Literature DB >> 25855245

Case of Sjögren-Larsson syndrome with a large deletion in the ALDH3A2 gene confirmed by single nucleotide polymorphism array analysis.

Nagwa E A Gaboon1,2, Musharraf Jelani3,4, Mona M Almramhi3, Hussein S A Mohamoud3,5, Jumana Y Al-Aama1,3.   

Abstract

Sjögren-Larsson syndrome (SLS) is a neurocutaneous disorder inherited in an autosomal recessive fashion. SLS patients are characterized by lipid metabolism error, primarily leading to cardinal signs of ichthyosis, spasticity and mental retardation. Additional signs include short stature, epilepsy, retinal abnormalities and photophobia. More than 90 mutations of the ALDH3A2 gene have been reported for SLS, and such variants can be successfully detected at a rate of 94% by direct DNA sequencing. We performed direct sequencing of ALDH3A2 gene from the index patient, however, no mutation could be detected. HumanCytoSNPs12 array analysis and subsequent targeted single nucleotide polymorphism analysis revealed a novel deletion mutation at chromosome 17p11.2. This 67-Kb region includes the first five coding exons of ALDH3A2, and is flanked by rs2245639 and rs962801. To the best of our knowledge, this mutation is novel and our findings broaden the mutation spectrum of ALDH3A2 causing SLS phenotype.
© 2015 Japanese Dermatological Association.

Entities:  

Keywords:  67-Kb deletion; ALDH3A2; Saudi Arabia; Sjögren-Larsson syndrome; homozygous

Mesh:

Substances:

Year:  2015        PMID: 25855245     DOI: 10.1111/1346-8138.12861

Source DB:  PubMed          Journal:  J Dermatol        ISSN: 0385-2407            Impact factor:   4.005


  8 in total

1.  Phenotypic and mutational spectrum of thirty-five patients with Sjögren-Larsson syndrome: identification of eleven novel ALDH3A2 mutations and founder effects.

Authors:  Mohamed S Abdel-Hamid; Mahmoud Y Issa; Hasnaa M Elbendary; Sherif F Abdel-Ghafar; Karima Rafaat; Heba Hosny; Marian Girgis; Ghada M H Abdel-Salam; Maha S Zaki
Journal:  J Hum Genet       Date:  2019-07-05       Impact factor: 3.172

2.  Genetics and prospective therapeutic targets for Sjögren-Larsson Syndrome.

Authors:  William B Rizzo
Journal:  Expert Opin Orphan Drugs       Date:  2016-03-10       Impact factor: 0.694

Review 3.  Recent advances in understanding ichthyosis pathogenesis.

Authors:  Nareh V Marukian; Keith A Choate
Journal:  F1000Res       Date:  2016-06-24

4.  A Novel Missense Mutation in the CLPP Gene Causing Perrault Syndrome Type 3 in a Turkish Family.

Authors:  Fatma Dursun; Hussein Sheikh Ali Mohamoud; Noreen Karim; Muhammad Naeem; Musharraf Jelani; Heves Kırmızıbekmez
Journal:  J Clin Res Pediatr Endocrinol       Date:  2016-04-18

5.  Genotype and phenotype variability in Sjögren-Larsson syndrome.

Authors:  Maximilian Weustenfeld; Reiner Eidelpes; Matthias Schmuth; William B Rizzo; Johannes Zschocke; Markus A Keller
Journal:  Hum Mutat       Date:  2018-11-26       Impact factor: 4.878

Review 6.  Clinical and genetic spectrums of 413 North African families with inherited retinal dystrophies and optic neuropathies.

Authors:  Aymane Bouzidi; Hicham Charoute; Majida Charif; Ghita Amalou; Mostafa Kandil; Abdelhamid Barakat; Guy Lenaers
Journal:  Orphanet J Rare Dis       Date:  2022-05-12       Impact factor: 4.303

7.  Identification of a novel deletion within ALDH3A2 gene in an Iranian Family with Sjögren-Larsson Syndrome.

Authors:  Maryam Taghdiri; Atie Kashef; Majid Fardaei; Mohammad Miryounesi
Journal:  Clin Case Rep       Date:  2017-11-22

8.  Sjogren-Larsson Syndrome: A case series of five members from an extended family with a novel mutation.

Authors:  Kamel T Abidi; Naglaa M Kamal; Ayman A Bakkar A; Maram Alotaibi; Haifa Asseri; Kawthar A Bokari
Journal:  Mol Genet Genomic Med       Date:  2020-09-15       Impact factor: 2.183

  8 in total

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