A rare phenomenon of twin spotting: Phacomatosis pigmentokeratotica

Phacomatosis pigmentokeratotica is a rare form of cutaneous mosaicism characterized by epidermal nevus usually nevus sebaceous, papular speckled lentiginous nevus, and extracutaneous anomalies. This type of twin-spot phenomenon (didymosis) is due to postzygotic crossing-over resulting in two homozyg...

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Published inIndian journal of paediatric dermatology Vol. 20; no. 3; pp. 267 - 270
Main Authors Wankhade, Vaishali, Shah, Vrutika, Singh, Rajesh, Mukhi, Jayesh
Format Journal Article
LanguageEnglish
Published Wolters Kluwer India Pvt. Ltd 01.07.2019
Medknow Publications and Media Pvt. Ltd
Wolters Kluwer Medknow Publications
Subjects
Analysis
Care and treatment
Child health
Cutaneous mosaicism
Medical research
Nevus
nevus sebaceous
Rare diseases
skeletal
speckled lentiginous nevus
Stem cells
twin-spot phenomenon
speckled lentiginous nevus
twin-spot phenomenon
Cutaneous mosaicism
skeletal
nevus sebaceous
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Abstract Phacomatosis pigmentokeratotica is a rare form of cutaneous mosaicism characterized by epidermal nevus usually nevus sebaceous, papular speckled lentiginous nevus, and extracutaneous anomalies. This type of twin-spot phenomenon (didymosis) is due to postzygotic crossing-over resulting in two homozygous daughter cells, representing the stem cells of the two distinct types of nevi and leading to additional extracutaneous defects. We report a case of this rare syndrome in a 4-year-old male child associated with skeletal anomalies.
AbstractList Phacomatosis pigmentokeratotica is a rare form of cutaneous mosaicism characterized by epidermal nevus usually nevus sebaceous, papular speckled lentiginous nevus, and extracutaneous anomalies. This type of twin-spot phenomenon (didymosis) is due to postzygotic crossing-over resulting in two homozygous daughter cells, representing the stem cells of the two distinct types of nevi and leading to additional extracutaneous defects. We report a case of this rare syndrome in a 4-year-old male child associated with skeletal anomalies.
Audience Academic
Author Shah, Vrutika
Mukhi, Jayesh
Wankhade, Vaishali
Singh, Rajesh
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Keywords speckled lentiginous nevus
twin-spot phenomenon
Cutaneous mosaicism
skeletal
nevus sebaceous
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References Wollenberg (key-10.4103/2319-7250.261883-6) 2002
Stosiek (key-10.4103/2319-7250.261883-4) 1994
Karia (key-10.4103/2319-7250.261883-1) 2018
Aschinberg (key-10.4103/2319-7250.261883-5) 1977
Happle (key-10.4103/2319-7250.261883-2) 2013
Oh (key-10.4103/2319-7250.261883-3) 2012
Gamayunov (key-10.4103/2319-7250.261883-7) 2016
References_xml – start-page: 278
  volume-title: Extensive linear epidermal nevus associated with hemangiomas of bones and Vitamin-D-resistant rickets
  year: 1994
  ident: key-10.4103/2319-7250.261883-4
  publication-title: Dermatology
  contributor:
    fullname: Stosiek
– start-page: 120
  issn: 0378-6323
  volume-title: Phacomatosis pigmentokeratotica: A very rare twin spotting phenomenon
  year: 2018
  ident: key-10.4103/2319-7250.261883-1
  publication-title: Indian J Dermatol Venereol Leprol
  contributor:
    fullname: Karia
– start-page: 1923
  volume-title: Phacomatosis pigmentokeratotica is a “pseudodidymosis”
  year: 2013
  ident: key-10.4103/2319-7250.261883-2
  publication-title: J Invest Dermatol
  contributor:
    fullname: Happle
– start-page: 56
  volume-title: Vitamin D-resistant rickets associated with epidermal nevus syndrome: Demonstration of a phosphaturic substance in the dermal lesions
  year: 1977
  ident: key-10.4103/2319-7250.261883-5
  publication-title: J Pediatr
  contributor:
    fullname: Aschinberg
– start-page: 1444
  volume-title: Phacomatosis pigmentokeratotica without extracutaneous abnormalities: A case study involving a preterm baby
  year: 2012
  ident: key-10.4103/2319-7250.261883-3
  publication-title: J Korean Med Sci
  contributor:
    fullname: Oh
– start-page: 564
  volume-title: Phacomatosis pigmentokeratotica or the Schimmelpenning-Feuerstein-Mims syndrome?
  year: 2016
  ident: key-10.4103/2319-7250.261883-7
  publication-title: Clin Case Rep
  contributor:
    fullname: Gamayunov
– start-page: 55
  volume-title: Phacomatosis pigmentokeratotica (Happle) in a 23-year-old man
  year: 2002
  ident: key-10.4103/2319-7250.261883-6
  publication-title: Acta Derm Venereol
  contributor:
    fullname: Wollenberg
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SubjectTerms Analysis
Care and treatment
Child health
Cutaneous mosaicism
Medical research
Nevus
nevus sebaceous
Rare diseases
skeletal
speckled lentiginous nevus
Stem cells
twin-spot phenomenon
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Title A rare phenomenon of twin spotting: Phacomatosis pigmentokeratotica
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