Romagnuolo Maurizio, Moltrasio Chiara, Iannone Claudia, Gattinara Maurizio, Cambiaghi Stefano, Marzano Angelo Valerio
Dermatology Unit, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.
Department of Pathophysiology and Transplantation, Università degli Studi di Milano, Milan, Italy.
Front Med (Lausanne). 2023 May 31;10:1197273. doi: 10.3389/fmed.2023.1197273. eCollection 2023.
Chronic recurrent multifocal osteomyelitis (CRMO) is a rare autoinflammatory disease, clinically characterized by chronic and recurrent episodes of osteoarticular inflammation, that generally presents in children and adolescents. From a dermatological point-of-view, CMRO can be associated with skin rashes mainly including psoriasis, palmoplantar pustulosis and acne. Pyoderma gangrenosum (PG) is a rare immune-mediated inflammatory skin disease classified within the spectrum of neutrophilic dermatoses that, in some cases, has been reported as cutaneous manifestation in CMRO patients. This paper presents a 16-year female patient diagnosed with CMRO, who presented PG lesions located on the lower leg, that arose after the administration of the tumour necrosis factor (TNF)-α inhibitor adalimumab. Cases of PG have been reported in patients being treated with certain medications, including TNF-α antagonists, leading to classified them in a setting aptly termed "drug-induced PG." In this paper, we discuss the co-occurrence of PG and CRMO, in the light of recent evidence on the pathogenesis of both diseases and giving ample space to a literature review on drug induced PG. In our case, it is plausible that PG could be considered a cutaneous manifestation of CRMO, although the mechanisms underlying this intriguingly relationship remain to be fully unraveled.
慢性复发性多灶性骨髓炎（CRMO）是一种罕见的自身炎症性疾病，临床特征为骨关节炎症的慢性反复发作，多见于儿童和青少年。从皮肤病学角度来看，CRMO可伴有皮疹，主要包括银屑病、掌跖脓疱病和痤疮。坏疽性脓皮病（PG）是一种罕见的免疫介导性炎症性皮肤病，属于嗜中性皮肤病范畴，在某些情况下，已被报道为CRMO患者的皮肤表现。本文介绍了一名16岁诊断为CRMO的女性患者，其小腿出现PG皮损，该皮损在使用肿瘤坏死因子（TNF）-α抑制剂阿达木单抗后出现。已有PG病例在接受某些药物治疗的患者中被报道，包括TNF-α拮抗剂，这导致将其归类于“药物性PG”这一恰当命名的情况。在本文中，我们根据这两种疾病发病机制的最新证据，对PG和CRMO的共现情况进行了讨论，并对药物性PG的文献综述给予了充分篇幅。在我们的病例中，PG可被认为是CRMO的一种皮肤表现，尽管这种有趣关系背后的机制仍有待充分阐明。
