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伴有嗜中性皮病的与 Pyrin 相关的自身炎症:一例报告。

Pyrin-associated autoinflammation with neutrophilic dermatosis: A case report.

作者信息

Bueno-Molina Rocío C, Hernández-Rodríguez Juan-Carlos, Zulueta-Dorado Teresa, Pereyra-Rodriguez Jose-Juan

机构信息

Department of Dermatology, Virgen del Rocio University Hospital, Seville, Spain.

Department of Pathological Anatomy, Virgen del Rocio University Hospital, Seville, Spain.

出版信息

J Dermatol. 2024 Dec;51(12):1702-1706. doi: 10.1111/1346-8138.17390. Epub 2024 Jul 28.

Abstract

Pyrin-associated autoinflammation with neutrophilic dermatosis (PAAND) is a rare, monogenic, autoinflammatory disorder caused by mutations in exon 2 of the MEFV gene. Characterized by neutrophilic dermatosis, recurrent fever, and arthralgia, this syndrome presents a diagnostic challenge due to its low prevalence and varied clinical manifestations. Here, we present the case of a 49-year-old Spanish male with severe hidradenitis suppurativa and pyoderma gangrenosum with a heterozygous variant (p.E244K) in the MEFV gene, consistent with PAAND syndrome. This variant has only been documented in one other case with notable similarities. Both patients share Spanish ancestry and present a severe form of hidradenitis suppurativa. Treatment of the disorder presents challenges due to its variable response to standard therapies. Anti-interleukin-1 agents, such as anakinra or anti-tumor necrosis factor (TNF)-α are the therapeutic approaches supported by the most substantial evidence. Our findings highlight the importance of genetic evaluation of MEFV mutations in individuals with neutrophilic dermatosis and systemic symptoms.

摘要

伴有嗜中性皮肤病的吡啉相关自身炎症(PAAND)是一种罕见的单基因自身炎症性疾病,由MEFV基因第2外显子的突变引起。该综合征以嗜中性皮肤病、反复发热和关节痛为特征,因其患病率低且临床表现多样,给诊断带来了挑战。在此,我们报告一例49岁的西班牙男性病例,该患者患有严重的化脓性汗腺炎和坏疽性脓皮病,其MEFV基因存在杂合变异(p.E244K),符合PAAND综合征。此变异仅在另一例有显著相似之处的病例中被记录过。两名患者均有西班牙血统,且均表现为严重形式的化脓性汗腺炎。由于该疾病对标准疗法的反应不一,其治疗颇具挑战。抗白细胞介素-1药物,如阿那白滞素或抗肿瘤坏死因子(TNF)-α是有最充分证据支持的治疗方法。我们的研究结果强调了对患有嗜中性皮肤病和全身症状的个体进行MEFV基因突变基因评估的重要性。

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