伴有嗜中性粒细胞皮肤病的 Pyrin 相关自身炎症:病例报告

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

嗜中性粒细胞皮炎(Pyrin-associated autoinflammation with neutrophilic dermatosis,PAAND)是一种罕见的单基因自身炎症性疾病,由 MEFV 基因第 2 外显子突变引起。该综合征以嗜中性粒细胞皮肤病、反复发热和关节痛为特征,由于发病率低且临床表现多样,因此给诊断带来了挑战。在此,我们介绍了一例 49 岁的西班牙男性患者,他患有严重的化脓性扁桃体炎和脓皮病,其 MEFV 基因存在杂合变异(p.E244K),与 PAAND 综合征一致。该变异体仅在另一例病例中出现过,且具有显著的相似性。这两名患者都有西班牙血统,并患有严重的化脓性扁桃体炎。这种疾病对标准疗法的反应不一,给治疗带来了挑战。抗白细胞介素-1药物,如阿那金拉(anakinra)或抗肿瘤坏死因子(TNF)-α,是目前证据最充分的治疗方法。我们的研究结果突显了对患有嗜中性皮肤病和全身症状的个体进行MEFV基因突变遗传评估的重要性。
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Pyrin-associated autoinflammation with neutrophilic dermatosis: A case report.

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.

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