Blastomycosis presenting as multiple splenic abscesses : Case report and review of the literature

1Department of Surgery, King Saud University, Riyadh, Saudi Arabia; 2Department of Radiology, Diagnostic Services, Boundary Trails Health Centre, Winkler; 3Department of Surgery; 4Department of Diagnostic Imaging; 5Department of Medical Microbiology; 6Department of Pathology; 7Section of Infectious Diseases, Department of Medicine, University of Manitoba, Winnipeg, Manitoba Correspondence: Dr John M Embil, Health Sciences Centre, University of Manitoba, MS 673-820 Sherbrook Street, Winnipeg, Manitoba R3A 1R9. Telephone 204-787-4654, fax 204-787-4699, e-mail jembil@hsc.mb.ca Blastomycosis is an uncommon granulomatous infection caused by the thermally dimorphic fungus Blastomyces dermatitidis, which exists in the mycelial form in the soil of warm, moist, wooded areas that are rich in organic debris (1-6). Conidia are inhaled when the mycelia are disturbed. At body temperature, they convert to thick-walled budding yeast (7,8). Hematogenous dissemination is presumed to result in extrapulmonary blastomycosis. Blastomycosis presents most commonly with pulmonary disease but may also present with skin lesions, abscesses or osteomyelitis, which may lead to misdiagnosis or delay in diagnosis. The endemic area for blastomycosis includes the Ohio and Mississippi river basins, the regions that border the Great Lakes and northwestern Ontario (Figure 1) (9). Blastomycosis may have a variety of different clinical manifestations (10), of which the pulmonary manifestations are the most frequent (11,12). There are a few reports of individuals presenting with splenomegaly and left upper quadrant discomfort as the initial manifestation, subsequently leading to a diagnosis of splenic abscess caused by B dermatitidis (13-17). We report a case of blastomycosis presenting with multiple splenic abscesses and review the previously published reports regarding this condition. Case presentation A 31-year-old Canadian Aboriginal man from northwestern Ontario presented with a two-month history of left upper quadrant pain beginning in early spring. He denied cough, fevers, chills and weight loss. His medical history was negative for diabetes or immune system disorders. There was a history of alcohol abuse until six years before presentation. The patient worked as a carpenter in the winter and forest firefighter from May through September. Physical examination revealed a tender left upper quadrant mass. Chest radiography was normal. A computed tomographic (CT) scan showed multiple lowattenuation lesions within the spleen, a collection between the splenic tip and splenic flexure of the colon, and several small adrenal lesions (Figure 2). There were no signs or symptoms of adrenal insufficiency. A tentative diagnosis of lymphoma was considered and the patient was referred to a surgeon, but he chose not to present for his evaluation for the next five months. He presented to the surgeon because the left upper quadrant pain had become severe during the two weeks before surgical evaluation and was associated with nausea and vomiting. Weight loss (18 kg) was noted, but there were no constitutional symptoms, respiratory Case report


Blastomycosis presenting as multiple splenic abscesses: Case report and review of the literature
Sami Al-Nassar MD FRCSC 1 , Tracy MacNair MD FRCPC 2 , Jeremy Lipschitz MD FRCSC 3 , Howard Greenberg MD FRCPC 4 , Elly Trepman MD 5 , Sate Hamza MD FRCPC 6 , John M Embil MD FRCPC 5,7   1 Department of Surgery, King Saud University, Riyadh, Saudi Arabia; 2 Department of Radiology, Diagnostic Services, Boundary Trails Health Centre, Winkler; 3 Department of Surgery; 4 Department of Diagnostic Imaging; 5 Department of Medical Microbiology; 6 Department of Pathology; 7 Section of Infectious Diseases, Department of Medicine, University of Manitoba, Winnipeg, Manitoba Correspondence: Dr John M Embil, Health Sciences Centre, University of Manitoba, MS 673-820 Sherbrook Street, Winnipeg, Manitoba R3A 1R9.Telephone 204-787-4654, fax 204-787-4699, e-mail jembil@hsc.mb.caB lastomycosis is an uncommon granulomatous infection caused by the thermally dimorphic fungus Blastomyces dermatitidis, which exists in the mycelial form in the soil of warm, moist, wooded areas that are rich in organic debris (1)(2)(3)(4)(5)(6).Conidia are inhaled when the mycelia are disturbed.At body temperature, they convert to thick-walled budding yeast (7,8).Hematogenous dissemination is presumed to result in extrapulmonary blastomycosis.Blastomycosis presents most commonly with pulmonary disease but may also present with skin lesions, abscesses or osteomyelitis, which may lead to misdiagnosis or delay in diagnosis.The endemic area for blastomycosis includes the Ohio and Mississippi river basins, the regions that border the Great Lakes and northwestern Ontario (Figure 1) (9).
Blastomycosis may have a variety of different clinical manifestations (10), of which the pulmonary manifestations are the most frequent (11,12).There are a few reports of individuals presenting with splenomegaly and left upper quadrant discomfort as the initial manifestation, subsequently leading to a diagnosis of splenic abscess caused by B dermatitidis (13)(14)(15)(16)(17).We report a case of blastomycosis presenting with multiple splenic abscesses and review the previously published reports regarding this condition.

