Literature DB >> 31282596

Expanding the phenotypic spectrum of MBOAT7-related intellectual disability.

Joseph E Jacher1, Nikita Roy2, Mohammad Ghaziuddin2, Jeffrey W Innis1,3,4.   

Abstract

MBOAT7 gene pathogenic variants are a newly discovered and rare cause for intellectual disability, autism spectrum disorder (ASD), seizures, truncal hypotonia, appendicular hypertonia, and below average head sizes (ranging from -1 to -3 standard deviations). There have been only 16 individuals previously reported who have MBOAT7-related intellectual disability, all of whom were younger than 10 years old and from consanguineous relationships. Thus, there is a lack of phenotypic information for adolescent and adult individuals with this disorder. Medical genetics and psychiatric evaluations in a 14-year-old female patient with a history of global developmental delay, intellectual disability, overgrowth with macrocephaly, metrorrhagia, seizures, basal ganglia hyperintensities, nystagmus, strabismus with amblyopia, ASD, anxiety, attention deficit hyperactivity disorder (ADHD), aggressive outbursts, and hyperphagia included a karyotype, methylation polymerase chain reaction for Prader-Willi/Angelman syndrome, chromosome microarray, and whole exome sequencing (WES), ADOS2, and ADI-R. WES identified a homozygous, likely pathogenic variant in the MBOAT7 gene (c.855-2A>G). This is the oldest known patient with MBOAT7-related intellectual disability, whose unique features compared with previously described individuals include overgrowth with macrocephaly, metrorrhagia, ophthalmological abnormalities, basal ganglia hyperintensities, unspecified anxiety disorder, and ADHD; combined type; and hyperphagia with the absence of appendicular hypertonia and cortical atrophy. More individuals need to be identified in order to delineate the full clinical spectrum of this disorder.
© 2019 Wiley Periodicals, Inc.

Entities:  

Keywords:  MBOAT7; intellectual disability; whole exome sequencing

Mesh:

Substances:

Year:  2019        PMID: 31282596     DOI: 10.1002/ajmg.b.32749

Source DB:  PubMed          Journal:  Am J Med Genet B Neuropsychiatr Genet        ISSN: 1552-4841            Impact factor:   3.568


  2 in total

1.  Functional and Structural Changes in the Membrane-Bound O-Acyltransferase Family Member 7 (MBOAT7) Protein: The Pathomechanism of a Novel MBOAT7 Variant in Patients With Intellectual Disability.

Authors:  Jiwon Lee; Amen Shamim; Jongho Park; Ja-Hyun Jang; Ji Hye Kim; Jeong-Yi Kwon; Jong-Won Kim; Kyeong Kyu Kim; Jeehun Lee
Journal:  Front Neurol       Date:  2022-04-18       Impact factor: 4.086

2.  Homozygous variants in the HEXB and MBOAT7 genes underlie neurological diseases in consanguineous families.

Authors:  Shazia Khan; Lettie E Rawlins; Gaurav V Harlalka; Muhammad Umair; Asmat Ullah; Shaheen Shahzad; Muhammad Javed; Emma L Baple; Andrew H Crosby; Wasim Ahmad; Asma Gul
Journal:  BMC Med Genet       Date:  2019-12-18       Impact factor: 2.103

  2 in total

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