Literature DB >> 27265460

Phenotypes and Genotypes in Five Children with Congenital Insensitivity to Pain with Anhidrosis.

Qing-Li Wang1, Shanna Guo2, Guangyou Duan2, Ying Ying2, Penghao Huang2, Jing Yu Liu3, Xianwei Zhang4.   

Abstract

BACKGROUND: Congenital insensitivity to pain with anhidrosis is an extremely rare hereditary disorder linked to variants in NTRK1. Our goal was to characterize the clinical features and the genetic basis of the disorder in Chinese patients.
METHODS: Patients were enrolled via social networking. Clinical features were investigated by interview, chart review, and physical examination. DNA was extracted from peripheral blood to genotype NTRK1 in patients and their parents. Variants identified were checked against a control cohort by high-throughput sequencing, and the effects of these variants were assessed in silico.
RESULTS: Clinical features in five patients were cataloged, and six loss-of-function NTRK1 variants were identified, including a frameshift variant c.963delG, a nonsense variant c.1804C>T, an intron variant c.851-33T>A, and three missense variants c.1802T>G, c.2074C>T, and c.2311C>T.
CONCLUSIONS: The results expand the spectrum of clinical and genetic features of congenital insensitivity to pain with anhidrosis and will help facilitate analysis of genotype-phenotype association in the future.
Copyright © 2016 The Authors. Published by Elsevier Inc. All rights reserved.

Entities:  

Keywords:  NTRK1; congenital insensitivity to pain with anhidrosis; genotype; phenotype; variant

Mesh:

Substances:

Year:  2016        PMID: 27265460     DOI: 10.1016/j.pediatrneurol.2016.04.006

Source DB:  PubMed          Journal:  Pediatr Neurol        ISSN: 0887-8994            Impact factor:   3.372


  6 in total

1.  Anesthetic management during adenotonsillectomy for twins with congenital insensitivity to pain with anhidrosis: two case reports.

Authors:  Cong Wang; Xianwei Zhang; Shanna Guo; Jiaoli Sun; Ningbo Li
Journal:  J Med Case Rep       Date:  2017-08-25

2.  Heterogeneity of clinical features and mutation analysis of NTRK1 in Han Chinese patients with congenital insensitivity to pain with anhidrosis.

Authors:  Ningbo Li; Shanna Guo; Qingli Wang; Guangyou Duan; Jiaoli Sun; Yi Liu; Jin Zhang; Cong Wang; Changmao Zhu; Jingyu Liu; Xianwei Zhang
Journal:  J Pain Res       Date:  2019-01-22       Impact factor: 3.133

3.  Phenotypic and genotypic features of a pair of Chinese identical twins with congenital insensitivity to pain and anhidrosis: A case report.

Authors:  Ningbo Li; Jiaoli Sun; Shanna Guo; Yi Liu; Cong Wang; Changmao Zhu; Xianwei Zhang
Journal:  Medicine (Baltimore)       Date:  2018-11       Impact factor: 1.889

4.  Splice modulating antisense oligonucleotides restore some acid-alpha-glucosidase activity in cells derived from patients with late-onset Pompe disease.

Authors:  May Thandar Aung-Htut; Kristin A Ham; Michel Tchan; Russell Johnsen; Frederick J Schnell; Sue Fletcher; Steve D Wilton
Journal:  Sci Rep       Date:  2020-04-21       Impact factor: 4.379

Review 5.  Phenotypic heterogeneity of intellectual disability in patients with congenital insensitivity to pain with anhidrosis: A case report and literature review.

Authors:  Zhenlei Liu; Jiaqi Liu; Gang Liu; Wenjian Cao; Sen Liu; Yixin Chen; Yuzhi Zuo; Weisheng Chen; Jun Chen; Yu Zhang; Shishu Huang; Guixing Qiu; Philip F Giampietro; Feng Zhang; Zhihong Wu; Nan Wu
Journal:  J Int Med Res       Date:  2018-04-05       Impact factor: 1.671

6.  Clinical, genomics and networking analyses of a high-altitude native American Ecuadorian patient with congenital insensitivity to pain with anhidrosis: a case report.

Authors:  Andrés López-Cortés; Ana Karina Zambrano; Patricia Guevara-Ramírez; Byron Albuja Echeverría; Santiago Guerrero; Eliana Cabascango; Andy Pérez-Villa; Isaac Armendáriz-Castillo; Jennyfer M García-Cárdenas; Verónica Yumiceba; Gabriela Pérez-M; Paola E Leone; César Paz-Y-Miño
Journal:  BMC Med Genomics       Date:  2020-08-17       Impact factor: 3.063

  6 in total

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