Case presentation
A 31-year-old Canadian Aboriginal man from northwestern Ontario presented with a two-month history of left upper quadrant pain beginning in early spring.He denied cough, fevers, chills and weight loss.His medical history was negative for diabetes or immune system disorders.There was a history of alcohol abuse until six years before presentation.The patient worked as a carpenter in the winter and forest firefighter from May through September.Physical examination revealed a tender left upper quadrant mass.Chest radiography was normal.A computed tomographic (CT) scan showed multiple lowattenuation lesions within the spleen, a collection between the splenic tip and splenic flexure of the colon, and several small adrenal lesions (Figure 2).There were no signs or symptoms of adrenal insufficiency.
A tentative diagnosis of lymphoma was considered and the patient was referred to a surgeon, but he chose not to present for his evaluation for the next five months.He presented to the surgeon because the left upper quadrant pain had become severe during the two weeks before surgical evaluation and was associated with nausea and vomiting.Weight loss (18 kg) was noted, but there were no constitutional symptoms, respiratory A 31-year-old Canadian Aboriginal man from northwestern Ontario presented with left upper quadrant pain and a tender left upper quadrant mass.Evaluation with a computed tomography scan showed multiple lesions within the spleen, a collection between the splenic tip and splenic flexure of the colon, and several small adrenal lesions.Computed tomographic-guided needle biopsy showed necrotizing granulomatous inflammation and multinucleated giant cells.Gomori's methenamine silver stain showed broad-based budding yeast consistent with Blastomyces dermatitidis.Abdominal symptoms resolved after two months of oral itraconazole.Multiple splenic abscesses are a rare presentation of blastomycosis and should be considered in the differential diagnosis of left upper quadrant abdominal pain in a patient with a history of travel or residence in a region endemic for B dermatitidis.
complaints or night sweats.The patient also noted a 2.5 cm palpable mass in the midline of the neck, present for the previous two weeks.The patient reported that a chest radiograph two months earlier was normal.
Although a repeat CT scan was requested, the patient did not attend for the study until four months later.At this time, the CT scan revealed persistence of the splenic and adrenal lesions.A percutaneous needle biopsy of the spleen showed necrotizing granulomatous inflammation and multinucleated giant cells.Gomori's methenamine silver stain showed broad-based budding yeast consistent with B dermatitidis (Figure 3).It was presumed that the neck mass was also due to blastomycosis.
The patient was treated with oral itraconazole (200 mg twice daily).He did not return for follow-up evaluation, but was successfully contacted six months after the biopsy.He stated that after two months of itraconazole, the left upper quadrant pain and the neck mass had completely resolved.He had independently discontinued the itraconazole after two months and did not return for further evaluation because of loss of insurance coverage as a result of marital separation.Further attempts to contact the patient were unsuccessful.

