:  2000       Impact factor: 3.172

2.  New mutations in two Dutch patients with early infantile galactosialidosis.

Authors:  J Groener; P Maaswinkel-Mooy; V Smit; M van der Hoeven; J Bakker; Y Campos; A d'Azzo
Journal:  Mol Genet Metab       Date:  2003-03       Impact factor: 4.797

3.  Congenital ascites as a presenting sign of lysosomal storage disease.

Authors:  J E Gillan; J A Lowden; K Gaskin; E Cutz
Journal:  J Pediatr       Date:  1984-02       Impact factor: 4.406

4.  Chemical chaperone treatment for galactosialidosis: Effect of NOEV on β-galactosidase activities in fibroblasts.

Authors:  Mohammad Arif Hossain; Katsumi Higaki; Michiko Shinpo; Eiji Nanba; Yoshiyuki Suzuki; Keiichi Ozono; Norio Sakai
Journal:  Brain Dev       Date:  2015-08-07       Impact factor: 1.961

5.  Infantile form of sialic acid storage disorder: clinical, ultrastructural, and biochemical studies in two siblings.

Authors:  M Tondeur; J Libert; E Vamos; F Van Hoof; G H Thomas; G Strecker
Journal:  Eur J Pediatr       Date:  1982-10       Impact factor: 3.183

6.  Juvenile galactosialidosis with attacks of neuropathic pain and absence of sialyloligosacchariduria.

Authors:  Niklas Darin; Mårten Kyllerman; Anna-Lena Hård; Claes Nordborg; Jan-Eric Månsson
Journal:  Eur J Paediatr Neurol       Date:  2008-12-18       Impact factor: 3.140

7.  A case of galactosialidosis with a homozygous Q49R point mutation.

Authors:  Naoko Matsumoto; Kenjiro Gondo; Johji Kukita; Katsumi Higaki; Rubigilda C Paragison; Eiji Nanba
Journal:  Brain Dev       Date:  2008-04-18       Impact factor: 1.961

Review 8.  Galactosialidosis: review and analysis of CTSA gene mutations.

Authors:  Anna Caciotti; Serena Catarzi; Rodolfo Tonin; Licia Lugli; Carmen Rodriguez Perez; Helen Michelakakis; Irene Mavridou; Maria Alice Donati; Renzo Guerrini; Alessandra d'Azzo; Amelia Morrone
Journal:  Orphanet J Rare Dis       Date:  2013-08-02       Impact factor: 4.123

  8 in total
  1 in total

Review 1.  Cathepsins in the Pathophysiology of Mucopolysaccharidoses: New Perspectives for Therapy.

Authors:  Valeria De Pasquale; Anna Moles; Luigi Michele Pavone
Journal:  Cells       Date:  2020-04-15       Impact factor: 6.600

  1 in total

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