Literature DB >> 26078655

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

Nihal Özdemir1, Emre Çelik1, Zafer Başlar2, Tiraje Celkan1.   

Abstract

Hermansky-Pudlak syndrome (HPS) is an autosomal recessive disease characterized with oculocutaneous albinism and platelet aggregation disorder. The clinical picture is highly variable and patients may present with different complaints., Ecchymoses usually develop in childhood; epistaxis, postoperative hemorrhage and gingival bleeding may be observed. Here we present 3 patients who were diagnosed with HPS in our clinic. In conclusion, HPS should be considered in all patients with ocular findings and albinism even though bleeding disorder is not described. Bleeding findings may be mild or unrecognized. Absence of secondary wave in platalet aggregation tests supports the diagnosis.

Entities:  

Keywords:  Hermnasky-Pudlack syndrome; bleeding disorder; thrombocyte dysfunction

Year:  2014        PMID: 26078655      PMCID: PMC4462281          DOI: 10.5152/tpa.2014.1071

Source DB:  PubMed          Journal:  Turk Pediatri Ars


  9 in total

1.  Albinism associated with hemorrhagic diathesis and unusual pigmented reticular cells in the bone marrow: report of two cases with histochemical studies.

Authors:  F HERMANSKY; P PUDLAK
Journal:  Blood       Date:  1959-02       Impact factor: 22.113

2.  Albinism and Hermansky-Pudlak syndrome in Puerto Rico.

Authors:  C J Witkop; M Nuñez Babcock; G H Rao; F Gaudier; C G Summers; F Shanahan; K R Harmon; D Townsend; H O Sedano; R A King
Journal:  Bol Asoc Med P R       Date:  1990-08

3.  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

4.  Successful bilateral lung transplantation for pulmonary fibrosis associated with the Hermansky-Pudlak syndrome.

Authors:  David J Lederer; Steven M Kawut; Joshua R Sonett; Efsevia Vakiani; Samuel L Seward; James G White; Jessie S Wilt; Charles C Marboe; William A Gahl; Selim M Arcasoy
Journal:  J Heart Lung Transplant       Date:  2005-10       Impact factor: 10.247

Review 5.  Hermansky-Pudlak syndrome: a disease of protein trafficking and organelle function.

Authors:  Maria L Wei
Journal:  Pigment Cell Res       Date:  2006-02

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

Authors:  D B Van Dorp; P W Wijermans; F Meire; G Vrensen
Journal:  Ophthalmic Paediatr Genet       Date:  1990-09

Review 7.  Congenital disorders associated with platelet dysfunctions.

Authors:  Paquita Nurden; Alan T Nurden
Journal:  Thromb Haemost       Date:  2008-02       Impact factor: 5.249

8.  Lethal hemophagocytic lymphohistiocytosis in Hermansky-Pudlak syndrome type II.

Authors:  Anselm Enders; Barbara Zieger; Klaus Schwarz; Ayami Yoshimi; Carsten Speckmann; Eva-Maria Knoepfle; Udo Kontny; Christoph Müller; Alan Nurden; Jan Rohr; Matthias Henschen; Ulrich Pannicke; Charlotte Niemeyer; Paquita Nurden; Stephan Ehl
Journal:  Blood       Date:  2006-03-21       Impact factor: 22.113

9.  Successful thyroidectomy in a patient with Hermansky-Pudlak syndrome treated with recombinant activated factor VII and platelet concentrates.

Authors:  A I del Pozo Pozo; V Jiménez-Yuste; A Villar; M Quintana; F Hernández-Navarro
Journal:  Blood Coagul Fibrinolysis       Date:  2002-09       Impact factor: 1.276

  9 in total
  2 in total

1.  Bleeding assessment in female patients with the Hermansky-Pudlak syndrome-A case series.

Authors:  Joel Rivera-Concepción; Jorge Acevedo-Canabal; Antonio Burés; Gustavo Vargas; Carmen Cadilla; Natalio J Izquierdo
Journal:  Eur J Haematol       Date:  2019-03-06       Impact factor: 2.997

2.  Unexpected intra-operative bleeding due to Hermansky-Pudlak Syndrome.

Authors:  Mustafa Ozgur; Bahar Yilmaz
Journal:  Indian J Anaesth       Date:  2015-06
  2 in total

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