Case report
Ulcerative nodules in a 40-year-old farmer with mycosis
fungoides: a case report
Sharina Macapagal, MD, Ma. Flordeliz Abad-Casintahan, MD, FPDS, FDSP-PDS and
Catherine Go-Teodosio, MD, FPDS
Jose R. Reyes Memorial Medical Center,
Manila, Philippines
Correspondence
Sharina Macapagal, MD
Jose R. Reyes Memorial Medical Center
Manila
Philippines
Email: sharinamacapagal@gmail.com
Funding source: None.
Conflict of interest: None.
doi: 10.1111/ijd.16204
from direct inoculation of the fungus through a break in the skin
Introduction
integrity. The clinical picture in affected individuals is variable
Cutaneous T-cell lymphoma (CTCL) is a malignancy of skin-
and can present as papules, nodules, and plaques. Once the
homing T-cells. As a result of the loss of the normal T-cell
diagnosis is established by histopathology and fungal culture,
receptor (TCR) and the disruption of the normal skin barrier,
management presents the next challenge as these organisms
there is increased susceptibility to opportunistic fungal infec-
are often highly resistant to most antifungal agents.5
tions, and mortality often results from infectious complications.
The treatment of CTCL becomes a challenge when the disease
is complicated with a mixed fungal infection as the latter must
Case report
be treated before the main problem can be addressed.
Mycosis fungoides (MF), a cutaneous T-cell lymphoma,
A case of a 40-year-old male is presented, with a 5-year his-
impairs both the cutaneous barrier and the immune response.
erythematous to hyperpigmented patches on anterior abdomen
Patients with this condition are often at high risk for developing
evolving to hypopigmented patches accompanied by weight
opportunistic infections such as cutaneous aspergillosis and
loss and inguinal lymphadenopathy. Findings of clinical and
scopulariopsis.1 Aspergillus and Scopulariopsis are found in soil
histopathologic examination were compatible with MF, plaque
and organic wastes.2–4 Cutaneous deep fungal infection results
stage. A skin punch biopsy and fungal culture were done
(a)
tory of generalized pruritus and diffuse hyperpigmentation with
(b)
Figure 1 Histopathologic findings of
aspergillosis: (a) Acute angle branching
hyphae within the granulomatous infiltrate
(Periodic acid–Schiff stain, 9100), (b)
Phialides with conidia radiating from the entire
round surface of the vesicle (wet mount)
ª 2022 the International Society of Dermatology.
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International Journal of Dermatology 2022
2
Case report
MF complicated by mixed deep fungal infection
(a)
(b)
(c)
(d)
(e)
(f)
(g)
(h)
(i)
(j)
(k)
(l)
(m)
(n)
(o)
Figure 2 Progress with treatment: (a-e) Diffuse xerosis of trunk and extremities with hyperpigmented and hypopigmented patches, ill-defined
pink plaques with ulcerations on the face, neck, left axillary area and left hand, and yellow to brown discoloration and hyperkeratosis of nail
plates of all the digits, (f-j) Complete response of lesions resulting in hypertrophic scars and ectropion of the left eye after 12 weeks of oral
itraconazole 100 mg/tab twice daily, (k-o) Marked improvement of lesions after 4 weeks of amphotericin B 50 mg/vial once daily
because of a suspicion of a possible fungal infection and
revealed Aspergillus spp. (Fig. 1). The patient has commenced
lesions. Hence, the patient was admitted and was started on
amphotericin B 50 mg intravenously once a day for 4 weeks
on itraconazole 100 mg twice daily. The ulcerations and nod-
and showed marked improvement of the lesions (Fig. 2). The
ules on the face and neck were completely responsive to
medication was well tolerated despite moderate elevation in
treatment, with significant residual scarring (Fig. 2). However,
creatinine level. With the challenge in clearing the fungal infec-
in the sixth month of treatment, there were recrudescent nod-
tion as well as renal impairment, chemotherapy for MF was
ules on the right and left lateral neck. A biopsy, fungal culture,
significantly delayed.
and matrix-assisted laser desorption ionization-time of flight
(MALDI-TOF) mass spectrometry were done and showed
Scopulariopsis spp. (Fig. 3) which was sensitive to ampho-
Discussion
tericin B, itraconazole, posaconazole, and voriconazole. The
As a result of profound immunosuppression and impaired skin
patient was referred to the infectious disease specialist for
barrier, patients with MF are prone to infectious complications
therapeutic
with Staphylococcus aureus as the most commonly identified
evaluation.
Itraconazole
200 mg
daily
was
increased to 400 mg for 1 month with no improvement of
International Journal of Dermatology 2022
pathogenic cause.6
ª 2022 the International Society of Dermatology.
