Literature DB >> 24926462

A novel de novo mutation within PHEX gene in a young girl with hypophosphatemic rickets and review of literature.

Chong Kun Cheon1, Hoon Sang Lee1, Su Yung Kim1, Min Jung Kwak2, Gu-Hwan Kim3, Han-Wook Yoo3.   

Abstract

X-linked hypophosphatemia (XLH) is the most common form of familial hypophosphatemic rickets and it is caused by loss-of-function mutations in the PHEX gene. Recently, a wide variety of PHEX gene defects in XLH have been revealed; these include missense mutations, nonsense mutations, splice site mutations, insertions, and deletions. Recently, we encountered a 2-year-9-month-old female with sporadic hypophosphatemic rickets. She underwent osteotomy, dental abscess was evident, and there was severe bowing of the legs. A low serum phosphorus level in combination with elevated serum alkaline phosphatase activity and normal serum calcium is suggestive of hypophosphatemic rickets. PHEX gene analysis revealed a splice acceptor site mutation, c.934-1G>T (IVS8(-1)G>T), at the intron8 and exon9 junction. To the best of our knowledge, this mutation is novel and has not been reported. The results of this study expand and improve our understanding of the clinical and molecular characteristics and the global pool of patients with sporadic hypophosphatemic rickets.

Entities:  

Keywords:  Hunam PHEX protein; Hypophospatemic rickets; Mutation

Year:  2014        PMID: 24926462      PMCID: PMC4049552          DOI: 10.6065/apem.2014.19.1.36

Source DB:  PubMed          Journal:  Ann Pediatr Endocrinol Metab        ISSN: 2287-1012


  18 in total

1.  A novel PHEX mutation in a Korean patient with sporadic hypophosphatemic rickets.

Authors:  Juwon Kim; Kyu Hyun Yang; Ji Sun Nam; Jong Rak Choi; Jaewoo Song; Myungsook Chang; Kyung-A Lee
Journal:  Ann Clin Lab Sci       Date:  2009       Impact factor: 1.256

2.  Mutational survey of the PHEX gene in patients with X-linked hypophosphatemic rickets.

Authors:  Shoji Ichikawa; Elizabeth A Traxler; Selina A Estwick; Leah R Curry; Michelle L Johnson; Andrea H Sorenson; Erik A Imel; Michael J Econs
Journal:  Bone       Date:  2008-06-18       Impact factor: 4.398

3.  Genomic organization of the human PEX gene mutated in X-linked dominant hypophosphatemic rickets.

Authors:  F Francis; T M Strom; S Hennig; A Böddrich; B Lorenz; O Brandau; K L Mohnike; M Cagnoli; C Steffens; S Klages; K Borzym; T Pohl; C Oudet; M J Econs; P S Rowe; R Reinhardt; T Meitinger; H Lehrach
Journal:  Genome Res       Date:  1997-06       Impact factor: 9.043

4.  Growth in PHEX-associated X-linked hypophosphatemic rickets: the importance of early treatment.

Authors:  Catherine Quinlan; Katie Guegan; Amaka Offiah; Richard O' Neill; Melanie P Hiorns; Sian Ellard; Detlef Bockenhauer; William Van't Hoff; Aoife M Waters
Journal:  Pediatr Nephrol       Date:  2011-11-20       Impact factor: 3.714

Review 5.  Novel PHEX nonsense mutation in a patient with X-linked hypophosphatemic rickets and review of current therapeutic regimens.

Authors:  T Kienitz; M Ventz; E Kaminsky; M Quinkler
Journal:  Exp Clin Endocrinol Diabetes       Date:  2011-05-06       Impact factor: 2.949

6.  Genetic diagnosis of X-linked dominant Hypophosphatemic Rickets in a cohort study: tubular reabsorption of phosphate and 1,25(OH)2D serum levels are associated with PHEX mutation type.

Authors:  Marcos Morey; Lidia Castro-Feijóo; Jesús Barreiro; Paloma Cabanas; Manuel Pombo; Marta Gil; Ignacio Bernabeu; José M Díaz-Grande; Lourdes Rey-Cordo; Gema Ariceta; Itxaso Rica; José Nieto; Ramón Vilalta; Loreto Martorell; Jaime Vila-Cots; Fernando Aleixandre; Ana Fontalba; Leandro Soriano-Guillén; José M García-Sagredo; Sixto García-Miñaur; Berta Rodríguez; Saioa Juaristi; Carmen García-Pardos; Antonio Martínez-Peinado; José M Millán; Ana Medeira; Oana Moldovan; Angeles Fernandez; Lourdes Loidi
Journal:  BMC Med Genet       Date:  2011-09-08       Impact factor: 2.103

