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Cutaneous leishmaniasis mimicking dactylitis in a patient with rheumatoid arthritis treated with certolizumab

Leishmaniasis is a rare chronic protozoal disease that involves the mononuclear phagocytic system. Since 2004, 37 cases of patients diagnosed with leishmaniasis while on treatment with different types of biological anti-tumour necrosis factor therapies have been reported. We present a case of an extraordinary form of cutaneous leishmaniasis in a patient treated with certolizumab, an association not previously reported, to our knowledge. A 52-year-old woman presented with a 4-month history of lesions on her right palm and right sole. Her medical history included refractory rheumatoid arthritis, for which she was taking certolizumab. She lived in the countryside and owned a dog. On physical examination, infiltrated eczematous-like papules were seen on the finger-pad of the first finger of the patient’s right hand and on the medial aspect of the great toe of her right foot (Fig. 1). The initial diagnosis was eczema, and betamethasone ointment was prescribed once daily for 2 weeks. At the follow-up visit, the lesions had worsened and a reddish-brown papule had appeared on the anterior aspect of the right thigh (Fig. 1). Two punch biopsies were taken, one from the right thigh and the other from the right sole. Histological examination of these revealed a mild lichenoid pattern with a dense lymphohistiocytic infiltrate throughout the whole dermis, and many intracytoplasmic inclusions in macrophages were observed in sections stained with haematoxylin and eosin and Giemsa (Fig. 2). Laboratory investigations did not show any blood test abnormalities, and abdominal ultrasonography did not show any hepatosplenomegaly. PCR and cultures of both skin biopsies were positive for Leishmania spp. and negative for fungal and mycobacterial infections. Blood leishmania PCR and bone marrow biopsy and culture were negative. Therefore, we considered that our patient had multifocal cutaneous leishmaniasis. She was successfully treated with intramuscular meglumine antimoniate for 20 days (20 mg/kg/day) (Fig. 1). No recurrence was observed within 1 year of follow-up. Anti-TNF therapies are associated with reactivation of silent granulomatous infections such as tuberculosis and leishmaniasis. It is important to highlight that in immunosuppressed patients, leishmaniasis may have an atypical clinical presentation. The clinical findings of our patient (eczematous-like lesions and finger swelling resembling dactylitis) were remarkable because they have not been reported in the literature. In addition, there was no visceral involvement despite basal immunosuppressive treatment and multiple cutaneous lesions. This is not a common finding, as anti-TNF drugs are associated with a high risk of developing visceral leishmaniasis. The term “mutifocal cutaneus leishmaniasis” was first coined by Paradisi et al. in 2005. in an immunocompetent patient. One year later, Maniscalco et al. reported 29 immunocompetent patients with multifocal cutaneous leishmaniasis. However, the first case related to anti-TNF therapy was published in 2009 by Schneider et al. To date, 37 cases of anti-TNF-induced leishmaniasis have now been reported, which 40.5% had cutaneous manifestations only (54.1% had visceral involvement). Correspondence: Dr Jose Herrerias-Moreno, Corporaci o Sanit aria Parc Tauli, Hospital Universitari de Sabadell, Universitat Aut onoma de Barcelona, Parc Taul ı s/n, 08208, Sabadell (Barcelona), Spain E-mail: jose.herrerias.moreno@gmail.com