CASE REPORT
Unexpected Blastomyces dermatitidis Etiology of Fungal Sinusitis and Erosive Palatal Infection in a Diabetic Patient Wheaton Franciscan Laboratory, Milwaukee, Wisconsin, USAa; College of Health Sciences, University of Wisconsin—Milwaukee, Milwaukee, Wisconsin, USAb; Midwest Infectious Disease Associates, S.C., New Berlin, Wisconsin, USAc
We present what is believed to be the initial report of hard-palate infection caused by Blastomyces dermatitidis. The organism was cultivated from biopsy material obtained from a diabetic patient presenting with complaints of headache and malaise. Radiologic findings revealed a malignant-appearing soft-tissue mass with paranasal sinus base destruction.
CASE REPORT
A
79-year-old female presented for evaluation of a 4- to 5-day history of malaise and bilateral frontal lobe headache. Associated pain was characterized as moderate to severe. The patient was withdrawn and uncooperative during the examination. Weakness, dizziness, loss of appetite, and dysphagia also characterized the course of illness. Family members intimated that the patient had lost 30 to 40 pounds over the previous month. Past medical history was significant for type 2 diabetes mellitus, aortic valve replacement, depression, osteoarthritis, and hyperlipidemia. The patient was a nonsmoker and did not consume alcohol. No history of chronic headaches, cerebrovascular accident, transient ischemic attack, and psychiatric or neurologic maladies was revealed. At the time of presentation, the patient resided in a Wisconsin assisted-living facility. The patient was also a past resident of Tennessee, though the timing of this was unclear. Vital signs were stable upon presentation (temperature, 98.3°F; heart rate, 67 beats per min; blood pressure, 143/66), with the exception of slight tachypnea (20 inhalations per min). Pulse oximetry was 98% on 2 liters of supplemental oxygen. A chest X-ray indicated mild chronic obstructive pulmonary disease without acute disease. The patient had elevated laboratory values for peripheral leukocytes (15,800/l, with increased neutrophils [79.0%] and decreased lymphocytes [11.0%] upon differential), serum glucose (226 mg/dl; upper limit of normal, 99 mg/dl), serum C-reactive protein (135.4 mg/liter; upper limit of normal, 8 mg/liter), and Westergren sedimentation rate (92 mm/h; upper limit of normal, 20 mm/h). A decreased serum albumin level (2.8 g/dl; lower limit of normal, 3.2 g/dl) was also documented. Two sets of blood cultures yielded no growth. Due to leukocyte esterase and microscopic leukocyte (5 to 10 per high-power field) findings from a urinalysis, diagnosis of a urinary tract infection was entertained, but culture was not pursued. The patient was treated with empirical vancomycin at 1,250 mg administered intravenously (i.v.) every 12 h (q12h) and levofloxacin at 750 mg i.v. q48h and subsequently discharged. During the hospitalization, the physical finding of an approximately 4-cm-diameter soft granulomatous mass on the hard palate of the posterior pharynx with mild anterior cervical lymphadenopathy prompted outpatient follow-up. An initial maxillofacial computed-tomography (CT) scan exhibited findings most consistent with chronic parasinusitis with extensive opacification of the ethmoid and sphenoid sinuses. Chest radiology was noncontrib-
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utory. Follow-up maxillofacial radiology revealed a malignantappearing soft-tissue mass in the posterior aspect of the hard bony palate, with slight infiltration and destruction of the base of the paranasal sinuses (Fig. 1). At the time of surgical intervention, differential diagnosis included squamous cell carcinoma, metastatic disease, and adenoid cystic carcinoma of the palate. A biopsy specimen excised from the hard palate (Fig. 2) revealed an abundance of granulation tissue, an abundance of inflammatory cells (primarily neutrophils), and rare multinucleated giant cells. Observation of innumerable broad-based budding yeast forms (Fig. 2), subsequently demonstrated to be Gomori methenamine silver stain positive and mucicarmine stain negative, narrowed the preliminary differential diagnosis to fungal disease caused by yeasts (including blastomycosis). This impression was supported by negative results for Cryptococcus neoformans serum antigen testing (1 day postbiopsy) and reactive complement fixation and Blastomyces dermatitidisspecific antigen results available 7 and 10 days postbiopsy, respectively, from reference laboratories. Antigen was quantitated at 12.49 ng/ml from urine (moderately positive reference range, 2.0 to 14.7 ng/ml; MiraVista Diagnostics, Indianapolis, IN). Laboratory diagnosis was confirmed by cultivation of characteristic B. dermatitidis mycelial growth from the biopsy specimen following 13 days of incubation on mycobiotic agar (Remel, Lenexa, KS) in 30°C ambient air and subsequent oligonucleotide hybridization with B. dermatitidis-specific rRNA (Accuprobe Blastomyces Dermatitidis Culture Identification Test; Gen-Probe, Incorporated, San Diego, CA). No fungal studies of respiratory secretions were undertaken. The patient received intravenous liposomal amphotericin B at 5 mg/kg of body weight daily for 6 weeks; step-down fluconazole (1) was administered through a percutaneous gastrostomy tube (400 mg q24h), as additional lesions on her scalp and right foot were also culture positive for B. dermatitidis. Fluconazole was sub-
Received 14 May 2014 Returned for modification 24 May 2014 Accepted 30 May 2014 Published ahead of print 4 June 2014 Editor: P. Bourbeau Address correspondence to Erik Munson, [email protected]. Copyright © 2014, American Society for Microbiology. All Rights Reserved. doi:10.1128/JCM.01392-14
p. 3130 –3133
August 2014 Volume 52 Number 8
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Jane Thomas,a Erik Munson,a,b John C. Christiansonc
Case Report
FIG 1 Sagittal (A), coronal (B), and transverse (C) sections of maxillofacial CT scan with contrast; thick arrows indicate the site of soft-tissue mass, and narrow arrows indicate evidence of bone erosion.
