Acta Dermato-Venereologica 99-13CompleteContent | Page 35

SHORT COMMUNICATION 1313 Successful Treatment of Recurrent Cutaneous Purpureocillium lilacinum (Paecilomyces lilacinus) Infection with Posaconazole and Surgical Debridement Wei-Yu CHEN 1 , Siao-Ru LIN 2 and Sung-Jen HUNG 1 * Department of Dermatology, and 2 Department of Laboratory Medicine, Hualien Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation, and Tzu Chi University, Hualien, Taiwan. *E-mail: [email protected] 1 Accepted Sep 18, 2019; E-published Sep 18, 2019 Purpureocillium lilacinum, previously known as Paecilo- myces lilacinus, is a saprophytic and filamentous mould that exists in soil, decaying food, and paper. It can produce conidia and spores in human tissue, but it seldom results in infectious disease because of its low virulence (1, 2). Nonetheless, P. lilacinus has become an emerging patho- gen in both immunocompromised and immunocompetent hosts recently (3–5). A chronic recalcitrant cutaneous ulcer induced by P. lilacinum is rare, and there is no consensus regarding its treatment. Treatment response to traditional antifungal agents, such as amphotericin B, fluconazole, griseofulvin, and echinocandins, was poor. Newer triazo- les, including voriconazole and posaconazole, have lower minimum inhibitory concentration (MIC) in vitro. Pre- viously reported patients with cutaneous infection treated with voriconazole responded well. However, only one patient was treated with posaconazole for 4 weeks but was lost to follow-up (3, 6). Herein, we describe a rare case of recurrent cutaneous P. lilacinum infection in a patient with Evans’ syndrome who was successfully treated with oral posaconazole and surgical debridement. CASE REPORT A male farmer in his 40s presented with painful papules on his right arm for a month and ulcerations on his left shin for 6 months. The lesions started as asymptomatic small erythematous papules that slowly progressed to painful ulcers surrounded by purpuric papules. The patient stated that he did not experience any trauma or have insect bites. He had a history of Evans’ syndrome and was treated with oral eltrombopag (50 mg/day) and prednisolone (10 mg/day) for more than 10 years. Azathioprine, mycophenolate, and dapsone were also given when acute exacerbation occurred. These lesions had a poor response to amoxicillin/clavulanic acid, levofloxacin, and fluconazole. The physical examination revealed a 1.5-cm necrotic ulcer with several satellite, purpuric papules, and sinus tract and pus formation on the patient’s right arm (Fig. 1A). Several 0.5-cm ulcerative and purpuric papules were found on his left shin (Fig. 1B). Subsequently, an incisional biopsy was performed. The histopathologic examination showed diffuse suppurative and granulomatous inflammation extending from the dermis to the subcutis layer without vasculitis or caseous necrosis. Non- pigmented spores and septate hyphae with a positive periodic acid-Schiff stain were found (Fig. 2A, B). Results of the tis- sue culture showed a woolly colony with a faint lilac colour in the centre and white colour in the peripheral area (Fig. 2C). Slide cultures revealed septate branching hyaline hyphae with elongated, tapering phialides and chain-like conidiophores (Fig. 2D). Cutaneous hyalohyphomycosis caused by Purpureocillium infection was diagnosed, based on findings from the mycological examinations and fungal culture. Genotypic identification of the clinical isolate by sequencing the internal transcribed spacer (ITS) region of the ribosomal RNA gene (7) confirmed P. lilacinum infection. The nucleotide sequence identity of the region was 100% P. lilacinum and matched the ribosomal RNA gene sequence deposited in the National Center for Biotechnology Information (GenBank: MK713625.1). Oral voriconazole (400 mg daily) was given for 12 weeks, and his wounds healed completely. However, a recurrent deep necrotic ulcer surrounded by haemorrhagic bullae and papules developed on the same site on his left shin 5 months later after discontinuing voriconazole (Fig. 1C). Results of the biopsy and culture showed that the patient had recurrent P. lilacinum hyalohyphomycosis. The susceptibility test showed that the minimum inhibitory concentra- tion (MIC) values for voriconazole and posaconazole were 0.5 mg/l and 1 mg/l. Then voriconazole was switched to oral posaconazole (300 mg daily) for 11 weeks, and surgical debridement followed by split-thickness skin grafting was performed. No recurrence was noted for more than 2 years (Fig. 1D). DISCUSSION P. lilacinum can invade multiple human organs and cause va- rious infections. Oculomycosis accounted for the most cases (51.3%), and it led to cutaneous and subcutaneous infections (35.3%). The prevalence of cutaneous infection is still unknown. Forty-two cases were reported from 1977 to 2004 (1). It can be transmitted by both direct inoculation and hematogenous dissemination. Outbreaks induced by contaminated skin cream have been reported (8). The predisposing factors of the infection are malignancy, Fig. 1. Photographs of the patient’s clinical course. A) A necrotic ulcer with peripheral sinus tract formation on the right forearm. B) Several brownish to violaceous papules and sinus tracts on the left anterior shin. C) A huge necrotic ulcer with peripheral haemorrhagic vesicles and bullae has developed after discontinuation of voriconazole for 5 months. D) Ulcers and bullae are resolved after posaconazole treatment (300 mg/day) for 11 weeks and surgical debridement with split-thickness skin grafting. This is an open access article under the CC BY-NC license. www.medicaljournals.se/acta Journal Compilation © 2019 Acta Dermato-Venereologica. doi: 10.2340/00015555-3320 Acta Derm Venereol 2019; 99: 1313–1314