Literature DB >> 29641717

Do you know this syndrome? Ichthyosis associated with neurological condition and alteration of hairs.

Luciana Baptista Pereira1, Neusa Yuriko Sakai Valente2, Vanessa Barreto Rocha1.   

Abstract

Trichothiodystrophy refers to a heterogeneous group of rare genetic diseases that affects neuroectodermal-derived tissues with multisystem involvement. The hallmark of these syndromes is the deficiency of sulfur in hair matrix proteins, leading to short and brittle hair. Few cases of this rare disorder have been published. The authors report a case of trichothiodystrophy in a male infant with ichthyosis, photosensitivity, spastic paraparesis, short stature, and neurologic and psychomotor retardation. Diagnosis was based on clinical and microscopic features of hair samples.

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Year:  2018        PMID: 29641717      PMCID: PMC5871382          DOI: 10.1590/abd1806-4841.20187727

Source DB:  PubMed          Journal:  An Bras Dermatol        ISSN: 0365-0596            Impact factor:   1.896


  7 in total

1.  Photosensitive form of trichothiodystrophy associated with a novel mutation in the XPD gene.

Authors:  Birka Brauns; Steffen Schubert; Janin Lehmann; Petra Laspe; Andrea Körner; Knut Brockmann; Michael P Schön; Steffen Emmert
Journal:  Photodermatol Photoimmunol Photomed       Date:  2015-12-15       Impact factor: 3.135

Review 2.  Trichothiodystrophy: a systematic review of 112 published cases characterises a wide spectrum of clinical manifestations.

Authors:  S Faghri; D Tamura; K H Kraemer; J J Digiovanna
Journal:  J Med Genet       Date:  2008-06-25       Impact factor: 6.318

3.  Brittle Hair, Photosensitivity, Brain Hypomyelination and Immunodeficiency: Clues to Trichothiodystrophy.

Authors:  Evangelia Farmaki; Natalia Nedelkopoulou; Florentina Delli; Kosmas Sarafidis; Dimitrios I Zafeiriou
Journal:  Indian J Pediatr       Date:  2016-07-30       Impact factor: 1.967

Review 4.  Understanding photodermatoses associated with defective DNA repair: Photosensitive syndromes without associated cancer predisposition.

Authors:  Yik Weng Yew; Cerrene N Giordano; Graciela Spivak; Henry W Lim
Journal:  J Am Acad Dermatol       Date:  2016-11       Impact factor: 11.527

Review 5.  Trichothiodystrophy: update on the sulfur-deficient brittle hair syndromes.

Authors:  P H Itin; A Sarasin; M R Pittelkow
Journal:  J Am Acad Dermatol       Date:  2001-06       Impact factor: 11.527

Review 6.  Trichothiodystrophy: from basic mechanisms to clinical implications.

Authors:  M Stefanini; E Botta; M Lanzafame; D Orioli
Journal:  DNA Repair (Amst)       Date:  2010-01-02

7.  [Trichothiodystrophy: PIBIDS syndrome].

Authors:  R Jiménez-Puya; J C Moreno-Giménez; F Camacho-Martínez; J Ferrando-Barbera; R Grimalt
Journal:  Actas Dermosifiliogr       Date:  2007-04
  7 in total
  1 in total

1.  PIBIDS syndrome in two Brazilian siblings.

Authors:  Kerstin Taniguchi Abagge; Felipe Haupenthal; Gabriella Yamashita Felber; Salmo Raskin
Journal:  BMJ Case Rep       Date:  2018-12-22
  1 in total

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