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Erythrokeratodermia variabilis(EKV; EKVP; PSEK)

MedGen UID:
75587
Concept ID:
C0265961
Disease or Syndrome
Synonym: ERYTHROKERATODERMIA VARIABILIS ET PROGRESSIVA
SNOMED CT: Erythrokeratodermia variabilis (70041004); Congenital poikiloderma (70041004); Mendes da Costa syndrome (70041004)
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).
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.
 
Monarch Initiative: MONDO:0017851
OMIM®: 133200
OMIM® Phenotypic series: PS133200
Orphanet: ORPHA316

Definition

The erythrokeratodermias are a clinically variable and genetically heterogeneous group of inherited disorders characterized by widespread erythematous plaques, stationary or migratory, associated with nonmigratory hyperkeratoses (summary by Ishida-Yamamoto et al., 1997). The condition is usually present at birth or occurs during the first year but may begin later in childhood or even in early adulthood. Lesions preferentially affect the face, buttocks, and extensor surfaces of the limbs. Palmoplantar keratoderma occurs in about half the cases, but hair, nails, and teeth are not affected (summary by Macfarlane et al., 1991). Genetic Heterogeneity of Erythrokeratodermia Variabilis et Progressiva See EKVP2 (617524), caused by mutation in the GJB4 gene (605425); EKVP3 (617525), caused by mutation in the GJA1 gene (121014); EKVP4 (617526), caused by mutation in the KDSR gene (136440); EKVP5 (617756), caused by mutation in the KRT83 gene (602765); EKVP6 (618531), caused by mutation in the TRPM4 gene (606936); and EKPV7 (619209), caused by mutation in the PERP gene (609301). [from OMIM]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVErythrokeratodermia variabilis
Follow this link to review classifications for Erythrokeratodermia variabilis in Orphanet.

Professional guidelines

PubMed

Richard G, Brown N, Rouan F, Van der Schroeff JG, Bijlsma E, Eichenfield LF, Sybert VP, Greer KE, Hogan P, Campanelli C, Compton JG, Bale SJ, DiGiovanna JJ, Uitto J
J Invest Dermatol 2003 Apr;120(4):601-9. doi: 10.1046/j.1523-1747.2003.12080.x. PMID: 12648223
DiGiovanna JJ, Robinson-Bostom L
Am J Clin Dermatol 2003;4(2):81-95. doi: 10.2165/00128071-200304020-00002. PMID: 12553849
van der Schroeff JG, Ruiter DJ, Bots GT
Arch Dermatol Res 1982;274(3-4):339-48. doi: 10.1007/BF00403739. PMID: 6187301

Recent clinical studies

Etiology

Hotz A, Fölster-Holst R, Oji V, Bourrat E, Frank J, Marrakchi S, Ennouri M, Wankner L, Komlosi K, Alter S, Fischer J
Genes (Basel) 2024 Feb 24;15(3) doi: 10.3390/genes15030288. PMID: 38540347Free PMC Article
DiGiovanna JJ, Robinson-Bostom L
Am J Clin Dermatol 2003;4(2):81-95. doi: 10.2165/00128071-200304020-00002. PMID: 12553849
Happle R, van de Kerkhof PC, Traupe H
Dermatologica 1987;175 Suppl 1:107-24. doi: 10.1159/000248867. PMID: 2961628
van der Schroeff JG, Nijenhuis LE, Meera Khan P, Bernini LF, Schreuder GM, van Loghem E, Volkers WS, Went LN
Hum Genet 1984;68(2):165-8. doi: 10.1007/BF00279308. PMID: 6437964
Maekawa Y, Yasaka S
J Dermatol 1977 Aug;4(4):147-50. doi: 10.1111/j.1346-8138.1977.tb01028.x. PMID: 15461342

Diagnosis

Mikilita ES, Hernandez IP, Boff AL, Kiszewski AE
An Bras Dermatol 2020 Jul-Aug;95(4):484-489. Epub 2020 May 5 doi: 10.1016/j.abd.2019.09.030. PMID: 32482553Free PMC Article
Wu JP, Ge HS, Yang C
Chin Med J (Engl) 2020 Feb 20;133(4):501-502. doi: 10.1097/CM9.0000000000000633. PMID: 31977560Free PMC Article
Wang H, Xu Z, Lee BH, Vu S, Hu L, Lee M, Bu D, Cao X, Hwang S, Yang Y, Zheng J, Lin Z
J Invest Dermatol 2019 May;139(5):1089-1097. Epub 2018 Dec 5 doi: 10.1016/j.jid.2018.10.044. PMID: 30528822
Takeichi T, Sugiura K, Hsu CK, Nomura T, Takama H, Simpson MA, Shimizu H, McGrath JA, Akiyama M
Acta Derm Venereol 2016 May;96(4):557-9. doi: 10.2340/00015555-2307. PMID: 26632638
Santesteban Muruzábal R, Hervella Garcés M, Ros Martín C
Actas Dermosifiliogr 2015 Oct;106(8):667-8. Epub 2014 Dec 12 doi: 10.1016/j.ad.2014.11.002. PMID: 25512236

