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Diencephalic-mesencephalic junction dysplasia

MedGen UID:
1641855
Concept ID:
C4707858
Disease or Syndrome
Synonym: Diencephalic-mesencephalic junction dysplasia syndrome
SNOMED CT: Diencephalic mesencephalic junction dysplasia (766871009)
Modes of inheritance:
Autosomal recessive inheritance
MedGen UID:
141025
Concept ID:
C0441748
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in individuals with two pathogenic alleles, either homozygotes (two copies of the same mutant allele) or compound heterozygotes (whereby each copy of a gene has a distinct mutant allele).
 
Monarch Initiative: MONDO:0017868
OMIM® Phenotypic series: PS251280
Orphanet: ORPHA319192

Definition

A rare genetic non-syndromic cerebral malformation with characteristics of severe intellectual disability, progressive postnatal microcephaly, axial hypotonia, spastic quadriparesis, seizures and facial dysmorphism (bushy eyebrows, hairy forehead, broad nasal root, long flat philtrum, V-shaped upper lip). Additionally, talipes equinovarus, non-obstructive cardiomyopathy, persistent hyperplastic primary vitreous, obstructive hydrocephalus and autistic features may also be associated. On brain magnetic resonance imaging, the ''butterfly sign'' is characteristically observed and cortical calcifications, agenesis of the corpus callosum, ventriculomegaly, brainstem dysplasia and cerebellar vermis hypoplasia have also been described. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • Diencephalic-mesencephalic junction dysplasia

Professional guidelines

PubMed

Lawrence AK, Whitehead MT, Kruszka P, Sanapo L, Yano S, Tanpaiboon P, Muenke M, Fraser JL, du Plessis AJ
Prenat Diagn 2021 May;41(6):778-790. Epub 2021 Feb 26 doi: 10.1002/pd.5909. PMID: 33522008

Recent clinical studies

Etiology

Jacobs E, Whitehead MT
Pediatr Radiol 2023 Jan;53(1):121-130. Epub 2022 Jul 22 doi: 10.1007/s00247-022-05439-y. PMID: 35867110
Accogli A, Goergen S, Izzo G, Mankad K, Krajden Haratz K, Parazzini C, Fahey M, Menzies L, Baptista J, Carpineta L, Tortora D, Fulcheri E, Gaetano Vellone V, Paladini D, Spaccini L, Toto V, Trayers C, Ben Sira L, Reches A, Malinger G, Salpietro V, De Marco P, Srour M, Zara F, Capra V, Rossi A, Severino M
Ann Clin Transl Neurol 2021 Oct;8(10):2004-2012. Epub 2021 Sep 12 doi: 10.1002/acn3.51448. PMID: 34510796Free PMC Article
Lawrence AK, Whitehead MT, Kruszka P, Sanapo L, Yano S, Tanpaiboon P, Muenke M, Fraser JL, du Plessis AJ
Prenat Diagn 2021 May;41(6):778-790. Epub 2021 Feb 26 doi: 10.1002/pd.5909. PMID: 33522008
Whitehead MT, Najim N
AJNR Am J Neuroradiol 2020 Apr;41(4):729-735. Epub 2020 Feb 27 doi: 10.3174/ajnr.A6446. PMID: 32115420Free PMC Article

Diagnosis

Fileva N, Severino M, Tortora D, Ramaglia A, Paladini D, Rossi A
J Clin Ultrasound 2023 Feb;51(2):283-299. doi: 10.1002/jcu.23423. PMID: 36785503
Lawrence AK, Whitehead MT, Kruszka P, Sanapo L, Yano S, Tanpaiboon P, Muenke M, Fraser JL, du Plessis AJ
Prenat Diagn 2021 May;41(6):778-790. Epub 2021 Feb 26 doi: 10.1002/pd.5909. PMID: 33522008
Mądry J, Szlufik S, Koziorowski D, Królicki L, Friedman A
Neurol Neurochir Pol 2017 Nov-Dec;51(6):514-518. Epub 2017 Aug 16 doi: 10.1016/j.pjnns.2017.08.005. PMID: 28864328
Severino M, Righini A, Tortora D, Pinelli L, Parazzini C, Morana G, Accorsi P, Capra V, Paladini D, Rossi A
AJNR Am J Neuroradiol 2017 Aug;38(8):1643-1646. Epub 2017 Jun 8 doi: 10.3174/ajnr.A5245. PMID: 28596193Free PMC Article
Abdel Razek AA, Castillo M
J Comput Assist Tomogr 2016 Jan-Feb;40(1):14-25. doi: 10.1097/RCT.0000000000000340. PMID: 26599961

Prognosis

Whitehead MT, Najim N
AJNR Am J Neuroradiol 2020 Apr;41(4):729-735. Epub 2020 Feb 27 doi: 10.3174/ajnr.A6446. PMID: 32115420Free PMC Article
Guemez-Gamboa A, Çağlayan AO, Stanley V, Gregor A, Zaki MS, Saleem SN, Musaev D, McEvoy-Venneri J, Belandres D, Akizu N, Silhavy JL, Schroth J, Rosti RO, Copeland B, Lewis SM, Fang R, Issa MY, Per H, Gumus H, Bayram AK, Kumandas S, Akgumus GT, Erson-Omay EZ, Yasuno K, Bilguvar K, Heimer G, Pillar N, Shomron N, Weissglas-Volkov D, Porat Y, Einhorn Y, Gabriel S, Ben-Zeev B, Gunel M, Gleeson JG
Ann Neurol 2018 Nov;84(5):638-647. Epub 2018 Oct 4 doi: 10.1002/ana.25327. PMID: 30178464Free PMC Article

Clinical prediction guides

Accogli A, Goergen S, Izzo G, Mankad K, Krajden Haratz K, Parazzini C, Fahey M, Menzies L, Baptista J, Carpineta L, Tortora D, Fulcheri E, Gaetano Vellone V, Paladini D, Spaccini L, Toto V, Trayers C, Ben Sira L, Reches A, Malinger G, Salpietro V, De Marco P, Srour M, Zara F, Capra V, Rossi A, Severino M
Ann Clin Transl Neurol 2021 Oct;8(10):2004-2012. Epub 2021 Sep 12 doi: 10.1002/acn3.51448. PMID: 34510796Free PMC Article
Whitehead MT, Najim N
AJNR Am J Neuroradiol 2020 Apr;41(4):729-735. Epub 2020 Feb 27 doi: 10.3174/ajnr.A6446. PMID: 32115420Free PMC Article
Guemez-Gamboa A, Çağlayan AO, Stanley V, Gregor A, Zaki MS, Saleem SN, Musaev D, McEvoy-Venneri J, Belandres D, Akizu N, Silhavy JL, Schroth J, Rosti RO, Copeland B, Lewis SM, Fang R, Issa MY, Per H, Gumus H, Bayram AK, Kumandas S, Akgumus GT, Erson-Omay EZ, Yasuno K, Bilguvar K, Heimer G, Pillar N, Shomron N, Weissglas-Volkov D, Porat Y, Einhorn Y, Gabriel S, Ben-Zeev B, Gunel M, Gleeson JG
Ann Neurol 2018 Nov;84(5):638-647. Epub 2018 Oct 4 doi: 10.1002/ana.25327. PMID: 30178464Free PMC Article

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