Literature DB >> 8491872

Trichothiodystrophy associated with photosensitivity, gonadal failure, and striking osteosclerosis.

C McCuaig1, D Marcoux, J E Rasmussen, M M Werner, N E Gentner.   

Abstract

Three new unrelated cases of PIBIDS (Photosensitivity, Ichthyosis, Brittle, sulfur-deficient hair [trichothiodystrophy], Impaired intelligence, Decreased fertility, and Short stature) are reported. Decreased survival of skin fibroblast lines after UVB exposure was found. All three male patients had hypogonadism and primary end-organ gonadal failure. Striking osteosclerosis was present in all three patients. To the best of our knowledge the third patient is the first reported case of a black man with PIBIDS.

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Year:  1993        PMID: 8491872     DOI: 10.1016/0190-9622(93)70109-7

Source DB:  PubMed          Journal:  J Am Acad Dermatol        ISSN: 0190-9622            Impact factor:   11.527


  8 in total

1.  Cerebellar and cerebral atrophy in trichothiodystrophy.

Authors:  Hye-Kyung Yoon; Michael A Sargent; Julie S Prendiville; Kenneth J Poskitt
Journal:  Pediatr Radiol       Date:  2005-05-24

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.  Central nervous system dysmyelination in PIBI(D)S syndrome: a further case.

Authors:  P A Battistella; A Peserico
Journal:  Childs Nerv Syst       Date:  1996-02       Impact factor: 1.475

Review 4.  Premature aging and cancer in nucleotide excision repair-disorders.

Authors:  K Diderich; M Alanazi; J H J Hoeijmakers
Journal:  DNA Repair (Amst)       Date:  2011-06-15

5.  Central osteosclerosis with trichothiodystrophy.

Authors:  Emma L Wakeling; Michele Cruwys; Mohnish Suri; Angela F Brady; Sarah E Aylett; Christine Hall
Journal:  Pediatr Radiol       Date:  2004-05-18

6.  Subcutaneous calcification as a delayed complication of radiotherapy: a case report and review of the literature.

Authors:  Zoltán Zaka; János Fodor; Nóra Udvarhelyi; Zsolt Orosz; Miklós Kásler
Journal:  Pathol Oncol Res       Date:  2008-08-28       Impact factor: 3.201

7.  Bone fragility and decline in stem cells in prematurely aging DNA repair deficient trichothiodystrophy mice.

Authors:  Karin E M Diderich; Claudia Nicolaije; Matthias Priemel; Jan H Waarsing; Judd S Day; Renata M C Brandt; Arndt F Schilling; Sander M Botter; Harrie Weinans; Gijsbertus T J van der Horst; Jan H J Hoeijmakers; Johannes P T M van Leeuwen
Journal:  Age (Dordr)       Date:  2011-08-04

8.  Trichothiodystrophy with dysmyelination and central osteosclerosis.

Authors:  J H Harreld; E C Smith; N S Prose; P K Puri; D P Barboriak
Journal:  AJNR Am J Neuroradiol       Date:  2010-01       Impact factor: 4.966

  8 in total

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