Blood · 2014
Pearson marrow pancreas syndrome in patients suspected to have Diamond-Blackfan anemia.
СтатьяПоддержка NIH: внешняя программаПоддержка исследования: вне правительства СШАНазвание и аннотация: Перевод пока не подготовлен
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Gagne Katelyn E, Ghazvinian Roxanne, Yuan Daniel, Zon Rebecca L, Storm Kelsie, Mazur-Popinska Magdalena, Andolina Laura, Bubala Halina, Golebiowska Sydonia, Higman Meghan A, Kalwak Krzysztof, Kurre Peter, Matysiak Michal, Niewiadomska Edyta, Pels Salley, Petruzzi Mary Jane, Pobudejska-Pieniazek Aneta, Szczepanski Tomasz, Fleming Mark D, Gazda Hanna T, Agarwal Suneet
Аннотация
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.