Literature DB >> 16355816

Hypertrichosis cubiti (hairy elbow syndrome): a clue to a malformation syndrome.

Agata Polizzi1, Piero Pavone, Elisabetta Ciancio, Clementina La Rosa, Giovanni Sorge, Martino Ruggieri.   

Abstract

Hypertrichosis cubiti (HC) or hairy elbow syndrome (OMIM # 139600) consists of a localised form of long vellus hair on the extensor surfaces of the distal third of the upper arm and the proximal third of the forearm bilaterally, or occasionally on other parts of the body. In the 28 cases reported in the literature so far the elbow hair abnormality was either isolated or associated with short stature or other physical abnormalities. Most of these cases were sporadic, but autosomal dominant as well as autosomal recessive inheritance patterns have been postulated. We report on three unrelated girls (aged 7 to 11 years) of whom one presented with excess hair in the elbows alone and the other two had associated abnormalities including short stature, dysmorphic facial features and mental retardation. The literature on this subject has been reviewed and the authors focus on cases of HC with associated anomalies. A pathogenic explanation by somatic mosaicism is proposed.

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Year:  2005        PMID: 16355816     DOI: 10.1515/jpem.2005.18.10.1019

Source DB:  PubMed          Journal:  J Pediatr Endocrinol Metab        ISSN: 0334-018X            Impact factor:   1.634


  8 in total

1.  Whole exome sequencing reveals a MLL de novo mutation associated with mild developmental delay and without 'hairy elbows': expanding the phenotype of Wiedemann-Steiner syndrome.

Authors:  Dora Steel; Vincenzo Salpietro; Rahul Phadke; Matthew Pitt; Giulia Gentile; Ahmed Massoud; Leigh Batten; Anu Bashamboo; Ken Mcelreavey; Anand Saggar; Maria Kinali
Journal:  J Genet       Date:  2015-12       Impact factor: 1.166

Review 2.  Intellectual Disability: When the Hypertrichosis Is a Clue.

Authors:  Lidia Pezzani; Donatella Milani; Gianluca Tadini
Journal:  J Pediatr Genet       Date:  2015-09-28

3.  Global transcriptional disturbances underlie Cornelia de Lange syndrome and related phenotypes.

Authors:  Bo Yuan; Davut Pehlivan; Ender Karaca; Nisha Patel; Wu-Lin Charng; Tomasz Gambin; Claudia Gonzaga-Jauregui; V Reid Sutton; Gozde Yesil; Sevcan Tug Bozdogan; Tulay Tos; Asuman Koparir; Erkan Koparir; Christine R Beck; Shen Gu; Huseyin Aslan; Ozge Ozalp Yuregir; Khalid Al Rubeaan; Dhekra Alnaqeb; Muneera J Alshammari; Yavuz Bayram; Mehmed M Atik; Hatip Aydin; B Bilge Geckinli; Mehmet Seven; Hakan Ulucan; Elif Fenercioglu; Mustafa Ozen; Shalini Jhangiani; Donna M Muzny; Eric Boerwinkle; Beyhan Tuysuz; Fowzan S Alkuraya; Richard A Gibbs; James R Lupski
Journal:  J Clin Invest       Date:  2015-01-09       Impact factor: 14.808

4.  De novo mutations in MLL cause Wiedemann-Steiner syndrome.

Authors:  Wendy D Jones; Dimitra Dafou; Meriel McEntagart; Wesley J Woollard; Frances V Elmslie; Muriel Holder-Espinasse; Melita Irving; Anand K Saggar; Sarah Smithson; Richard C Trembath; Charu Deshpande; Michael A Simpson
Journal:  Am J Hum Genet       Date:  2012-07-12       Impact factor: 11.025

5.  West syndrome followed by juvenile myoclonic epilepsy: a coincidental occurrence?

Authors:  Salvatore Mangano; Rosaria Nardello; Gabriele Tripi; Giuliana Giordano; Chiara Spitaleri; Giuseppa Renata Mangano; Antonina Fontana
Journal:  BMC Neurol       Date:  2013-05-24       Impact factor: 2.474

6.  Hypertrichosis cubiti, a case report and literature review.

Authors:  Vivian E T Tng; Sally de Zwaan
Journal:  Clin Case Rep       Date:  2015-12-09

Review 7.  Congenital generalized hypertrichosis: the skin as a clue to complex malformation syndromes.

Authors:  Piero Pavone; Andrea D Praticò; Raffaele Falsaperla; Martino Ruggieri; Marcella Zollino; Giovanni Corsello; Giovanni Neri
Journal:  Ital J Pediatr       Date:  2015-08-05       Impact factor: 2.638

8.  Syringomyelia and Bone Malformations in the Setting of Hypertrichosis Cubiti (Hairy Elbow Syndrome).

Authors:  Vito Pavone; Gianluca Testa; Raffaele Falsaperla; Giuse Sessa
Journal:  J Orthop Case Rep       Date:  2015 Apr-Jun
  8 in total

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