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dc.contributor.author
Bottega, Roberta  
dc.contributor.author
Nicchia, Elena  
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Alfano, Caterina  
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Glembotsky, Ana Claudia  
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Pastore, Annalisa  
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Bertaggia Calderara, Debora  
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Bisig, Bettina  
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Duchosal, Michel A.  
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Arbesú, Guillermo  
dc.contributor.author
Alberio, Lorenzo  
dc.contributor.author
Heller, Paula Graciela  
dc.contributor.author
Savoia, Anna  
dc.date.available
2018-06-05T13:27:51Z  
dc.date.issued
2017-02  
dc.identifier.citation
Bottega, Roberta; Nicchia, Elena; Alfano, Caterina; Glembotsky, Ana Claudia; Pastore, Annalisa; et al.; Gray platelet syndrome: Novel mutations of the NBEAL2 gene; Wiley-liss, Div John Wiley & Sons Inc; American Journal Of Hematology; 92; 2; 2-2017; E20-E22  
dc.identifier.issn
0361-8609  
dc.identifier.uri
http://hdl.handle.net/11336/47272  
dc.description.abstract
Gray platelet syndrome (GPS) is a rare inherited macrothrombocytopenia characterized by reduction of a-granules in platelets and megakaryocytes associated with mild-tomoderate bleeding and myelofibrosis [1]. As reported in at least 28 unrelated families [1], GPS is caused by mutations of NBEAL2, the gene encoding for the neurobeachin-like-2 protein. NBEAL2 is a member of the family containing the BEACH (BEige And Chediak Higashi) domain, a conserved region involved in vesicular trafficking that may be critical for the a-granule development [2]. Here, we report novel mutations of NBEAL2 in two affected individuals (P1 and P2), who were previously diagnosed with immune thrombocytopenia (ITP) and then suspected to have GPS because of absence of azurophilic granules on May-Grunwald-Giemsa staining. In P1, sequencing analysis identified an € homozygous missense variant (c.6212G > C; p.Arg2071Pro; Supporting Information Fig. S1). Since her parents were not available for the segregation analysis, we hypothesised that the two mutant alleles were identical by descent because of homozygosity of all the polymorphic markers at the NBEAL2 locus (data not shown). Moreover, the potential hemizygous condition was excluded using statistical analyses of NBEAL2 amplicon coverage as previously reported (Supporting Information Fig. S1C) [3]. In P2, we detected one maternal nonsense (c.3839C > T; p.Arg1280*) and one paternal missense (c.6477C > G; p.His2159Gln; Supporting Information Fig. S1). The three NBEAL2 variants are reported in SNPs databases but with a minor allele frequency <0.01%.  
dc.format
application/pdf  
dc.language.iso
eng  
dc.publisher
Wiley-liss, Div John Wiley & Sons Inc  
dc.rights
info:eu-repo/semantics/openAccess  
dc.rights.uri
https://creativecommons.org/licenses/by-nc-sa/2.5/ar/  
dc.subject
Nbeal2  
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Gray Platelet Syndrome  
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Platelets  
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Mutations  
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Salud Ocupacional  
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Ciencias de la Salud  
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CIENCIAS MÉDICAS Y DE LA SALUD  
dc.title
Gray platelet syndrome: Novel mutations of the NBEAL2 gene  
dc.type
info:eu-repo/semantics/article  
dc.type
info:ar-repo/semantics/artículo  
dc.type
info:eu-repo/semantics/publishedVersion  
dc.date.updated
2018-06-04T17:20:45Z  
dc.journal.volume
92  
dc.journal.number
2  
dc.journal.pagination
E20-E22  
dc.journal.pais
Estados Unidos  
dc.journal.ciudad
Nueva York  
dc.description.fil
Fil: Bottega, Roberta. Università degli Studi di Trieste; Italia  
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Fil: Nicchia, Elena. Università degli Studi di Trieste; Italia  
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Fil: Alfano, Caterina. King's College London; Reino Unido  
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Fil: Glembotsky, Ana Claudia. Consejo Nacional de Investigaciones Científicas y Técnicas; Argentina. Universidad de Buenos Aires. Facultad de Medicina. Instituto de Investigaciones Médicas; Argentina  
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Fil: Pastore, Annalisa. King's College London; Reino Unido  
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Fil: Bertaggia Calderara, Debora. Universite de Lausanne; Suiza  
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Fil: Bisig, Bettina. Universite de Lausanne; Suiza  
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Fil: Duchosal, Michel A.. Universite de Lausanne; Suiza  
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Fil: Arbesú, Guillermo. Hospital Pediátrico Dr. Humberto Notti; Argentina  
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Fil: Alberio, Lorenzo. Universite de Lausanne; Suiza  
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Fil: Heller, Paula Graciela. Consejo Nacional de Investigaciones Científicas y Técnicas; Argentina. Universidad de Buenos Aires. Facultad de Medicina. Instituto de Investigaciones Médicas; Argentina  
dc.description.fil
Fil: Savoia, Anna. Università degli Studi di Trieste; Italia. Institute for Maternal and Child Health; Italia  
dc.journal.title
American Journal Of Hematology  
dc.relation.alternativeid
info:eu-repo/semantics/altIdentifier/doi/https://dx.doi.org/10.1002/ajh.24610  
dc.relation.alternativeid
info:eu-repo/semantics/altIdentifier/url/https://onlinelibrary.wiley.com/doi/abs/10.1002/ajh.24610