Here we present the first case in Asia

Here, we present the first case in Asia of sarcoidosis with bilateral leg lymphedema as the initial presentation. Notably, cutaneous manifestation is common in sarcoidosis. This case reminds us to consider sarcoidosis in the differential diagnosis of leg lymphedema, thereby prompting early diagnosis and treatment.
Introduction Paecilomyces species are saprophytic molds found ubiquitously in the environment. Although they were deemed as laboratory contaminants in the past, recently there has been an increase in the number of Paecilomyces-related infections. The most commonly reported pathogenic species are Paecilomyces lilacinus, Paecilomyces variotii, and Paecilomyces marquandii. Clinically, cutaneous and subcutaneous infection was the second most common manifestation, following oculomycosis. Most of these cases were found in immunocompromised patients who were under post-transplantation status, with hematological malignancies or AIDS. The main route of cutaneous infection reported in the literature was direct cutaneous inoculation through the colonization of clinical materials, such as applying contaminated purchase 4-ethylphenyl sulfate lotion or using incompletely sterilized central venous catheter, although some patients contracted the infection through dog bite, contaminated water after flooding, or wounds of mechanical trauma.Paecilomyces infection is an emerging hyalohyphomycosis in humans, but there is relative inexperience in treating this infection. In this paper, we present the case of a cutaneous P. lilacinus infection in an elderly but immunocompetent patient, who had been successfully treated with oral itraconazole. We also reviewed all the cases of cutaneous P. lilacinus infection in immunocompetent patients reported in English literature to provide treatment guidance for physicians in the future.
Case Report An 87-year-old Taiwanese man, with no known immunocompromised status, presented with a progressive, tender, erythematous plaque on the ventral surface of the right forearm for 2 weeks (Figure 1A). Before the skin lesion developed, he alleged that there were some itchy rashes over the area, and excoriated wounds due to frequent scratching were noted by his daughter. Under the initial impression of cellulitis, he was admitted to our hospital and empiric treatment with intravenous oxacillin was initiated. Nevertheless, due to unresponsiveness to the treatment for 1 week, we decided to perform skin biopsy and tissue culture to diagnose the underlying condition. Histopathologic analysis of the skin specimen revealed suppurative granulomas with pathogens showing nonpigmented, septated, branching hyphae, which were highlighted by periodic acid–Schiff-diastase (PAS-D) stain (Figures 1C and 1D). The tissue culture on Sabouraud dextrose agar (SDA) showed whitish floccose colonies with central lilac discoloration and brownish pigmentation on the reverse side of the culture plate. However, due to the lack of malt extract agar in our hospital, we did not demonstrate fungal culture result on it. The fungal morphology under a microscope revealed erection of the conidiophores with clustered tenpin-shaped phialides and round to oval nonbranching conidia (Figures 1D, 2A, and 2B). The pathogen was identified to be P. lilacinus. Moreover, we conducted polymerase chain reaction with primers internal transcribed spacers 1 (ITS1) and ITS4 to amplify the ITS1, 5.8S RNA gene, and ITS2 regions of the fungal DNA. The resulting sequence, composed of 573 nucleotides, was compared with the public database (http://blast.ncbi.nlm.nih.gov/Blast.cgi) and the results best matched with P. lilacinus, showing 100% similarities. According to the final diagnosis of cutaneous P. lilacinus infection, treatment with oral itraconazole (200 mg/d) was subsequently initiated and the erythematous plaque on his right forearm resolved after a 4-week treatment (Figure 1B).
Discussion P. lilacinus is a saprophytic mold found ubiquitously in the environment; however, it has now become an emerging pathogen causing localized to severe systemic infection. Most of the reported cases were under immunocompromised status and the main predisposing factors included solid organ transplantation, bone marrow transplantation, corticosteroid therapy, liver cirrhosis, diabetes mellitus, AIDS, and primary immunodeficiency, such as chronic granulomatous disease. However, an increasing number of events occurring in healthy people had been reported recently. This growing incidence strongly suggests that the pathogen P. lilacinus is no longer only an opportunistic pathogen and that the worldwide disease burden could have been underestimated.