Literature DB >> 25040319

Goltz syndrome and PORCN mosaicism.

David A Stevenson1, Meghan Chirpich, Yvonne Contreras, Heather Hanson, Karin Dent.   

Abstract

Goltz syndrome, also known as focal dermal hypoplasia, is characterized primarily by ectodermal and mesodermal defects. Manifestations include cutis aplasia, dermal hypoplasia, papillomas, chorioretinal colobomas, absent/dysplastic teeth, and skeletal anomalies. Goltz syndrome is an X-linked disorder due to mutations in PORCN, with a predominance of females affected. Germline mutations in PORCN are thought to result in embryonically lethality in males. We present a boy with a phenotype consistent with Goltz syndrome with low-level mosaicism for a novel mutation in PORCN from peripheral blood (c.956dupA; p.Asn320GlufsX99).
© 2014 The International Society of Dermatology.

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Year:  2014        PMID: 25040319      PMCID: PMC4245318          DOI: 10.1111/ijd.12605

Source DB:  PubMed          Journal:  Int J Dermatol        ISSN: 0011-9059            Impact factor:   2.736


  26 in total

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Journal:  Acta Derm Venereol       Date:  2001 Jun-Jul       Impact factor: 4.437

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Journal:  J Am Acad Dermatol       Date:  1981-03       Impact factor: 11.527

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Authors:  Said Alkindi; Malcolm Battin; Salim Aftimos; Diana Purvis
Journal:  Pediatr Dermatol       Date:  2012-11-07       Impact factor: 1.588

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Authors:  W L Broughton; J E Weaver; M C Bibro; B J White
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  4 in total

1.  Management of pedal fibrovascular papillomas in Goltz-Gorlin syndrome.

Authors:  Andrew J DeCrescenzo; Olga S Bachilo; Eric L Cole; Michael G Wilkerson
Journal:  JAAD Case Rep       Date:  2016-08-05

Review 2.  WNT Signaling and Bone: Lessons From Skeletal Dysplasias and Disorders.

Authors:  Yentl Huybrechts; Geert Mortier; Eveline Boudin; Wim Van Hul
Journal:  Front Endocrinol (Lausanne)       Date:  2020-04-09       Impact factor: 5.555

3.  Goltz syndrome in males: A clinical report of a male patient carrying a novel PORCN variant and a review of the literature.

Authors:  Sofia Frisk; Catherine Grandpeix-Guyodo; Karin Popovic Silwerfeldt; Helgi Thor Hjartarson; Dimitris Chatzianastassiou; Irina Magnusson; Tobias Laurell; Ann Nordgren
Journal:  Clin Case Rep       Date:  2018-09-21

4.  Novel insights into PORCN mutations, associated phenotypes and pathophysiological aspects.

Authors:  Annabelle Arlt; Nicolai Kohlschmidt; Andreas Hentschel; Enrika Bartels; Claudia Groß; Ana Töpf; Pınar Edem; Nora Szabo; Albert Sickmann; Nancy Meyer; Ulrike Schara-Schmidt; Jarred Lau; Hanns Lochmüller; Rita Horvath; Yavuz Oktay; Andreas Roos; Semra Hiz
Journal:  Orphanet J Rare Dis       Date:  2022-01-31       Impact factor: 4.123

  4 in total

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