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Novel contiguous gene deletion in peruvian girl with Trichothiodystrophy type 4 and glutaric aciduria type 3

  • Jorge La Serna-Infantes
    ,
  • Miguel Chávez Pastor
    ,
  • Milana Trubnykova
    ,
  • Félix Chavesta Velásquez
    ,
  • Flor Vásquez Sotomayor
    ,
  • Hospital Guillermo Almenara Irigoyen
    ,
  • Instituto Nacional de Salud del Niño
    ,
Research Output:
Contribution to journal
Article
Peer-review

Publication Information

Output type

Research Output:
Contribution to journal
Article
Peer-review

Original language

English

Pages from-to (Number of pages)

Pages 388-392 (5 pages)

Journal (Volume, Issue Number)

European Journal of Medical Genetics (Volume 61, Issue 7)

Publication milestones

  • Published - 07/2018

Publication status

Published - 07/2018

ISSN

1769-7212

Publication IDs

  • Scopus: 85042196535
  • PubMed: 29421601

Abstract

Trichothiodystrophy type 4 is a rare autosomal recessive and ectodermal disorder, characterized by dry, brittle, sparse and sulfur-deficient hair and other features like intellectual disability, ichthyotic skin and short stature, caused by a homozygous mutation in MPLKIP gene. Glutaric aciduria type 3 is caused by a homozygous mutation in SUGCT gene with no distinctive phenotype. Both genes are localized on chromosome 7 (7p14). We report an 8-year-old female with short stature, microcephaly, development delay, intellectual disability and hair characterized for dark, short, coarse, sparse and brittle associated to classical trichorrhexis microscopy pattern. Chromosome microarray analysis showed a 125 kb homozygous pathogenic deletion, which includes genes MPLKIP and SUGCT, not described before. This is the first case described in Peru of a novel contiguous gene deletion of Trichothiodystrophy type 4 and Glutaric aciduria type 3 performed by chromosome microarray analysis, highlighting the contribution and importance of molecular technologies on diagnosis of rare genetic conditions.