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Ermine phenotype

MedGen UID:
346466
Concept ID:
C1856899
Disease or Syndrome
Synonyms: O'Doherty syndrome; Pigmentary disorder with hearing loss
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:0009196
OMIM®: 227010
Orphanet: ORPHA999

Definition

A rare deafness characterized by the association of bilateral sensorineural hearing loss and white hair with scattered black tufts, as well as skin areas of hyper- and hypopigmentation. Additional reported features include global developmental delay and moderate intellectual disability, growth retardation, microcephaly, hypotonia, mild dysmorphic facial features (deeply set eyes, broad nasal bridge, slight bowing of the upper lip), retinal depigmentation, anomalies of the fingers and toes, and white matter abnormalities on brain imaging. [from ORDO]

Clinical features

From HPO
Sensorineural hearing loss disorder
MedGen UID:
9164
Concept ID:
C0018784
Disease or Syndrome
A type of hearing impairment in one or both ears related to an abnormal functionality of the cochlear nerve.
Intellectual disability, mild
MedGen UID:
10044
Concept ID:
C0026106
Mental or Behavioral Dysfunction
Mild intellectual disability is defined as an intelligence quotient (IQ) in the range of 50-69.
Vitiligo
MedGen UID:
22677
Concept ID:
C0042900
Disease or Syndrome
Generalized well circumscribed patches of leukoderma that are generally distributed over symmetric body locations and is due to autoimmune destruction of melanocytes.
White hair
MedGen UID:
66797
Concept ID:
C0239804
Finding
Hypopigmented hair that appears white.
White eyelashes
MedGen UID:
332275
Concept ID:
C1836736
Finding
White color (lack of pigmentation) of the eyelashes.
White eyebrow
MedGen UID:
373165
Concept ID:
C1836737
Finding
White color (lack of pigmentation) of the eyebrow.
Spotty hyperpigmentation
MedGen UID:
812509
Concept ID:
C3806179
Finding
Abnormal iris pigmentation
MedGen UID:
331733
Concept ID:
C1834387
Finding
Abnormal pigmentation of the iris.

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVErmine phenotype
Follow this link to review classifications for Ermine phenotype in Orphanet.

Recent clinical studies

Etiology

Gironi LC, Colombo E, Brusco A, Grosso E, Naretto VG, Guala A, Di Gregorio E, Zonta A, Zottarelli F, Pasini B, Savoia P
Medicina (Kaunas) 2019 Jul 7;55(7) doi: 10.3390/medicina55070345. PMID: 31284637Free PMC Article

Diagnosis

Gironi LC, Colombo E, Brusco A, Grosso E, Naretto VG, Guala A, Di Gregorio E, Zonta A, Zottarelli F, Pasini B, Savoia P
Medicina (Kaunas) 2019 Jul 7;55(7) doi: 10.3390/medicina55070345. PMID: 31284637Free PMC Article
Zarate YA, Pacheco MC, Bove KE, Gorlin R, Zhao H, Hopkin RJ
Am J Med Genet A 2009 Jun;149A(6):1253-6. doi: 10.1002/ajmg.a.32815. PMID: 19449401

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