Mendonca Leonardo Oliveira, Prado Alex Isidoro, Costa Izelda Maria Carvalho, Bandeira Marcia, Dyer Rafael, Barros Samar Freschi, Khöler Karen Francine, Fonseca Luiz Augusto Marcondes, Kalil Jorge, Castro Fabio Morato, Toledo-Barros Myrthes Anna Maragna
Clinical Immunology and Allergy, School of Medicine, University of São Paulo, São Paulo, Brazil.
Laboratory for Immunological Investigation (LIM-19), Heart Institute, University of São Paulo, São Paulo, Brazil.
Front Immunol. 2021 Apr 14;12:586320. doi: 10.3389/fimmu.2021.586320. eCollection 2021.
Since the first description of the syndrome of sideroblastic anemia with immunodeficiency, fevers and development delay (SIFD), clinical pictures lacking both neurological and hematological manifestations have been reported. Moreover, prominent skin involvement, such as with relapsing erythema nodosum, is not a common finding. Up to this moment, no genotype and phenotype correlation could be done, but mild phenotypes seem to be located in the N or C part. B-cell deficiency is a hallmark of SIFD syndrome, and multiple others immunological defects have been reported, but not high levels of double negative T cells. Here we report a Brazilian patient with a novel phenotype of SFID syndrome, carrying multiple immune defects and harboring a novel mutation on TRNT1 gene.
自从首次描述伴有免疫缺陷、发热和发育迟缓的铁粒幼细胞贫血综合征(SIFD)以来,已报告了缺乏神经和血液学表现的临床症状。此外,突出的皮肤受累情况,如复发性结节性红斑,并不常见。到目前为止,尚无法建立基因型与表型的相关性,但轻度表型似乎位于N端或C端。B细胞缺陷是SIFD综合征的一个标志,并且已报告了多种其他免疫缺陷,但未发现高水平的双阴性T细胞。在此,我们报告一名患有新型SIFD综合征表型的巴西患者,其携带多种免疫缺陷并在TRNT1基因上存在一个新的突变。