DisCUssion
The current patient's presentation, with abdominal pain and splenic lesions, is an infrequently described manifestation of blastomycosis.He responded well to oral antifungal chemotherapy and did not require splenectomy.
All of the reported patients with splenic abscesses resulting from B dermatitidis had contact with geographical regions endemic for B dermatitidis (Table 1).The diagnosis of splenic abscess resulting from B dermatitidis in previously reported cases involved varied radiographic investigations, including radionuclide investigations (Table 1).More recently, CT has led to a decreased need for radionuclide studies, and CT-guided splenic aspiration has facilitated biopsy, as in the present case, obviating the need for surgical intervention.
In early reports, chronic abscesses were reported to be either sterile or caused exclusively by aerobic bacteria, but the spectrum of microorganisms reported from chronic abscesses has changed, likely as a consequence of improved microbiological techniques for the recovery of anaerobes and fungi and a heightened awareness that parasites may be responsible for   A contrast-enhanced computed axial tomographic scan of the abdomen revealing numerous tiny avascular low-density lesions that appear to be confluent splenic abscesses (18)(19)(20)(21)(22)(23)(24)(25)(26)(27)(28)(29)(30)(31)(32).The apparent increase in reports of fungi, mainly Candida species isolated from splenic abscesses, may be a result of intensified chemotherapy and corticosteroid regimens for patients with neoplasms and inflammatory conditions requiring immunosuppression.Abscesses from parasites and other unusual microorganisms such as Nocardia species and mycobacteria have also been reported, attributed to immunosuppression, injection drug use and HIV infection (23).
The patient presented in the spring after having reported two months of symptoms.The time of presentation and clinical manifestations are compatible with previous studies reporting a seasonal variation of blastomycosis (33).Localized pneumonias typically present one to six months after initial exposure, with the presumed exposure occurring in the summer months.Four to nine months after the primary exposure, reactivation or slow progression of asymptomatic infection resulting in isolated extrapulmonary or disseminated hematogenous disease is observed.Our patient's presentation is compatible with these time lines (33).
Although rare, splenic abscess caused by B dermatitidis should be considered in an individual with left upper quadrant abdominal pain who has a history of travel or residence in a region endemic for B dermatitidis.With CT scanning, CT-guided biopsy, ultrasonography and antifungal chemotherapy, splenectomy may be avoided in some cases (14)(15).
The present patient was lost to follow-up, but if possible, patients should be followed because of the potential for chronicity or recurrence.DisCLosUre: There is no financial support or proprietary interest to report.
Inc.All rights reserved s al-nassar, t Macnair, J Lipschitz, et al.Blastomycosis presenting as multiple splenic abscesses: Case report and review of the literature.Can J infect Dis Med Microbiol 2010;21(1):53-56.

Figure 1 )
Figure 1) Geographical regions endemic for blastomycosis (modified from references 12 and 34).USA United States of America

Figure 2 )
Figure2) A contrast-enhanced computed axial tomographic scan of the abdomen revealing numerous tiny avascular low-density lesions that appear to be confluent

aCKnoWLeDgeMents:
The authors thank Ms Carolyn Garlinski, Infection Prevention and Control Unit, Health Sciences Centre, Winnipeg, Manitoba, for secretarial assistance and Dr Zhaolin Xu, Department of Pathology, Dalhousie University, Halifax, Nova Scotia, for reviewing the histology.

TAble 1 Cases of blastomycosis with splenic abscess Case (reference) 13,14 15 16 17 Current
*Constitutional: night sweats, fever, chills, malaise and/or fatigue; Pulmonary: thoracic pain, decreased air entry or cough; Abdominal: pain, tenderness, guarding and/or palpable splenomegaly; † Ultrasonographic findings included splenic enlargement, hyper-or hypoechogenicity, and cysts; Abdominal computed tomography (CT) scan findings included splenic enlargement and low-density lesions replacing parenchyma; Radionuclide scanning in one case included a gallium 67 scan (photopenia in spleen) and technetium 99m sulfur colloid scan (splenomegaly and photopenia); Fine-needle aspiration in both cases done was diagnostic of blastomycosis; ‡ Laparotomy showed ascites; splenectomy could not be done because of dense adhesions.NR Not reported; TB Reactive tuberculin test