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Porokeratosis of Mibelli(POROK1)

MedGen UID:
181842
Concept ID:
C0949506
Disease or Syndrome
Synonym: Porokeratosis, Mibelli
SNOMED CT: Porokeratosis of Mibelli (80432009); Mibelli's disease (80432009)
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.
Not genetically inherited
MedGen UID:
988794
Concept ID:
CN307044
Finding
Source: Orphanet
clinical entity without genetic inheritance.
 
Monarch Initiative: MONDO:0019141
OMIM®: 175800; 607622
Orphanet: ORPHA735

Definition

Porokeratosis is a rare skin disorder characterized by one or more annular plaques with a surrounding raised horny border that spreads centrifugally. Variants of porokeratosis have been described that differ in morphologic shapes, distribution, and clinical course (Schamroth et al., 1997). However, as noted by Sybert (2010), several families with expression of more than one variant of porokeratosis among members, and several individuals expressing more than one variant, have been reported, suggesting that the distinctions among these variants may be artificial. Mutations in the MVK gene have been found to cause multiple types of porokeratosis, which have been described as porokeratosis of Mibelli, porokeratoma, genital porokeratosis, hyperkeratotic porokeratosis, and linear porokeratosis. The preferred title of this entry was formerly 'Porokeratosis 1, Mibelli Type; POROK1.' Genetic Heterogeneity of Porokeratosis Also see porokeratosis-2 (POROK2; 175850), mapped to chromosome 12q24; POROK3 (175900), caused by mutation in the MVK gene (251170) on chromosome 12q24; POROK4 (607728), mapped to chromosome 15q25-q26; POROK5 (612293), mapped to chromosome 1p31; POROK6 (612353), mapped to chromosome 1p31; POROK7 (614714), caused by mutation in the MVD gene (603236) on chromosome 16q24; POROK8 (616063), caused by mutation in the SLC17A9 gene (612107) on chromosome 20q13; and POROK9 (616631), caused by mutation in the FDPS gene (134629) on chromosome 1q22. A palmoplantar form of punctate porokeratosis has also been described (PPKP2; 175860). Genotype/Phenotype Correlations Zhang et al. (2015) screened 12 isoprenoid genes in 134 Chinese probands with porokeratosis and identified mutations in the MVK, MVD, PMVK, and FDPS genes in 113 patients. The authors noted that giant plaque-type porokeratosis ptychotropica with lesion diameters of at least 5 cm appeared to be uniquely associated with mutation in MVK; it was observed in 19 (50%) of 38 MVK probands, but not in patients with mutations in any of the other 3 genes or in the 21 probands in whom no mutation was found. MVK patients also showed the widest range in terms of the number and size of lesions, as well as presence of porokeratosis subtypes. In patients with MVD mutations, the age of onset ranged from 5 to 70 years, and lesion diameters were generally less than 2 cm. In addition, 6 of the 62 MVD probands exhibited solar facial porokeratosis, which was not seen in any other patients. Localized genital porokeratosis and porokeratoma appeared to be uniquely associated with mutation in the PMVK gene, whereas patients with mutations in the FDPS gene had more than 500 lesions, all with diameters of 1 cm or less. [from OMIM]

Clinical features

From HPO
Parakeratosis
MedGen UID:
10572
Concept ID:
C0030436
Disease or Syndrome
Abnormal formation of the keratinocytes of the epidermis characterized by persistence of nuclei, incomplete formation of keratin, and moistness and swelling of the keratinocytes.
Porokeratosis
MedGen UID:
56518
Concept ID:
C0162839
Congenital Abnormality
A clonal disorder of keratinization with one or multiple atrophic patches surrounded by a clinically and histologically distinctive hyperkeratotic ridgelike border called the cornoid lamella.

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVPorokeratosis of Mibelli
Follow this link to review classifications for Porokeratosis of Mibelli in Orphanet.

Professional guidelines

PubMed

Vargas-Mora P, Morgado-Carrasco D, Fustà-Novell X
Actas Dermosifiliogr (Engl Ed) 2020 Sep;111(7):545-560. Epub 2020 May 13 doi: 10.1016/j.ad.2020.03.005. PMID: 32401728
Weidner T, Illing T, Miguel D, Elsner P
Am J Clin Dermatol 2017 Aug;18(4):435-449. doi: 10.1007/s40257-017-0271-3. PMID: 28283894
Gonçalves JC
Arch Dermatol 1973 Jul;108(1):131-2. doi: 10.1001/archderm.1973.01620220085026. PMID: 4732842

Recent clinical studies

Etiology

Zaar O, Polesie S, Navarrete-Dechent C, Errichetti E, Akay BN, Jaimes J, Cabo H, Cohen Sabban E, Paoli J
J Eur Acad Dermatol Venereol 2021 Oct;35(10):2091-2096. Epub 2021 Jun 25 doi: 10.1111/jdv.17439. PMID: 34077570
Vargas-Mora P, Morgado-Carrasco D, Fustà-Novell X
Actas Dermosifiliogr (Engl Ed) 2020 Sep;111(7):545-560. Epub 2020 May 13 doi: 10.1016/j.ad.2020.03.005. PMID: 32401728
Ottoni LQ, Kakizaki P, Pinheiro RR, Sittart JA, Valente NY
An Bras Dermatol 2016 Sep-Oct;91(5 suppl 1):131-133. doi: 10.1590/abd1806-4841.20164253. PMID: 28300920Free PMC Article
Zeng K, Zhang QG, Li L, Duan Y, Liang YH
Arch Dermatol Res 2014 Oct;306(8):749-55. Epub 2014 Apr 30 doi: 10.1007/s00403-014-1465-7. PMID: 24781643
Ferreira FR, Santos LD, Tagliarini FA, Lira ML
An Bras Dermatol 2013 Nov-Dec;88(6 Suppl 1):179-82. doi: 10.1590/abd1806-4841.20132721. PMID: 24346913Free PMC Article

