Download Severe Oculofacial Sequelae of Cutaneous Blastomyces dermatitidis

Case Reports
Severe Oculofacial Sequelae of
Cutaneous Blastomyces dermatitidis
Joseph S. Schmutz, M.D.*, Nicholas A. Ramey, M.D.*,
Gregory M. Gauthier, M.D.†, and
Mark J. Lucarelli, M.D., F.A.C.S.*
Abstract: This study reports a case of Blastomyces
dermatitidis soft tissue infection resulting in a disfiguring
lower eyelid ectropion from cicatricial and postinflammatory
cutaneous changes. Primary treatment included intravenous
amphotericin B followed by long-term oral itraconazole,
which resulted in complete remission of the disease without
debridement, after which cicatricial ectropion was repaired
surgically with scar release, full-thickness skin graft, and
temporary Frost tarsorraphy. Cutaneous blastomycosis
may cause severe oculofacial sequelae, ranging from eyelid
ectropion to widespread facial cicatrix, and may mimic
other more common infectious processes, in addition to
malignancy. Recommended antifungal therapy includes
induction with intravenous amphotericin B and a long course
of oral antifungals, preferably coordinated in conjunction
with an infectious disease specialist. Ectropion repair should
be delayed until the inflammatory response has completely
healed. If the ocular surface is compromised or nearby
ocular structures are threatened, primary debridement and
repair may be indicated.
B
lastomyces dermatitidis, the etiologic agent of blastomycosis, causes pyogranulomatous inflammation. Infection is
primarily acquired by inhalation of aerosolized spores following
disruption of soil to cause pneumonia, which can be followed
by lymphohematogenous dissemination to the skin. This pathogen can also cause primary cutaneous disease by a penetrating
injury to the skin. This report illustrates a patient with severe
facial and periocular blastomycosis complicated by cicatricial
lower eyelid ectropion. Primary treatment and secondary repair
are discussed. The medical literature is reviewed for recent
clinical and microbiologic updates on blastomycosis. Informed
written consent was obtained for the use of clinical photographs
from the patient, and this study is Health Insurance Portability
and Accountability Act (HIPAA) compliant.
CASE REPORT
A 45-year-old man in August of 2010 was evaluated for
bilateral facial lesions (Figure), OS irritation, malaise, and weight
loss. He developed periauricular and cervical lymphadenopathy,
and purulent nodules involving his face, trunk, and upper and
lower extremities 14 months prior to presentation. Over a period
of several weeks after initial presentation, several cutaneous
lesions spontaneously resolved, but those on his face and buttocks
persisted. In 2009, he sought medical attention at an urgent care
*Department of Ophthalmology, Oculoplastic, Facial Cosmetic and
Orbital Surgery Service; and †Department of Medicine, University of
Wisconsin, Madison, Wisconsin, U.S.A.
Accepted for publication June 1, 2014.
Supported, in part, by an unrestricted grant from Research to Prevent
Blindness Inc., New York, NY, to the Department of Ophthalmology and
Visual Sciences.
This work has not been previously presented.
The authors have no financial or conflicts of interest to disclose.
Address correspondence and reprint requests to Joseph Schmutz, M.D.,
University of Wisconsin, Madison, WI 53705. E-mail: [email protected]
DOI: 10.1097/IOP.0000000000000245
Ophthal Plast Reconstr Surg, Vol. XX, No. XX, 2014
facility for the facial lesions. He was prescribed clindamycin for
10 days but was unable to complete the antibiotic course due to
the development of a drug rash. From December 2009 to August
2010, the patient experienced progression of his facial and buttock
lesions. He was diagnosed with cutaneous Blastomycosis based
on skin biopsy demonstrating broad-based budding yeast and a
positive urine Blastomyces antigen. Culture of the skin lesions
grew B. dermatitidis. Following induction therapy with liposomal
amphotericin B, which was stopped at 9 days due to development
of a morbilliform rash from the amphotericin, he was treated with
oral itraconazole solution for 9 months. Itraconazole levels were
monitored monthly and were therapeutic (>1 μg/ml) throughout the treatment course. Therapy was discontinued due to side
effects of headache, nausea, and fatigue. Surgical debridement
of infected tissue was not required to clear the active infection, as
appropriate medical treatment yielded dramatic results. (Figure).
