Literature DB >> 24138047

The hermansky-pudlak syndrome: clinical features and imperatives from an ophthalmic perspective.

Andrew J Schneier1, Anne B Fulton.   

Abstract

The Hermansky-Pudlak Syndrome (HPS) is a rare, autosomal recessive condition comprising nine genetically heterogeneous entities that feature oculocutaneous albinism (OCA) and bleeding tendency as their principal clinical manifestations. The pathogenesis of HPS involves disturbances in the biogenesis and trafficking of lysosome-related organelles. While the ophthalmologist is trained to address the ocular manifestations of OCA, it is critical for the provider to consider HPS when examining OCA patients as its systemic sequelae may be associated with morbidity and mortality. If there is suspicion of HPS in a patient with albinism, the ophthalmologist should enlist the aid of consultants to confirm the diagnosis and monitor for systemic features. As the nine HPS subtypes explored in this article vary widely in the character and severity of their associated systemic manifestations, some authors advocate determining the specific gene defect in each HPS patient in order to optimize care and provide anticipatory guidance.

Entities:  

Mesh:

Year:  2013        PMID: 24138047     DOI: 10.3109/08820538.2013.825280

Source DB:  PubMed          Journal:  Semin Ophthalmol        ISSN: 0882-0538            Impact factor:   1.975


  4 in total

1.  Refractive errors, visual impairment, and the use of low-vision devices in albinism in Malawi.

Authors:  M Schulze Schwering; N Kumar; D Bohrmann; G Msukwa; K Kalua; P Kayange; M S Spitzer
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  2015-02-12       Impact factor: 3.117

2.  Membrane-Associated Transporter Protein (MATP) Regulates Melanosomal pH and Influences Tyrosinase Activity.

Authors:  Bum-Ho Bin; Jinhyuk Bhin; Seung Ha Yang; Misun Shin; Yeon-Ju Nam; Dong-Hwa Choi; Dong Wook Shin; Ai-Young Lee; Daehee Hwang; Eun-Gyung Cho; Tae Ryong Lee
Journal:  PLoS One       Date:  2015-06-09       Impact factor: 3.240

3.  Hermansky-Pudlak syndrome: Mutation update.

Authors:  Marjan Huizing; May C V Malicdan; Jennifer A Wang; Hadass Pri-Chen; Richard A Hess; Roxanne Fischer; Kevin J O'Brien; Melissa A Merideth; William A Gahl; Bernadette R Gochuico
Journal:  Hum Mutat       Date:  2020-01-23       Impact factor: 4.700

4.  Whole-Exome Sequencing Identified a Novel Homozygous Frameshift Mutation of HPS3 in a Consanguineous Family with Hermansky-Pudlak Syndrome.

Authors:  Zhao-Xia Wang; Yi-Hui Liu; Yi Dong; Ya-Li Li; Tie-Yu Tang; Liang-Liang Fan
Journal:  Biomed Res Int       Date:  2021-09-24       Impact factor: 3.411

  4 in total

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