Diagnosis

Zaar O, Polesie S, Navarrete-Dechent C, Errichetti E, Akay BN, Jaimes J, Cabo H, Cohen Sabban E, Paoli J
J Eur Acad Dermatol Venereol 2021 Oct;35(10):2091-2096. Epub 2021 Jun 25 doi: 10.1111/jdv.17439. PMID: 34077570
Vargas-Mora P, Morgado-Carrasco D, Fustà-Novell X
Actas Dermosifiliogr (Engl Ed) 2020 Sep;111(7):545-560. Epub 2020 May 13 doi: 10.1016/j.ad.2020.03.005. PMID: 32401728
Elfatoiki FZ, Soussi W, Chiheb S, Jabri L, Benchikhi H
Pan Afr Med J 2015;20:195. Epub 2015 Mar 3 doi: 10.11604/pamj.2015.20.195.5003. PMID: 26113926Free PMC Article
Laino L, Pala S, Innocenzi D, Accappaticcio G, Van Steensel MA
Eur J Dermatol 2004 May-Jun;14(3):190-2. PMID: 15246948
Rothman IL, Wirth PB, Klaus MV
Int J Dermatol 1992 Jan;31(1):52-4. doi: 10.1111/j.1365-4362.1992.tb03522.x. PMID: 1737691

Therapy

Weidner T, Illing T, Miguel D, Elsner P
Am J Clin Dermatol 2017 Aug;18(4):435-449. doi: 10.1007/s40257-017-0271-3. PMID: 28283894
Elfatoiki FZ, Soussi W, Chiheb S, Jabri L, Benchikhi H
Pan Afr Med J 2015;20:195. Epub 2015 Mar 3 doi: 10.11604/pamj.2015.20.195.5003. PMID: 26113926Free PMC Article
Venkatarajan S, LeLeux TM, Yang D, Rosen T, Orengo I
Dermatol Online J 2010 Dec 15;16(12):10. PMID: 21199636
Montes-De-Oca-Sánchez G, Tirado-Sánchez A, García-Ramírez V
J Dermatolog Treat 2006;17(5):319-20. doi: 10.1080/09546630600944116. PMID: 17092865
Rothman IL, Wirth PB, Klaus MV
Int J Dermatol 1992 Jan;31(1):52-4. doi: 10.1111/j.1365-4362.1992.tb03522.x. PMID: 1737691

Prognosis

Kindem S, Serra-Guillén C, Sorní G, Guillén C, Sanmartín O
JAMA Dermatol 2015 Jan;151(1):85-6. doi: 10.1001/jamadermatol.2014.2697. PMID: 25409424
Malek J, Chedraoui A, Kibbi AG, Ghosn S
Am J Dermatopathol 2009 Aug;31(6):604-6. doi: 10.1097/DAD.0b013e3181a09948. PMID: 19590410
Dereli T, Ozyurt S, Ozturk G
J Dermatol 2004 Mar;31(3):223-7. doi: 10.1111/j.1346-8138.2004.tb00659.x. PMID: 15187342
Leow YH, Soon YH, Tham SN
Ann Acad Med Singap 1996 Nov;25(6):837-41. PMID: 9055013
McCullough TL, Lesher JL Jr
Pediatr Dermatol 1994 Sep;11(3):267-70. doi: 10.1111/j.1525-1470.1994.tb00601.x. PMID: 7971564

Clinical prediction guides

Zaar O, Polesie S, Navarrete-Dechent C, Errichetti E, Akay BN, Jaimes J, Cabo H, Cohen Sabban E, Paoli J
J Eur Acad Dermatol Venereol 2021 Oct;35(10):2091-2096. Epub 2021 Jun 25 doi: 10.1111/jdv.17439. PMID: 34077570
Koley S, Mandal RK, Bar C
Int J Dermatol 2014 Sep;53(9):1119-24. Epub 2014 Jun 5 doi: 10.1111/ijd.12495. PMID: 24898929
Tan LS, Chong WS
Australas J Dermatol 2012 May;53(2):e40-4. Epub 2012 Jan 19 doi: 10.1111/j.1440-0960.2011.00856.x. PMID: 22571584
Jurecka W, Neumann RA, Knobler RM
J Am Acad Dermatol 1991 Jan;24(1):96-101. doi: 10.1016/0190-9622(91)70019-x. PMID: 1999539
Inamoto N, Watanabe T, Nakamura K
J Am Acad Dermatol 1984 Aug;11(2 Pt 2):357-61. doi: 10.1016/s0190-9622(84)70172-1. PMID: 6237130

Recent systematic reviews

Weidner T, Illing T, Miguel D, Elsner P
Am J Clin Dermatol 2017 Aug;18(4):435-449. doi: 10.1007/s40257-017-0271-3. PMID: 28283894

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