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DOI 10.1111/ejh.12596

Медицинская библиотека · European journal of haematology · 2015

A multicentre observational study for early diagnosis of Gaucher disease in patients with Splenomegaly and/or Thrombocytopenia.

Motta Irene, Filocamo Mirella, Poggiali Erika, Stroppiano Marina, Dragani Alfredo, Consonni Dario, Barcellini Wilma, Gaidano Gianluca, Facchini Luca, Specchia Giorgina, Cappellini Maria Domenica, Splenomegaly Gaucher Disease study group

СтатьяМногоцентровое исследованиеНаблюдательное исследованиеПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

Gaucher disease (GD) is the most common lysosomal disorder resulting from deficient activity of the β-glucosidase enzyme that causes accumulation of glucosylceramide in the macrophage-monocyte system. Notably, because of non-specific symptoms and a lack of awareness, patients with GD experience long diagnostic delays. The aim of this study was to apply a diagnostic algorithm to identify GD type 1 among adults subjects referred to Italian haematology outpatient units because of splenomegaly and/or thrombocytopenia and, eventually, to estimate the prevalence of GD in this selected population. One hundred and ninety-six subjects (61 females, 135 males; mean age 47.8 ± 18.2 years) have been enrolled in the study and tested for β-glucosidase enzyme activity on dried blood spot (DBS). Seven of 196 patients have been diagnosed with GD, (5 females and 2 males) with mean age 31.8 ± 8.2 years, with a prevalence of 3.6% (with a prevalence of 3.6% (I95% CI 1.4-7.2; 1/28 patients) in this population. These results show that the use of an appropriate diagnostic algorithm and a simple diagnostic method, such as DBS, are important tools to facilitate the diagnosis of a rare disease even for not disease-expert physicians.

Для цитирования: Motta Irene, Filocamo Mirella, Poggiali Erika, Stroppiano Marina, Dragani Alfredo, Consonni Dario, Barcellini Wilma, Gaidano Gianluca, Facchini Luca, Specchia Giorgina, Cappellini Maria Domenica, Splenomegaly Gaucher Disease study group. A multicentre observational study for early diagnosis of Gaucher disease in patients with Splenomegaly and/or Thrombocytopenia.. European journal of haematology. 2015. DOI: 10.1111/ejh.12596.

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