Evidence map›Paper›PMID 38418803›Full record

ReviewJournal of applied genetics2024

Novel FOXP2 variant associated with speech and language dysfunction in a Chinese family and literature review.

Fengyu Che, Chenhao Li, Liyu Zhang, Chenxi Qian, Lidangzhi Mo, Benchang Li, Haibin Wu, Lifang Wang, Ying Yang

Erratum issuedAbstract readReview
PubMed Publisher
In one paragraph

Review in Journal of applied genetics, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
1.1field-weighted citation impact, top 22% 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

0 citing papers in PubMed, 2 citations in OpenAlex.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

9 authors at 3 institutions in 1 country.

Fengyu Che *Shaanxi Institute for Pediatric Diseases, Xi'an Children's Hospital, Xi'an, China.
Chenhao Li *Medical School of Chinese PLA, Beijing, China.
Liyu ZhangShaanxi Institute for Pediatric Diseases, Xi'an Children's Hospital, Xi'an, China.
Chenxi QianSouthern Medical University, Guangzhou, China.
Lidangzhi MoShaanxi Institute for Pediatric Diseases, Xi'an Children's Hospital, Xi'an, China.
Benchang LiShaanxi Institute for Pediatric Diseases, Xi'an Children's Hospital, Xi'an, China.
Haibin WuShaanxi Institute for Pediatric Diseases, Xi'an Children's Hospital, Xi'an, China.
Lifang Wang *Department of Child Health Care, Xi'an Children's Hospital, Xi'an, China. cm1666@sina.com.
Ying Yang *Shaanxi Institute for Pediatric Diseases, Xi'an Children's Hospital, Xi'an, China. yying1930@163.com.
Xi’an Children’s Hospital · CNPLA Academy of Military Science · CNSouthern Medical University · CN

Funding

Xi'an Children's Hospital research project 2021D01
6 · The paper itself

Abstract

Since its initial identification, the Forkhead Box P2 gene (FOXP2) has maintained its singular status as the archetypal monogenic determinant implicated in Mendelian forms of human speech and language impairments. Despite the passage of two decades subsequent to its discovery, extant literature remains disproportionately sparse with regard to case-specific instances and loci of mutational perturbations. The objective of the current investigation centers on furnishing an enriched delineation of both its clinical manifestations and its mutational heterogeneity. Clinical phenotypes and peripheral blood samples were assiduously amassed from familial subjects. Whole-exome sequencing and Sanger sequencing methodologies were deployed for the unambiguous identification of potential genetic variants and for corroborating their co-segregation within the family pedigree. An exhaustive review of published literature focusing on patients manifesting speech and language disorders consequent to FOXP2 genetic anomalies was also undertaken. The investigation yielded the identification of a novel heterozygous variant, c.661del (p.L221Ffs*41), localized within the FOXP2 gene in the proband, an inheritance from his symptomatic mother. The proband presented with an array of symptoms, encompassing dysarthric speech, deficits in instruction comprehension, and communicative impediments. In comparison, the mother exhibited attenuated symptoms, including rudimentary verbalization capabilities punctuated by pronounced stuttering and dysarthria. A comprehensive analysis of articles archived in the Human Gene Mutation Database (HGMD) classified under "DM" disclosed the existence of 74 patients inclusive of the subjects under current examination, sub-divided into 19 patients with null variants, 5 patients with missense variants, and 50 patients with gross deletions or complex genomic rearrangements. A conspicuous predominance of delayed speech, impoverished current verbal abilities, verbal comprehension deficits, and learning difficulties were observed in patients harboring null or missense FOXP2 variants, as compared to their counterparts with gross deletions or complex rearrangements. Developmental delays, hypotonia, and craniofacial aberrations were exclusive to the latter cohort. The elucidated findings augment the existing corpus of knowledge on the genetic architecture influencing both the proband and his mother within this specified familial context. Of critical importance, these discoveries furnish a robust molecular framework conducive to the prenatal diagnostic evaluations of prospective progeny within this familial lineage.

Indexed as

Language DisordersSpeechChinaForkhead Transcription FactorsHumansMutationForkhead Transcription FactorsFOXP2 protein, humanFOXP2Genotype and phenotypeSpeech-language disorder

Identifiers

PMID38418803
OpenAlexW4392229800

What OpenQuestion holds

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Registered trials

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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.