7.  Three novel PHEX gene mutations in Japanese patients with X-linked hypophosphatemic rickets.

Authors:  K Sato; T Tajima; J Nakae; M Adachi; Y Asakura; K Tachibana; S Suwa; N Katsumata; T Tanaka; Y Hayashi; S Abe; M Murashita; K Okuhara; N Shinohara; K Fujieda
Journal:  Pediatr Res       Date:  2000-10       Impact factor: 3.756

8.  PHEX analysis in 118 pedigrees reveals new genetic clues in hypophosphatemic rickets.

Authors:  Céline Gaucher; Odile Walrant-Debray; Thy-Minh Nguyen; Laure Esterle; Michèle Garabédian; Frédéric Jehan
Journal:  Hum Genet       Date:  2009-02-15       Impact factor: 4.132

9.  Novel and de novo PHEX mutations in patients with hypophosphatemic rickets.

Authors:  Erdem Durmaz; Minjing Zou; Roua A Al-Rijjal; Essa Y Baitei; Sumaya Hammami; Iffet Bircan; Sema Akçurin; Brian Meyer; Yufei Shi
Journal:  Bone       Date:  2012-10-16       Impact factor: 4.398

10.  Mutational analysis of PHEX gene in X-linked hypophosphatemia.

Authors:  P H Dixon; P T Christie; C Wooding; D Trump; M Grieff; I Holm; J M Gertner; J Schmidtke; B Shah; N Shaw; C Smith; C Tau; D Schlessinger; M P Whyte; R V Thakker
Journal:  J Clin Endocrinol Metab       Date:  1998-10       Impact factor: 5.958

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  7 in total

Review 1.  Genetics of Refractory Rickets: Identification of Novel PHEX Mutations in Indian Patients and a Literature Update.

Authors:  Binata Marik; Arvind Bagga; Aditi Sinha; Pankaj Hari; Arundhati Sharma
Journal:  J Pediatr Genet       Date:  2018-01-28

2.  Dentoalveolar Abscesses Not Associated with Caries or Trauma: A Diagnostic Hallmark of Hypophosphatemic Rickets Initially Misdiagnosed as Hypochondroplasia.

Authors:  Silvia Elena Yacarini Paredes; Raquel Assed Bezerra Segato; Leila Daher Moreira; Alcides Moreira; Kranya Victoria Díaz Serrano; Clarissa Teles Rodrigues; Luciana Yamamoto Almeida; Jorge Esquiche León
Journal:  Head Neck Pathol       Date:  2017-11-30

3.  Whole Exome Sequencing Reveals Novel PHEX Splice Site Mutations in Patients with Hypophosphatemic Rickets.

Authors:  Sara L Ma; Virginia Vega-Warner; Christopher Gillies; Matthew G Sampson; Vijay Kher; Sidharth K Sethi; Edgar A Otto
Journal:  PLoS One       Date:  2015-06-24       Impact factor: 3.240

4.  Whole exome sequencing confirms the clinical diagnosis of Marfan syndrome combined with X-linked hypophosphatemia.

Authors:  Xunlun Sheng; Xue Chen; Bo Lei; Rui Chen; Hui Wang; Fangxia Zhang; Weining Rong; Ruoshui Ha; Yani Liu; Feng Zhao; Peizeng Yang; Chen Zhao
Journal:  J Transl Med       Date:  2015-06-04       Impact factor: 5.531

5.  X-linked Hypophosphatemic Rickets, del(2)(q37.1;q37.3) Deletion Syndrome and Mosaic Turner Syndrome, mos 45,X/46,X, del(2)(q37.1;q37.3) in a 3-year-old Female.

Authors:  Alaina P Vidmar; Brian Miyazaki; Pedro A Sanchez-Lara; Pisit Pitukcheewanont
Journal:  J Bone Metab       Date:  2017-11-30

6.  Identification of a novel variant in the PHEX gene using targeted gene panel sequencing in a 24-month-old boy with hypophosphatemic rickets.

Authors:  Ha Young Jo; Jung Hyun Shin; Hye Young Kim; Young Mi Kim; Heirim Lee; Mi Hye Bae; Kyung Hee Park; Ja-Hyun Jang; Min Jung Kwak
Journal:  Ann Pediatr Endocrinol Metab       Date:  2020-03-31

7.  Phenotypes of a family with XLH with a novel PHEX mutation.

Authors:  Akiko Yamamoto; Toshiro Nakamura; Yasuhisa Ohata; Takuo Kubota; Keiichi Ozono
Journal:  Hum Genome Var       Date:  2020-03-31
  7 in total

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