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Disseminated blastomycosis presenting as oligoarticular septic arthritis in a 12-year-old girl.

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Arthritis & Rheumatism (Arthritis Care & Research)
Vol. 53, No. 1, February 15, 2005, pp 138 –141
DOI 10.1002/art.20916
© 2005, American College of Rheumatology
TRAINEE ROUNDS
Disseminated Blastomycosis Presenting as
Oligoarticular Septic Arthritis in a 12-Year-Old
Girl
ANDREW J. HEAD, LINDA K. MYERS, JEFFREY D. THOMPSON, STEVEN C. BUCKINGHAM,
ROBERT B. SKINNER, JR
Introduction
Septic arthritis due to blastomycosis is rare, and when
present is usually monarticular, although disease involving multiple joints has been described (1,2). Only 2 children have previously been reported to have arthritis as a
presenting symptom of blastomycosis (3,4). Both of these
cases were monarticular. We describe a case of an immunocompetent 12-year-old girl with disseminated blastomycosis who presented with skin lesions and arthritis involving 3 joints. To our knowledge, this is the first pediatric
patient reported with blastomycotic arthritis involving ⬎1
joint.
Case report
A previously healthy 12-year-old African American girl
from Tennessee was referred to our institution with a
1-month history of knee pain and papulopustular skin
lesions on the face that had not responded to treatment
courses with oral cephalexin followed by oral clindamycin. The review of systems was significant for low-grade
fever for 3 days and a 10-pound weight loss over 2 months.
Physical examination was remarkable for a temperature of
38.3°C and a limp favoring the left leg. Joint exam revealed
a moderate effusion in the left knee that was warm and
mildly tender. The right knee had a small effusion and
mild tenderness. The left ankle was slightly swollen,
warm, and tender as well. The rest of her joints were
unremarkable. The skin examination revealed 3 5– 6 cm
well-demarcated scaly verrucous plaques in varying stages
on her forehead and chin. She also had 3 discrete nonAndrew J. Head, MD, Linda K. Myers, MD, Jeffrey D.
Thompson, MD, Steven C. Buckingham, MD, Robert B. Skinner Jr, MD: University of Tennessee Health Science Center
and Children’s Foundation Research Center at LeBonheur
Children’s Medical Center, Memphis, Tennessee.
Address correspondence to Andrew J. Head, MD, Division
of Rheumatology, University of Tennessee Health Science
Center, G326 Coleman Building, 956 Court Avenue, Memphis, TN 38163. Email: headaj@hotmail.com.
Submitted for publication April 23, 2004; accepted in
revised form July 11, 2004.
138
AND
tender 2–3 cm purple nodules: 1 on her left upper arm, 1
on the left palm, and 1 on the left foot (Figure 1).
The peripheral blood leukocyte count was 15,000/mm3
(normal range 5,000 –13,000/mm3) with 78.4% neutrophils, the hemoglobin was 9.4 gm/dl (normal range 11–16
gm/dl), and the platelet count was 666,000/mm3 (normal
range 140,000 – 450,000/mm3).
Serum creatinine, aspartate aminotransferase, and alanine aminotransferase levels were normal. The erythrocyte
sedimentation rate was 98 mm/hour (normal range 0 –13
mm/hour) and the C-reactive protein level was 21.0 mg/dl
(normal range 0.3– 0.9 mg/dl). Blood cultures were negative. Arthrocentesis of the left knee yielded 10 cc of turbid
fluid. The synovial fluid leukocyte count was 31,600/mm3
with 82% neutrophils and the synovial fluid red blood cell
count was 11,800/mm3. Bacterial culture of the synovial
fluid was negative. Serum assays for rheumatoid factor,
antinuclear antibody, antistreptolysin-O antibody, human
immunodeficiency virus antibody, and HLA–B27 yielded
negative results. An evaluation of the patient’s immune
function revealed normal numbers of B and T cells and
normal responses to mitogens. Serum assays of total hemolytic complement and quantitative immunoglobulins
revealed normal results.
Radiographs of the knees demonstrated bilateral soft
tissue swelling and a cyst-like lucency within the medial
aspect of the right proximal tibial metaphysis. Magnetic
resonance imaging (MRI) of the knees revealed a lesion
involving the metaphysis extending into the epyphysis of
the medial right proximal tibia and a similar lesion at the
distal left femur. These lesions demonstrated a hyperintense signal with surrounding hypointense signal on T2
imaging, consistent with osteomyelitis or early Brodie’s
abscess (Figure 2). A technetium-99 bone scan showed
increased uptake in the right 7th and 8th ribs, right iliac
crest, left distal femur, right medial tibial plateau, right
distal tibia, and the occiput of the skull. A chest radiograph revealed perihilar infiltrates and computed tomography of the chest demonstrated left upper lobe consolidation with air bronchograms, a left-sided moderate
pleural effusion, a right-sided rib lesion, a calcified 3-mm
Blastomycosis Presenting as Oligoarticular Arthritis
139
Figure 1. Skin lesions in various stages. Clockwise from top left: forehead, left arm, left palm, medial left foot.
nodule at the right lung base, and multiple 3-mm hypodensities in the spleen consistent with abscesses. An MRI
scan of her brain showed no lesions.
A biopsy of one of the facial skin lesions was performed.
Routine microscopy with hematoxylin and eosin showed
epidermal hyperplasia with suppurative and granulomatous inflammation. Silver stain revealed thick-walled,
broad-based budding yeast diagnostic of blastomycosis
(Figure 3). Complement fixing antibodies to Blastomyces
dermatitidis were identified in her serum (titer 1:64 dilutions).
