Evidence map›Paper›PMID 33037390›Full record

ArticleJournal of human genetics2021

The identification of two pathogenic variants in a family with mild and severe forms of developmental delay.

Noriko Miyake, Shermineh Heydari, Masoud Garshasbi, Shinji Saitoh, Jafar Nasiri, Kohei Hamanaka, Atsushi Takata, Naomichi Matsumoto, Farnaz Hosseini Beheshti, Ahmad Reza Salehi Chaleshtori

Abstract readCase Reports
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In one paragraph

Article in Journal of human genetics, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.2field-weighted citation impact, top 38% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed, 2 citations in OpenAlex.

  1. Article
  2. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

10 authors at 4 institutions in 2 countries.

Noriko Miyake *Yokohama City University Graduate School of Medicine, Yokohama, 236-0004, Japan.
Shermineh HeydariDepartment of Medical Genetics, Faculty of Medical Sciences, Tarbiat Modares University, P.O. Box, 14115-331, Tehran, Iran.
Masoud GarshasbiDepartment of Medical Genetics, Faculty of Medical Sciences, Tarbiat Modares University, P.O. Box, 14115-331, Tehran, Iran.
Shinji SaitohDepartment of Pediatrics and Neonatology, Nagoya City University Graduate School of Medical Sciences, Nagoya, 467-8601, Japan.ORCID http://orcid.org/0000-0001-6911-3351
Jafar NasiriDepartment of Pediatric Neurology, Child Growth and Development Research Center & Research Institute for Primordial Prevention of Non-communicable Disease, Isfahan University of Medical Sciences, Isfahan, Iran.
Kohei HamanakaYokohama City University Graduate School of Medicine, Yokohama, 236-0004, Japan.
Atsushi TakataYokohama City University Graduate School of Medicine, Yokohama, 236-0004, Japan.
Naomichi MatsumotoYokohama City University Graduate School of Medicine, Yokohama, 236-0004, Japan.
Farnaz Hosseini BeheshtiDepartment of Pediatric Neurology, Child Growth and Development Research Center & Research Institute for Primordial Prevention of Non-communicable Disease, Isfahan University of Medical Sciences, Isfahan, Iran.
Ahmad Reza Salehi Chaleshtori *Department of Medical Genetics, Faculty of Medical Sciences, Tarbiat Modares University, P.O. Box, 14115-331, Tehran, Iran. ar.salehichaleshtori@modares.ac.ir.
Yokohama City University · JPTarbiat Modares University · IRIsfahan University of Medical Sciences · IRNagoya City University · JP

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Intellectual disability (ID) accounts for 1% of the general population, and it is caused by the interplay between the genetic and/or environmental factors. The genetic components responsible for the development of ID are highly heterogeneous, and the phenotype and severity of the disease vary in patients even if they have an identical pathological variant and/or belong to the same family. Herein, we reported two male siblings with ID in an Iranian family. By means of the whole-exome sequencing method, elder brother affected by a moderate form of ID exhibited a de novo missense variant in the KCNQ3 gene, while another sibling afflicted with a severe form of the disease exhibited a de novo in-frame deletion in the UBE3A gene. Both variants have been previously ascribed to similar clinical phenotypes. In addition, a genetic variant in the KCNQ3 gene was transmitted to his son, who had a mild form of ID. To our knowledge, all individuals with KCNQ3-related developmental delay show de novo variants in the KCNQ3 gene. Thus, this familial case exhibit milder phenotype that might extend the clinical spectrum of KCNQ3 pathogenic variants. In addition, the current report highlights the significance of the clinical evaluation and non-biased assessment of the genetic analysis.

Indexed as

Genetic Predisposition to DiseaseMutationChildDevelopmental DisabilitiesFemaleGenetic Association StudiesHumansIntellectual DisabilityKCNQ3 Potassium ChannelMalePedigreePhenotypeSiblingsUbiquitin-Protein LigasesKCNQ3 Potassium ChannelKCNQ3 protein, humanUBE3A protein, humanUbiquitin-Protein Ligases

Identifiers

PMID33037390
OpenAlexW3091945605

What OpenQuestion holds

Textmetadata
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.