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Guttmacher syndrome

MedGen UID:
401304
Concept ID:
C1867801
Disease or Syndrome
Synonyms: Autosomal dominant preaxial deficiency, postaxial polydactyly, and hypospadias; Preaxial deficiency, postaxial polydactyly and hypospadias
SNOMED CT: Preaxial deficiency, postaxial polydactyly, hypospadias syndrome (722452004); Guttmacher syndrome (722452004)
Modes of inheritance:
Autosomal dominant inheritance
MedGen UID:
141047
Concept ID:
C0443147
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 heterozygotes. In the context of medical genetics, an autosomal dominant disorder is caused when a single copy of the mutant allele is present. Males and females are affected equally, and can both transmit the disorder with a risk of 50% for each child of inheriting the mutant allele.
 
Gene (location): HOXA13 (7p15.2)
 
Monarch Initiative: MONDO:0008301
OMIM®: 176305
Orphanet: ORPHA2957

Definition

An extremely rare syndrome with characteristics of hypoplastic thumbs and halluces, fifth finger brachydactyly, postaxial polydactyly of the hands, short or uniphalangeal second toes with absent nails and hypospadias. It has been described in a father and his son and daughter. The affected patients have normal mental development. Except for postaxial polydactyly of the hands and uniphalangeal second toes with absent nails, features are in common with hand-foot-genital syndrome caused by mutations in the HOXA13 gene. In all three affected individuals, two different sequence alterations were identified in HOXA13 gene: a de novo missense mutation and a deletion in the promoter region of the gene, inherited from an unaffected parent, which may contribute to the phenotype in the affected individuals. The condition is inherited in an autosomal dominant manner. [from SNOMEDCT_US]

Clinical features

From HPO
Glandular hypospadias
MedGen UID:
452551
Concept ID:
C0452168
Congenital Abnormality
Short thumb
MedGen UID:
98469
Concept ID:
C0431890
Congenital Abnormality
Hypoplasia (congenital reduction in size) of the thumb.
Postaxial hand polydactyly
MedGen UID:
609221
Concept ID:
C0431904
Congenital Abnormality
Supernumerary digits located at the ulnar side of the hand (that is, on the side with the fifth finger).
Short 5th finger
MedGen UID:
334269
Concept ID:
C1842878
Congenital Abnormality
Hypoplasia (congenital reduction in size) of the fifth finger, also known as the little finger.
Short 2nd toe
MedGen UID:
867399
Concept ID:
C4021769
Anatomical Abnormality
Underdevelopment (hypoplasia) of the second toe.

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVGuttmacher syndrome
Follow this link to review classifications for Guttmacher syndrome in Orphanet.

Professional guidelines

PubMed

Hart SA, Nandi D, Backes CH, Cua CL
Pediatr Res 2021 Nov;90(5):1081-1085. Epub 2021 Mar 5 doi: 10.1038/s41390-021-01416-7. PMID: 33674738
Shovlin CL, Guttmacher AE, Buscarini E, Faughnan ME, Hyland RH, Westermann CJ, Kjeldsen AD, Plauchu H
Am J Med Genet 2000 Mar 6;91(1):66-7. doi: 10.1002/(sici)1096-8628(20000306)91:1<66::aid-ajmg12>3.0.co;2-p. PMID: 10751092

Recent clinical studies

Diagnosis

Imagawa E, Kayserili H, Nishimura G, Nakashima M, Tsurusaki Y, Saitsu H, Ikegawa S, Matsumoto N, Miyake N
Am J Med Genet A 2014 Sep;164A(9):2398-402. Epub 2014 Jun 16 doi: 10.1002/ajmg.a.36648. PMID: 24934387

Clinical prediction guides

Imagawa E, Kayserili H, Nishimura G, Nakashima M, Tsurusaki Y, Saitsu H, Ikegawa S, Matsumoto N, Miyake N
Am J Med Genet A 2014 Sep;164A(9):2398-402. Epub 2014 Jun 16 doi: 10.1002/ajmg.a.36648. PMID: 24934387

Supplemental Content

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