A Japanese boy with double diagnoses of 2p15p16.1 microdeletion syndrome and RP2-associated retinal disorder.


Journal

Human genome variation
ISSN: 2054-345X
Titre abrégé: Hum Genome Var
Pays: England
ID NLM: 101652445

Informations de publication

Date de publication:
17 Dec 2021
Historique:
received: 29 09 2021
accepted: 21 11 2021
revised: 16 11 2021
entrez: 18 12 2021
pubmed: 19 12 2021
medline: 19 12 2021
Statut: epublish

Résumé

2p15p16.1 microdeletion syndrome is a recently recognized congenital disorder characterized by developmental delay and dysmorphic features. RP2-associated retinal disorder (RP2-RD) is an X-linked inherited retinal disease with a childhood onset caused by a loss-of-function variant in the RP2 gene. Here, we describe a 14-year-old boy with double diagnoses of 2p15p16.1 microdeletion syndrome and RP2-RD. The recurrence risk of each condition and the indication for potential therapeutic options for RP2-RD are discussed.

Identifiants

pubmed: 34921139
doi: 10.1038/s41439-021-00178-2
pii: 10.1038/s41439-021-00178-2
pmc: PMC8683409
doi:

Types de publication

Journal Article

Langues

eng

Pagination

46

Informations de copyright

© 2021. The Author(s).

Références

Int J Mol Sci. 2019 Mar 26;20(6):
pubmed: 30917587
J Med Genet. 2007 Apr;44(4):269-76
pubmed: 16963482
JCI Insight. 2016 Mar 17;1(3):e85461
pubmed: 27699255
Clin Genet. 2004 Jan;65(1):7-10
pubmed: 15032968
Am J Med Genet A. 2017 Aug;173(8):2081-2087
pubmed: 28573701
J Med Genet. 2008 Feb;45(2):122-4
pubmed: 18245392
Am J Med Genet C Semin Med Genet. 2020 Sep;184(3):675-693
pubmed: 32875684
Am J Med Genet A. 2016 Sep;170(9):2338-48
pubmed: 27271787
Stem Cell Reports. 2020 Jul 14;15(1):67-79
pubmed: 32531192
Hum Mol Genet. 2015 Feb 15;24(4):972-86
pubmed: 25292197
J Med Genet. 2009 Sep;46(9):645-7
pubmed: 19724011
Mol Genet Genomic Med. 2017 May 22;5(4):429-437
pubmed: 28717667
Congenit Anom (Kyoto). 2015 Aug;55(3):125-32
pubmed: 25900130

Auteurs

Kazuki Yamazawa (K)

Medical Genetics Center, National Hospital Organization Tokyo Medical Center, Tokyo, Japan. kyamazawa@keio.jp.
Department of Pediatrics, National Hospital Organization Tokyo Medical Center, Tokyo, Japan. kyamazawa@keio.jp.

Kenji Shimizu (K)

Division of Medical Genetics, Saitama Children's Medical Center, Saitama, Japan.
Division of Medical Genetics and Cytogenetics, Shizuoka Children's Hospital, Shizuoka, Japan.

Hirofumi Ohashi (H)

Division of Medical Genetics, Saitama Children's Medical Center, Saitama, Japan.

Hidenori Haruna (H)

Department of Pediatrics and Adolescent Medicine, Juntendo University Faculty of Medicine, Tokyo, Japan.

Satomi Inoue (S)

Medical Genetics Center, National Hospital Organization Tokyo Medical Center, Tokyo, Japan.

Haruka Murakami (H)

Medical Genetics Center, National Hospital Organization Tokyo Medical Center, Tokyo, Japan.

Tatsuo Matsunaga (T)

Medical Genetics Center, National Hospital Organization Tokyo Medical Center, Tokyo, Japan.
Division of Hearing and Balance Research, National Institute of Sensory Organs, National Hospital Organization Tokyo Medical Center, Tokyo, Japan.

Takeshi Iwata (T)

Division of Molecular and Cellular Biology, National Institute of Sensory Organs, National Hospital Organization Tokyo Medical Center, Tokyo, Japan.

Kazushige Tsunoda (K)

Laboratory of Visual Physiology, Division of Vision Research, National Institute of Sensory Organs, National Hospital Organization Tokyo Medical Center, Tokyo, Japan.

Kaoru Fujinami (K)

Medical Genetics Center, National Hospital Organization Tokyo Medical Center, Tokyo, Japan. k.fujinami@ucl.ac.uk.
Laboratory of Visual Physiology, Division of Vision Research, National Institute of Sensory Organs, National Hospital Organization Tokyo Medical Center, Tokyo, Japan. k.fujinami@ucl.ac.uk.
UCL Institute of Ophthalmology, University College London, London, United Kingdom. k.fujinami@ucl.ac.uk.

Classifications MeSH