MF complicated by mixed deep fungal infection
(a)
Case report
(b)
Figure 3 Histopathologic findings of
scopulariopsis: (a) Long thin septated
hyphae with vesicular swellings found near
granulomas (Periodic acid–Schiff stain,
9100), (b) Hyaline septate hyphae with
brush-like annelides containing chains of
conidia (Wet mount)
Aspergillosis is the most frequent invasive mold infection in
immunocompromised patients.7 For this patient, in particular,
his long-term habitation in an orchard with probable exposure to
high spore counts,4 frequent incidental cutaneous injuries, and
impaired cutaneous barrier in MF were likely risk factors for
developing this infection.
Acknowledgment
The authors thank the investigators who helped in the article: Dr.
Deo Adiel F. Wong and Dr. Hester Gail Y. Lim-Bueser. Ethical
approval: The Institutional Review Board (IRB) of the hospital
approved the research protocol before commencing the study.
The Infectious Disease Society of America guidelines specify
three groups of antifungal agents for the treatment of aspergillosis, namely triazoles, amphotericin B, and echinocandin. Triazoles are the preferred agents for most patients.5 Amphotericin
B is often reserved for use in resource-limited settings in which
no alternative agents are available as in the case of our patient.
Scopulariopsis is frequently isolated from onychomycosis
patients. Cutaneous infection is rare but has been reported.4
Several drug susceptibility studies have shown that the organism is resistant to broad-spectrum antifungal agents including
amphotericin B, flucytosine, fluconazole, itraconazole, voriconazole, and terbinafine.6,8,9
Decisions on when to proceed with chemotherapy for MF
should involve both infectious disease specialists and hematologists/oncologists, as they must consider the progression of cutaneous infection during treatment versus the risk of death from
the underlying malignancy if the treatment is delayed.10,11 In
cases such as these, multiple biopsies, fungal cultures, and
MALDI-TOF may be needed to establish the etiology of the
opportunistic pathogens which can present as multiple coexisting infections in a single patient.
Conclusion
Treatment of aspergillosis and scopulariopsis remains difficult,
especially for immunocompromised patients who need to start
chemotherapy because of multiple drug interactions since
most antifungal agents are nephrotoxic. Given the high resistance to antifungal agents and its potential to disseminate in
immunocompromised hosts, treatment of a secondary opportunistic fungal infection serves as a roadblock to therapy.
ª 2022 the International Society of Dermatology.
References
1 Tsambiras PE, Patel S, Greene JN, et al. Infectious
complications of cutaneous T-cell lymphoma. Cancer Control
2001; 8: 185–188.
2 Kelati A, Gallouj S, Tahiri L, et al. Defining the mimics and
clinico-histological diagnosis criteria for mycosis fungoides to
minimize misdiagnosis. Int J Women’s Dermatol 2017; 3:
100–106.
3 Bernardeschi C, Foulet F, Ingen-Housz-Oro S, et al. Cutaneous
invasive aspergillosis. Medicine 2015; 94: e1018.
4 Liu X, Yang J, Ma W. Primary cutaneous aspergillosis caused
by Aspergillus.Fumigatus in an immunocompetent patient.
Medicine 2017; 96: e8916.
5 Patterson TF, Thompson GR, Denning DW, et al. Practice
guidelines for the diagnosis and management of aspergillosis:
2016 update by the Infectious Diseases Society of America. Clin
Infect Dis 2016; 63: e1–e60.
6 Rakita RM, Lease ED, Edelman JD, et al. Successful treatment
of Scopulariopsis infection in a lung transplant recipient. Am J
Transplant 2015; 15:2010.
7 Paredes K, Capilla J, Mayayo E, et al. Virulence and resistance
to antifungal therapies of Scopulariopsis species. Antimicrob
Agents Chemother 2016; 60: 2063–2068.
8 Sellier P, Monsuez J-J, Lacroix C, et al. Recurrent
subcutaneous infection due to Scopulariopsis brevicaulis in a
liver transplant recipient. Clin Infect Dis 2000; 30:820–823.
9 Shaver CM, Castilho JL, Cohen DN, et al. Fatal Scopulariopsis
infection in a lung transplant recipient: lessons of organ
procurement. Am J Transplant 2014; 14: 2893–2897.
10 Bruynzeel I, Starink TM. Granulomatous skin infection caused
by Scopulariopsis brevicaulis. J Am Acad Dermatol 1998; 39:
365–367.
zary
11 Kamijo H, Miyagaki T. Mycosis fungoides and Se
syndrome: updates and review of current therapy. Curr Treat
Options in Oncol 2021; 22: 10.
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