| Literature DB >> 24735966 |
Katelyn E Gagne1, Roxanne Ghazvinian2, Daniel Yuan2, Rebecca L Zon1, Kelsie Storm3, Magdalena Mazur-Popinska4, Laura Andolina5, Halina Bubala6, Sydonia Golebiowska7, Meghan A Higman5, Krzysztof Kalwak4, Peter Kurre3, Michal Matysiak7, Edyta Niewiadomska7, Salley Pels8, Mary Jane Petruzzi5, Aneta Pobudejska-Pieniazek6, Tomasz Szczepanski6, Mark D Fleming9, Hanna T Gazda10, Suneet Agarwal11.
Abstract
Pearson marrow pancreas syndrome (PS) is a multisystem disorder caused by mitochondrial DNA (mtDNA) deletions. Diamond-Blackfan anemia (DBA) is a congenital hypoproliferative anemia in which mutations in ribosomal protein genes and GATA1 have been implicated. Both syndromes share several features including early onset of severe anemia, variable nonhematologic manifestations, sporadic genetic occurrence, and occasional spontaneous hematologic improvement. Because of the overlapping features and relative rarity of PS, we hypothesized that some patients in whom the leading clinical diagnosis is DBA actually have PS. Here, we evaluated patient DNA samples submitted for DBA genetic studies and found that 8 (4.6%) of 173 genetically uncharacterized patients contained large mtDNA deletions. Only 2 (25%) of the patients had been diagnosed with PS on clinical grounds subsequent to sample submission. We conclude that PS can be overlooked, and that mtDNA deletion testing should be performed in the diagnostic evaluation of patients with congenital anemia.Entities:
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Year: 2014 PMID: 24735966 PMCID: PMC4102714 DOI: 10.1182/blood-2014-01-545830
Source DB: PubMed Journal: Blood ISSN: 0006-4971 Impact factor: 22.113