Acta Dermato-Venereologica 99-13CompleteContent | Page 35
SHORT COMMUNICATION
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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