Literature DB >> 2126369

The Hermansky-Pudlak syndrome. Variable reaction to 1-desamino-8D-arginine vasopressin for correction of the bleeding time.

D B Van Dorp1, P W Wijermans, F Meire, G Vrensen.   

Abstract

The effect of the synthetic vasopressin derivative 1-desamino-8D-arginine vasopressin (DDAVP = Minrin) on bleeding time was studied in nine patients with Hermansky-Pudlak syndrome; four of them were Dutch, five were Belgian. Shortening of bleeding time was observed in four of the patients with this type of storage pool disease; in one patient the response was equivocal, in two patients the response was not dramatic and in two there was no response at all. DDAVP may be useful in managing the bleeding disorder in some patients with Hermansky-Pudlak syndrome. Therefore, every patient with this syndrome should be tested with DDAVP as a preventive measure.

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Year:  1990        PMID: 2126369     DOI: 10.3109/13816819009020985

Source DB:  PubMed          Journal:  Ophthalmic Paediatr Genet        ISSN: 0167-6784


  4 in total

1.  Hermansky-pudlak syndrome: report of a case and review of the literature.

Authors:  Matthew T Hurford; Christopher Sebastiano
Journal:  Int J Clin Exp Pathol       Date:  2008-01-01

2.  A rare cause of thrombocyte dysfunction: Hermansky-Pudlak syndrome.

Authors:  Nihal Özdemir; Emre Çelik; Zafer Başlar; Tiraje Celkan
Journal:  Turk Pediatri Ars       Date:  2014-06-01

3.  Defective AP-3-dependent VAMP8 trafficking impairs Weibel-Palade body exocytosis in Hermansky-Pudlak Syndrome type 2 blood outgrowth endothelial cells.

Authors:  Ellie Karampini; Maaike Schillemans; Menno Hofman; Floris van Alphen; Martin de Boer; Taco W Kuijpers; Maartje van den Biggelaar; Jan Voorberg; Ruben Bierings
Journal:  Haematologica       Date:  2019-01-10       Impact factor: 9.941

4.  BLOC-2 subunit HPS6 deficiency affects the tubulation and secretion of von Willebrand factor from mouse endothelial cells.

Authors:  Jing Ma; Zhe Zhang; Lin Yang; Janos Kriston-Vizi; Daniel F Cutler; Wei Li
Journal:  J Genet Genomics       Date:  2016-10-21       Impact factor: 4.275

  4 in total

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