The patient was hospitalized and intravenous ceftriaxone was administered initially. By hospital day number 1
the result of the skin biopsy was learned, at which time the
ceftriaxone was stopped and intravenous amphotericin B
administered (50 mg daily or 0.8 mg/kg/day). The patient
defervesced within 48 hours of beginning amphotericin B.
The joint pain resolved with resultant improvement in
ambulation over a 1-week period. By hospital day number
8, she was discharged. The amphotericin B was continued
daily for 14 days, at which point the treatment was
changed to oral itraconazole 100 mg twice daily. One
month following admission, the patient was afebrile, reported no joint pains, and was ambulating without difficulty. In addition, at 1-month followup, the skin lesions
were resolving, but not completely healed.
Discussion
The clinical picture of pneumonia, septic arthritis, osteomyelitis, and verrucous skin lesions, with histopathologic
Figure 2. Plain radiograph (left) and T2-weighted magnetic resonance image (right) of the right knee show a 4 ⫻ 6 cm
spherical cyst with surrounding sclerosis within the medial aspect of the proximal tibial metaphysis extending into
the epiphysis, consistent with Brodie’s abscess.
140
Figure 3. Silver-stained skin biopsy specimen under high power
demonstrating thick-walled, broad-based budding yeast typical of
Blastomyces dermatitidis (arrow).
evidence of broad-based budding yeast and serologic evidence of B. dermatitidis infection together confirm the
diagnosis of disseminated blastomycosis in our patient.
Blastomycosis is caused by the dimorphic fungus B.
dermatitidis and is endemic in the midwestern, south
central, and south eastern United States (5). The annual
incidence of infection in the US is reported as 0.2/100,000
to 1.23/100,000, but reaches as high as 43.21/100,000 in
endemic areas (6,7). An underlying immunosuppressive
illness is reported in 12.5–25% of cases (7,8). Only 3–11%
of cases occur in children (3,4). Frequently infection is
asymptomatic, but disease involving multiple organ systems has been described (1– 4,6 –13).
As in our patient, the initial symptoms of blastomycosis
infection are frequently constitutional, including malaise,
fever, chills, and weight loss. The portal of entry is the
respiratory tract and the organism is inhaled as conidia,
initially causing pneumonitis. This process can be asymptomatic (as in our patient) or present as acute or chronic
pneumonia (5). Once within the alveolar macrophages, the
infection is either controlled by the host immune system
or spreads to distant organs hematogenously (10).
Our patient demonstrated the typical skin and other
organ involvement of disseminated blastomycosis. The
skin is involved in 60 – 80% of cases and the lesions can be
verrucous, ulcerative, or both. Dermal abscesses can form
and spontaneously drain or cause sinus tracts. Osteomyelitis is common and the most common bones involved are
(in descending order) vertebrae, pelvis, sacrum, skull, ribs,
and long bones (5).
Blastomycotic arthritis, although well described, is relatively uncommon (1– 4,6 –13). Joint pain is reported in
11% of patients and 8% have frank arthritis (11). The
arthritis is usually monarticular, but a few cases involving
multiple joints have been reported (1,2). Overall, the knee
joint is most frequently affected (10). Diagnostic delays
have been documented due to the ability of blastomycosis
to mimic other more common conditions, such as bacterial
arthritis (6). This was the case in our patient. Direct extension of osteomyelitis is a well-recognized cause of blastomycotic arthritis, but hematogenous seeding of the synovial membrane is thought to occur as well (11).
The synovial fluid of the blastomycotic joint is usually
Head et al
frankly purulent and, as in our patient, can mimic culturenegative bacterial arthritis (1). Reports in the literature
suggest the synovial fluid leukocyte count may vary from 450
to 100,000/mm3 with ⬎70% neutrophils (1,5). Synovial fluid
fungal cultures and KOH staining are useful in diagnosis and,
when performed, have a diagnostic yield of 82% (11). The
definitive diagnosis is made by culture or identification of the
organism from tissue samples or fluid (5).
To our knowledge there are 2 cases of blastomycotic
arthritis reported in children and both of these were monarthritic at presentation (3,4). Neither reported synovial
fluid analysis. The first patient was a 7-year-old girl who
presented with a 1-month history of left ankle pain and
swelling. This patient had a lytic lesion with a well-circumscribed border in the dome of the talus. She was
treated with curettage of the bone lesion and oral ketoconazole 100 mg/day for 2 months. At 2 years followup there
was no evidence of disease recurrence (3).
The second case was an 8-year-old boy who presented
with foot pain and constitutional symptoms. He developed
skin lesions and painful swelling of the right ankle and left
elbow within 2 months. He was treated with oral ketoconazole 150 mg/day for 2 months, but his condition worsened. Ketoconazole was withdrawn and intravenous amphotericin B initiated. He received a total of 1 mg/kg/day
of amphotericin B for 1 month and then itraconazole 100
mg/day for 3 months. One year after treatment he had
resolution of the skin lesions but residual arthralgias of the
right ankle (4).
Intravenous amphotericin is the first-line antifungal
agent for the management of life-threatening disseminated
blastomycosis; however, therapy for some patients without central nervous system involvement may be switched to
oral itraconazole after stabilization on intravenous amphotericin B (14). Our patient stabilized on a 2-week course of
intravenous amphotericin B and was changed to oral itraconazole. We plan to continue itraconazole for 12 months to treat
her osteomyelitis. She has reported no problems at followup
and has demonstrated ongoing clinical improvement.
In conclusion, septic arthritis due to blastomycosis is
rare, and when present is usually monarticular, but can
involve multiple joints and bones. The presence of pulmonary symptoms or typical skin lesions, if present, suggest
the diagnosis. Blastomycotic arthritis should be included
in the differential diagnosis of oligoarthritis in a patient
living in an endemic area, particularly if associated with
skin and bone lesions and constitutional symptoms.
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