Literature DB >> 3770739

Xeroderma pigmentosum (complementation group D) mutation is present in patients affected by trichothiodystrophy with photosensitivity.

M Stefanini, P Lagomarsini, C F Arlett, S Marinoni, C Borrone, F Crovato, G Trevisan, G Cordone, F Nuzzo.   

Abstract

We studied the response to UV irradiation in cells from four patients, from three apparently unrelated families, affected by trichothiodystrophy (TTD). They showed all the symptoms of this rare autosomal recessive disorder (brittle hair with reduced sulfur content, mental and physical retardation, ichthyosis, peculiar face) together with photosensitivity. We found a decreased rate of duplicative DNA synthesis in stimulated lymphocytes, reduced survival in fibroblasts, and very low levels of unscheduled DNA synthesis (UDS) in Go lymphocytes and fibroblasts after UV irradiation. Complementation studies showed that normal values of UDS are restored in heterokaryons obtained by fusion of TTD cells with normal and xeroderma pigmentosum (XP)-complementation group A-cells. In contrast the defect is not complemented by fusion with XP-complementation group D-fibroblasts.

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Year:  1986        PMID: 3770739     DOI: 10.1007/bf00282072

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  34 in total

1.  Ichthyosiform erythroderma, hair shaft abnormalities, and mental and growth retardation. A new recessive disorder.

Authors:  C H Tay
Journal:  Arch Dermatol       Date:  1971-07

2.  Sibs with mental and physical retardation and trichorrhexis nodosa with abnormal amino acid composition of the hair.

Authors:  R J Pollitt; F A Jenner; M Davies
Journal:  Arch Dis Child       Date:  1968-04       Impact factor: 3.791

3.  PIBI(D)S syndrome: a new entity with defect of the deoxyribonucleic acid excision repair system.

Authors:  F Crovato; A Rebora
Journal:  J Am Acad Dermatol       Date:  1985-10       Impact factor: 11.527

4.  Trichothiodystrophy: sulfur-deficient brittle hair as a marker for a neuroectodermal symptom complex.

Authors:  V H Price; R B Odom; W H Ward; F T Jones
Journal:  Arch Dermatol       Date:  1980-12

5.  Ichthyosis, brittle hair, impaired intelligence, decreased fertility and short stature (IBIDS syndrome).

Authors:  J L Jorizzo; D J Atherton; R G Crounse; R S Wells
Journal:  Br J Dermatol       Date:  1982-06       Impact factor: 9.302

6.  Cockayne's syndrome fibroblasts have increased sensitivity to ultraviolet light but normal rates of unscheduled DNA synthesis.

Authors:  A D Andrews; S F Barrett; F W Yoder; J H Robbins
Journal:  J Invest Dermatol       Date:  1978-05       Impact factor: 8.551

7.  The Tay syndrome (congenital ichthyosis with trichothiodystrophy).

Authors:  R Happle; H Traupe; H Gröbe; G Bonsmann
Journal:  Eur J Pediatr       Date:  1984-01       Impact factor: 3.183

8.  Low-sulfur hair syndrome associated with UVB photosensitivity and testicular failure.

Authors:  P A Lucky; N Kirsch; A W Lucky; D M Carter
Journal:  J Am Acad Dermatol       Date:  1984-08       Impact factor: 11.527

9.  The Sabinas syndrome.

Authors:  R R Howell; A I Arbisser; D S Parsons; C I Scott; U Fraustadt; W R Collie; R N Marshall; O C Ibarra
Journal:  Am J Hum Genet       Date:  1981-11       Impact factor: 11.025

10.  Trichothiodystrophy-neurotrichocutaneous syndrome of Pollitt: a report of two unrelated cases.

Authors:  M D King; C L Gummer; J B Stephenson
Journal:  J Med Genet       Date:  1984-08       Impact factor: 6.318

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  24 in total

1.  A variant of the Nijmegen breakage syndrome with unusual cytogenetic features and intermediate cellular radiosensitivity.

Authors:  R Tupler; G L Marseglia; M Stefanini; E Prosperi; L Chessa; T Nardo; A Marchi; P Maraschio
Journal:  J Med Genet       Date:  1997-03       Impact factor: 6.318

2.  Ocular manifestations of trichothiodystrophy.

Authors:  Brian P Brooks; Amy H Thompson; Janine A Clayton; Chi-Chao Chan; Deborah Tamura; Wadih M Zein; Delphine Blain; Casey Hadsall; John Rowan; Kristen E Bowles; Sikandar G Khan; Takahiro Ueda; Jennifer Boyle; Kyu-Seon Oh; John J DiGiovanna; Kenneth H Kraemer
Journal:  Ophthalmology       Date:  2011-09-28       Impact factor: 12.079

Review 3.  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

4.  Persistence of repair proteins at unrepaired DNA damage distinguishes diseases with ERCC2 (XPD) mutations: cancer-prone xeroderma pigmentosum vs. non-cancer-prone trichothiodystrophy.

Authors:  Jennifer Boyle; Takahiro Ueda; Kyu-Seon Oh; Kyoko Imoto; Deborah Tamura; Jared Jagdeo; Sikandar G Khan; Carine Nadem; John J Digiovanna; Kenneth H Kraemer
Journal:  Hum Mutat       Date:  2008-10       Impact factor: 4.878

5.  A mutation in the XPB/ERCC3 DNA repair transcription gene, associated with trichothiodystrophy.

Authors:  G Weeda; E Eveno; I Donker; W Vermeulen; O Chevallier-Lagente; A Taïeb; A Stary; J H Hoeijmakers; M Mezzina; A Sarasin
Journal:  Am J Hum Genet       Date:  1997-02       Impact factor: 11.025

6.  Bi-allelic TARS Mutations Are Associated with Brittle Hair Phenotype.

Authors:  Arjan F Theil; Elena Botta; Anja Raams; Desiree E C Smith; Marisa I Mendes; Giuseppina Caligiuri; Sarah Giachetti; Silvia Bione; Roberta Carriero; Giordano Liberi; Luca Zardoni; Sigrid M A Swagemakers; Gajja S Salomons; Alain Sarasin; Alan Lehmann; Peter J van der Spek; Tomoo Ogi; Jan H J Hoeijmakers; Wim Vermeulen; Donata Orioli
Journal:  Am J Hum Genet       Date:  2019-08-01       Impact factor: 11.025

7.  Trichothiodystrophy, xeroderma pigmentosum and PIBI(D)S syndrome.

Authors:  A Rebora; F Crovato
Journal:  Hum Genet       Date:  1988-01       Impact factor: 4.132

8.  A new nucleotide-excision-repair gene associated with the disorder trichothiodystrophy.

Authors:  M Stefanini; W Vermeulen; G Weeda; S Giliani; T Nardo; M Mezzina; A Sarasin; J I Harper; C F Arlett; J H Hoeijmakers
Journal:  Am J Hum Genet       Date:  1993-10       Impact factor: 11.025

Review 9.  Diseases associated with defective responses to DNA damage.

Authors:  Mark O'Driscoll
Journal:  Cold Spring Harb Perspect Biol       Date:  2012-12-01       Impact factor: 10.005

10.  Immune defects in families and patients with xeroderma pigmentosum and trichothiodystrophy.

Authors:  E Mariani; A Facchini; M C Honorati; E Lalli; E Berardesca; P Ghetti; S Marinoni; F Nuzzo; G C Astaldi Ricotti; M Stefanini
Journal:  Clin Exp Immunol       Date:  1992-06       Impact factor: 4.330

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