FB2026_03 , released September 17, 2026
Human Disease Model Report: intellectual disability, X-linked, MICPCH syndrome
Open Close
General Information
Name
intellectual disability, X-linked, MICPCH syndrome
FlyBase ID
FBhh0000865
Overview

Intellectual disability, X-linked, MICPCH syndrome is one of several diseases associated with defects in the human CASK gene; this disease exhibits X-linked dominant inheritance. Severe mutations and deletions of CASK are typically implicated in this disease. See the human disease model report for 'intellectual disability, X-linked, CASK-related' (FBhh0000867) for information on experimental results using Drosophila models of this and related diseases.

[updated Aug. 2018 by FlyBase; FBrf0222196]

Disease Summary Information
Parent Disease Summary: intellectual disability, X-linked, syndromic
Symptoms and phenotype

Intellectual disability is characterized by impairments in intellectual functioning and adaptive behavior; symptoms must be present before a child becomes 18 years old (http://medical-dictionary.thefreedictionary.com/mental+retardation; 2016.01.19).

Intellectual disability can be subdivided into syndromic forms, characterized by cognitive impairment accompanied by dysmorphic features, malformations or neurological abnormalities, and nonsyndromic forms, characterized by cognitive impairment without additional features (Basel-Vanagaite, 2008; DOI: 10.1002/9780470015902.a0021454).

Specific Disease Summary: intellectual disability, X-linked, MICPCH syndrome
OMIM report

[INTELLECTUAL DEVELOPMENTAL DISORDER WITH MICROCEPHALY AND PONTINE AND CEREBELLAR HYPOPLASIA; MICPCH](https://omim.org/entry/300749)

Human gene(s) implicated

[CALCIUM/CALMODULIN-DEPENDENT SERINE PROTEIN KINASE; CASK](https://omim.org/entry/300172)

Symptoms and phenotype

Mental retardation and microcephaly with pontine and cerebellar hypoplasia (MICPCH) is an X-linked disorder affecting females and characterized by severe intellectual disability, microcephaly, and variable degrees of pontocerebellar hypoplasia. Affected individuals have very poor psychomotor development, often without independent ambulation or speech, axial hypotonia with or without hypertonia, and distinctive facial dysmorphism (summary by Moog et al., 2011; pubmed:21954287). [from MIM:300749; 2018.08.14]

Genetics

MICPCH syndrome is caused by heterozygous mutation or deletion in the CASK gene, typically resulting in complete loss of function. [from MIM:300749 and MIM:300422; 2018.08.14]

Cellular phenotype and pathology
Molecular information

The CASK gene encodes a calcium/calmodulin-dependent serine protein kinase that is a member of the membrane-associated guanylate kinase (MAGUK) protein family. MAGUKs are scaffolding proteins associated with intercellular junctions (summary by Atasoy et al., 2007; pubmed:17287346) [from MIM:300172; 2018.08.14]

External links
Disease synonyms
mental retardation, X-linked, syndromic, Najm type
Mental retardation and microcephaly with pontine and cerebellar hypoplasia
MICPCH
MRXSNA
X-linked intellectual disability, Najm type
Ortholog Information
Human gene(s) in FlyBase
    Human gene (HGNC)
    D. melanogaster ortholog (based on DIOPT)
    Comments on ortholog(s)
    Other mammalian ortholog(s) used
      D. melanogaster Gene Information (0)
      Other Genes Used: Viral, Bacterial, Synthetic (0)
        Summary of Physical Interactions (0 groups)
        Alleles Reported to Model Human Disease (Disease Ontology) (0 alleles)
        Alleles Representing Disease-Implicated Variants
        Genetic Tools, Stocks and Reagents
        Sources of Stocks
        Contact lab of origin for a reagent not available from a public stock center.
        Bloomington Stock Center Disease Page
        Related mammalian, viral, bacterial, or synthetic transgenes
        Allele
        Transgene
        Publicly Available Stocks
        Selected Drosophila transgenes
        Allele
        Transgene
        Publicly Available Stocks
        RNAi constructs available
        Allele
        Transgene
        Publicly Available Stocks
        Selected Drosophila classical alleles
        Allele
        Allele class
        Mutagen
        Publicly Available Stocks
        References (3)