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Pseudo-TORCH syndrome(BLCPMG)

MedGen UID:
483678
Concept ID:
C3489725
Disease or Syndrome
Synonyms: BAND-LIKE CALCIFICATION WITH SIMPLIFIED GYRATION AND POLYMICROGYRIA; BLCPMG; PSEUDO-TORCH SYNDROME
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:0009626
OMIM®: 251290
OMIM® Phenotypic series: PS251290
Orphanet: ORPHA1229

Definition

Pseudo-TORCH syndrome-1 (PTORCH1) is an autosomal recessive neurologic disorder with characteristic clinical and neuroradiologic features that mimic intrauterine TORCH infection in the absence of evidence of infection. Affected individuals have congenital microcephaly, intracranial calcifications, simplified gyration and polymicrogyria, and severe developmental delay (Reardon et al., 1994; O'Driscoll et al., 2010). Crow et al. (2000, 2003) called attention to the phenotypic overlap of pseudo-TORCH syndrome and Aicardi-Goutieres syndrome (AGS; 225750), and even suggested that some cases may represent the same disorder. Congenital microcephaly, thrombocytopenia, hepatic dysfunction, and hepatosplenomegaly are usually associated with pseudo-TORCH syndrome and not with AGS, but some patients with AGS have shown these features. Genetic Heterogeneity of Pseudo-TORCH Syndrome See also PTORCH2 (617397), caused by mutation in the USP18 gene (607057) on chromosome 22q11, and PTORCH3 (618886), caused by mutation in the STAT2 gene (600556) on chromosome 12q13. [from OMIM]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVPseudo-TORCH syndrome

Professional guidelines

PubMed

Dell'Isola GB, Dini G, Culpepper KL, Portwood KE, Ferrara P, Di Cara G, Verrotti A, Lodolo M
World J Pediatr 2023 Jul;19(7):635-643. Epub 2023 Jan 17 doi: 10.1007/s12519-022-00679-2. PMID: 36650407Free PMC Article
Cetin Gedik K, Lamot L, Romano M, Demirkaya E, Piskin D, Torreggiani S, Adang LA, Armangue T, Barchus K, Cordova DR, Crow YJ, Dale RC, Durrant KL, Eleftheriou D, Fazzi EM, Gattorno M, Gavazzi F, Hanson EP, Lee-Kirsch MA, Montealegre Sanchez GA, Neven B, Orcesi S, Ozen S, Poli MC, Schumacher E, Tonduti D, Uss K, Aletaha D, Feldman BM, Vanderver A, Brogan PA, Goldbach-Mansky R
Ann Rheum Dis 2022 May;81(5):601-613. Epub 2022 Jan 27 doi: 10.1136/annrheumdis-2021-221814. PMID: 35086813Free PMC Article
Lanzi G, D'Arrigo S, Drumbl G, Uggetti C, Fazzi E
Funct Neurol 2003 Apr-Jun;18(2):71-5. PMID: 12911136

Recent clinical studies

Etiology

Kukulka NA, Zarei S, Glass J, Bouska C, Schroder J, Sen K
Am J Med Genet A 2024 Nov;194(11):e63804. Epub 2024 Jun 28 doi: 10.1002/ajmg.a.63804. PMID: 38942733
Cohen MC, Karaman I, Squier W, Farrel T, Whitby EH
Pediatr Dev Pathol 2012 Jan-Feb;15(1):45-9. Epub 2011 Jul 15 doi: 10.2350/10-01-0783-CR.1. PMID: 21762029
Sanchis A, Cerveró L, Bataller A, Tortajada JL, Huguet J, Crow YJ, Ali M, Higuet LJ, Martínez-Frías ML
J Pediatr 2005 May;146(5):701-5. doi: 10.1016/j.jpeds.2005.01.033. PMID: 15870678

Diagnosis

Panigrahy N, Bakhru S, Lingappa L, Chirla D
BMJ Case Rep 2022 Dec 29;15(12) doi: 10.1136/bcr-2022-249192. PMID: 36581356Free PMC Article
Ekinci F, Yildizdas RD, Horoz OO, Herguner O, Bisgin A
Eur J Med Genet 2020 Jun;63(6):103923. Epub 2020 Mar 30 doi: 10.1016/j.ejmg.2020.103923. PMID: 32240828
Cohen MC, Karaman I, Squier W, Farrel T, Whitby EH
Pediatr Dev Pathol 2012 Jan-Feb;15(1):45-9. Epub 2011 Jul 15 doi: 10.2350/10-01-0783-CR.1. PMID: 21762029
Watts P, Kumar N, Ganesh A, Sastry P, Pilz D, Levin AV, Chitayat D
Eye (Lond) 2008 May;22(5):730-3. Epub 2007 Dec 14 doi: 10.1038/sj.eye.6703058. PMID: 18084237
Vivarelli R, Grosso S, Cioni M, Galluzzi P, Monti L, Morgese G, Balestri P
Brain Dev 2001 Mar;23(1):18-23. doi: 10.1016/s0387-7604(00)00188-1. PMID: 11226724

Therapy

Meuwissen ME, Schot R, Buta S, Oudesluijs G, Tinschert S, Speer SD, Li Z, van Unen L, Heijsman D, Goldmann T, Lequin MH, Kros JM, Stam W, Hermann M, Willemsen R, Brouwer RW, Van IJcken WF, Martin-Fernandez M, de Coo I, Dudink J, de Vries FA, Bertoli Avella A, Prinz M, Crow YJ, Verheijen FW, Pellegrini S, Bogunovic D, Mancini GM
J Exp Med 2016 Jun 27;213(7):1163-74. Epub 2016 Jun 20 doi: 10.1084/jem.20151529. PMID: 27325888Free PMC Article

Prognosis

Cohen MC, Karaman I, Squier W, Farrel T, Whitby EH
Pediatr Dev Pathol 2012 Jan-Feb;15(1):45-9. Epub 2011 Jul 15 doi: 10.2350/10-01-0783-CR.1. PMID: 21762029
Abdel-Salam GM, Zaki MS, Saleem SN, Gaber KR
Am J Med Genet A 2008 Nov 15;146A(22):2929-36. doi: 10.1002/ajmg.a.32549. PMID: 18925673
Sanchis A, Cerveró L, Bataller A, Tortajada JL, Huguet J, Crow YJ, Ali M, Higuet LJ, Martínez-Frías ML
J Pediatr 2005 May;146(5):701-5. doi: 10.1016/j.jpeds.2005.01.033. PMID: 15870678

Clinical prediction guides

Mochida GH, Ganesh VS, Felie JM, Gleason D, Hill RS, Clapham KR, Rakiec D, Tan WH, Akawi N, Al-Saffar M, Partlow JN, Tinschert S, Barkovich AJ, Ali B, Al-Gazali L, Walsh CA
Am J Hum Genet 2010 Dec 10;87(6):882-9. Epub 2010 Nov 25 doi: 10.1016/j.ajhg.2010.10.026. PMID: 21109224Free PMC Article
Sanchis A, Cerveró L, Bataller A, Tortajada JL, Huguet J, Crow YJ, Ali M, Higuet LJ, Martínez-Frías ML
J Pediatr 2005 May;146(5):701-5. doi: 10.1016/j.jpeds.2005.01.033. PMID: 15870678
Crow YJ, Black DN, Ali M, Bond J, Jackson AP, Lefson M, Michaud J, Roberts E, Stephenson JB, Woods CG, Lebon P
J Med Genet 2003 Mar;40(3):183-7. doi: 10.1136/jmg.40.3.183. PMID: 12624136Free PMC Article

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