Therapy

Wu JP, Ge HS, Yang C
Chin Med J (Engl) 2020 Feb 20;133(4):501-502. doi: 10.1097/CM9.0000000000000633. PMID: 31977560Free PMC Article
Ishida-Yamamoto A
J Dermatol 2016 Mar;43(3):280-5. doi: 10.1111/1346-8138.13220. PMID: 26945536
Santesteban Muruzábal R, Hervella Garcés M, Ros Martín C
Actas Dermosifiliogr 2015 Oct;106(8):667-8. Epub 2014 Dec 12 doi: 10.1016/j.ad.2014.11.002. PMID: 25512236
Zhang L, Hong Y, Zheng S, Huo W, Qi R, Geng L, Chen HD, Gao XH
Dermatol Ther 2014 Jul-Aug;27(4):240-3. Epub 2014 Apr 22 doi: 10.1111/dth.12127. PMID: 24754264
Singh N, Thappa DM
Pediatr Dermatol 2010 Jan-Feb;27(1):111-3. doi: 10.1111/j.1525-1470.2009.01044.x. PMID: 20199434

Prognosis

Afzal G, Ahmed N, Jamal N, Zahoor F
J Pak Med Assoc 2024 Sep;74(9):1687-1689. doi: 10.47391/JPMA.9785. PMID: 39279078
Sampaio-Silva J, Batissoco AC, Jesus-Santos R, Abath-Neto O, Scarpelli LC, Nishimura PY, Galindo LT, Bento RF, Oiticica J, Lezirovitz K
Ann Hum Genet 2018 Jan;82(1):23-34. Epub 2017 Oct 17 doi: 10.1111/ahg.12213. PMID: 29044474
Richard G, Brown N, Rouan F, Van der Schroeff JG, Bijlsma E, Eichenfield LF, Sybert VP, Greer KE, Hogan P, Campanelli C, Compton JG, Bale SJ, DiGiovanna JJ, Uitto J
J Invest Dermatol 2003 Apr;120(4):601-9. doi: 10.1046/j.1523-1747.2003.12080.x. PMID: 12648223
DiGiovanna JJ, Robinson-Bostom L
Am J Clin Dermatol 2003;4(2):81-95. doi: 10.2165/00128071-200304020-00002. PMID: 12553849
Happle R, van de Kerkhof PC, Traupe H
Dermatologica 1987;175 Suppl 1:107-24. doi: 10.1159/000248867. PMID: 2961628

Clinical prediction guides

Charfeddine C, Laroussi N, Mkaouar R, Jouini R, Khayat O, Redissi A, Mosbah A, Dallali H, Chedly Debbiche A, Zaouak A, Fenniche S, Abdelhak S, Hammami-Ghorbel H
PLoS One 2021;16(10):e0258777. Epub 2021 Oct 20 doi: 10.1371/journal.pone.0258777. PMID: 34669720Free PMC Article
Sampaio-Silva J, Batissoco AC, Jesus-Santos R, Abath-Neto O, Scarpelli LC, Nishimura PY, Galindo LT, Bento RF, Oiticica J, Lezirovitz K
Ann Hum Genet 2018 Jan;82(1):23-34. Epub 2017 Oct 17 doi: 10.1111/ahg.12213. PMID: 29044474
Richard G, Brown N, Rouan F, Van der Schroeff JG, Bijlsma E, Eichenfield LF, Sybert VP, Greer KE, Hogan P, Campanelli C, Compton JG, Bale SJ, DiGiovanna JJ, Uitto J
J Invest Dermatol 2003 Apr;120(4):601-9. doi: 10.1046/j.1523-1747.2003.12080.x. PMID: 12648223
McFadden N, Oppedal BR, Ree K, Brandtzaeg P
Acta Derm Venereol 1987;67(4):284-8. PMID: 2445144
van der Schroeff JG, Nijenhuis LE, Meera Khan P, Bernini LF, Schreuder GM, van Loghem E, Volkers WS, Went LN
Hum Genet 1984;68(2):165-8. doi: 10.1007/BF00279308. PMID: 6437964

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