| Literature DB >> 30018023 |
Alessandra Fierabracci1, Marsha Pellegrino2, Federica Frasca3, Sara Sebnem Kilic4, Corrado Betterle5.
Abstract
APECED is a rare monogenic recessive disorder caused by mutations in the AIRE gene. In this manuscript, we report a male Turkish patient with APECED syndrome who presented with chronic mucocutaneous candidiasis associated with other autoimmune manifestations developed over the years. The presence of the homozygous R257X mutation of the AIRE gene confirmed the diagnosis of APECED syndrome. We further performed literature review in 23 published Turkish APECED patients and noted that Finnish major mutation R257X is common in Turks. In particular, we assessed retrospectively how often the Ferre/Lionakis criteria would have resulted in earlier diagnosis in Finns, Sardinians and Turks in respect to the classic criteria. Since an earlier diagnosis could have been possible in 18.8% of Turkish, in 23.8% of Sardinian and 38.55% of Finnish patients we reviewed from literature, Ferre/Lionakis criteria could indeed allow in future earlier initiation of immunomodulatory treatments, if found effective in future studies.Entities:
Keywords: APECED-AIRE gene-genotype-phenotype variability-Turkish population
Mesh:
Year: 2018 PMID: 30018023 DOI: 10.1016/j.clim.2018.06.012
Source DB: PubMed Journal: Clin Immunol ISSN: 1521-6616 Impact factor: 3.969