The verrucous lesions ultimately improved after several months
of the above regimen, leaving large cheek and left lower eyelid
cicatricial plaques in its wake. The resulting left lower eyelid
cicatricial ectropion was repaired in a single-stage procedure 9
months after finishing antifungal therapy. The scar was released.
A 35 mm × 17 mm full-thickness skin graft was taken from the
right retroauricular area to exactly match and fill the defect, and
then a lateral tarsal strip procedure was performed, followed by
temporary Frost tarsorraphy.
DISCUSSION
Blastomyces dermatitidis grows in the moist soil of
wooded areas rich in organic debris of the Mississippi and Ohio
River basins, United States, and Canadian regions bordering the
Great Lakes and the St. Lawrence River Valley. It is hyperendemic in north-central Wisconsin with an incidence up to 40
per 100,000 in certain counties.1 The fungus is thermally dimorphic, existing as a filamentous mold in the soil (22–25°C) and
as a pathogenic yeast in the tissue (37°C). Following disruption
of soil, aerosolized mycelial fragments and infectious spores
are inhaled into the lungs and converted into broad-based budding yeast. Once established in the lung, yeast can spread to
extrapulmonary sites, most often to the skin, bone, and genitourinary system.2,3 Integumentary manifestations occur in up
to 80% of patients with disseminated infection, with predilection for the face and exposed areas.4 Eyelid involvement with
systemic blastomycosis infection is rare, occurring in less than
2% of cases.5 Cutaneous blastomycosis may cause severe oculofacial sequelae, ranging from eyelid ectropion to widespread
facial cicatrix. Rarely, intraocular involvement and optic nerve
infection have also been reported.6
Although integumentary manifestations are common
after hematogenous dissemination, direct inoculation of the skin
is also possible after a penetrating injury.7
Cutaneous and systemic manifestations of blastomycosis may mimic other more common infectious processes and
malignancy.2,4 Understanding the risk factors associated with
acquisition of B. dermatitidis is important for prompt diagnosis.
Practice guidelines published by the Infectious Diseases Society
of America recommend that patients with severe infection be
treated with intravenous amphotericin B followed by a prolonged
course of oral antifungals such as itraconazole.8 Coordination
with an expert in infectious diseases is recommended.
Whenever possible, surgical correction of oculofacial
sequelae should be delayed until clearance of infection. Delay
may not be feasible when nearby ocular structures are threatened or when refractory lagophthalmos or severe retraction
threaten to compromise protection of the ocular surface. In
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Copyright © 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. Unauthorized reproduction of this article is prohibited.
Ophthal Plast Reconstr Surg, Vol. XX, No. XX, 2014
Case Reports
External photographs documenting severe active Blastomyces dermatitidis facial cellulitis (top left and center). Postinfectious
cicatricial left lower eyelid ectropion remained after 9 months of systemic antifungal treatment (top right). Postoperative photographs
demonstrate successful repair after scar release, full thickness skin graft, and temporary Frost tarsorraphy (below).
these cases, earlier surgical intervention should be considered.
In this case, the patient required 9 months of therapy for the
infection to completely clear, after which successful cicatricial
ectropion repair was accomplished.
In summary, B. dermatitidis infection can have severe
oculofacial sequelae. If possible, reconstruction should be
delayed until active infection is cured and cicatrix is no longer
progressing.
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of blastomycosis in a region of high endemicity in north central
Wisconsin. Clin Infect Dis 1992;15:629–35.
2. Saccente M, Woods GL. Clinical and laboratory update on blastomycosis. Clin Microbiol Rev 2010;23:367–81.
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2010;7:173–80.
4. Woofter MJ, Cripps DJ, Warner TF. Verrucous plaques on the face.
North American blastomycosis. Arch Dermatol 2000;136:547–50.
5.Bartley GB. Blastomycosis of the eyelid. Ophthalmology
1995;102:2020–3.
6. Pariseau B, Lucarelli MJ, Appen RE. Unilateral Blastomyces dermatitidis optic neuropathy case report and systematic literature review. Ophthalmology 2007;114:2090–4.
7.Shukla S, Singh S, Jain M, et al. Paediatric cutaneous blastomycosis: a rare case diagnosed on FNAC. Diagn Cytopathol
2009;37:119–21.
8. Chapman SW, Dismukes WE, Proia LA, et al.; Infectious Diseases
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© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Copyright © 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. Unauthorized reproduction of this article is prohibited.