August 2014 Volume 52 Number 8
The dimorphic fungus B. dermatitidis typically exists in warm, moist soil of wooded areas that is rich in organic debris (4–7). While specific gender, season, age, race, or vocation is not typically predictive of the onset of blastomycosis, exposure to soil often provides a common link in reports of sporadic disease and outbreaks (8). Regions of Wisconsin and Tennessee have been classified as areas of high endemicity for blastomycosis (9). Up to half of individuals infected with B. dermatitidis develop an asymptomatic illness. The organism has the potential to affect nearly every organ system during chronic disease (8). Studies of clinical blastomycosis published between 1956 and 1972 reported pulmonary, cutaneous, bone, and genitourinary involvement rates of 52% to 90%, 38% to 80%, 7% to 48%, and 10% to 33%, respectively (10–15). A paradigm shift toward mostly pulmonary disease has occurred in more-recent assessments. A total of 70% of blastomycosis cases in a Canadian survey revealed isolated pulmonary involvement (16). A total of 77% to 91% of single-manifestation blastomycosis in two United States regions of endemicity (17, 18) had pulmonary involvement, while between 0% to 3% and 4% to 6% had exclusive bone and cutaneous involvement, respectively. In their analysis of 326 blastomycosis cases, Chapman et al. (18) reported overall cutaneous and bone involvement rates of only 18% and 4%, respectively. Blastomycosis has the potential to affect most bones, though skeletal disease is typically noted in long bones, vertebrae, and ribs (19). The organism preferentially infects metaphyseal and epiphyseal portions of the bone (20). An approximately 3-decade survey of skeletal blastomycosis at a United States medical center revealed 31 cases; of those cases, 7 (23%) showed skull and facial bone involvement. However, no specific mention was made of palatal involvement (20). With respect to the current case report, a PubMed (United States National Library of Medicine/National Institutes of Health) search utilizing permutations of the terms blastomycosis, Blastomyces dermatitidis, palatal, soft palate, and hard palate uncovered no cases of palatal blastomycosis with a B. dermatitidis etiology. This search found two case series of oral South American blastomycosis (Paracoccidioides brasiliensis etiology) in the dental literature (21, 22). The two papers shared the common theme of chronic, ulcerative P. brasiliensis lesions in Brazilian patients presenting as the first signs and symptoms of South American blastomycosis. de Almeida et al. (21) reported on the clinical course of a patient who presented with oral pain from the maxillary left quadrant and of a second patient with persistent mouth ulcers for several months. Oral ketoconazole therapy brought about disease regression and subsequent resolution within 2 to 4 months. Of 36 patients with
jcm.asm.org 3131
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stituted for itraconazole within the step-down therapeutic component because the patient had poor oral intake and was less likely to tolerate and/or absorb itraconazole liquid formulation. Moreover, high-dose fluconazole regimens have been shown to be effective in the management of non-life-threatening blastomycosis (2, 3). The patient exhibited significant clinical resolution of palatal findings following approximately 4 weeks of amphotericin B therapy. However, final efficacy of the combination antifungal therapy for the palatal blastomycosis could not be determined, as the patient expired less than 3 months later due to myocardial infarction.
Case Report
painful, chronic proliferative or ulcerative oral lesions in the series described by Sposto et al. (22), 17 (47%) exhibited disease in the palate and 23 (64%) had subsequent pulmonary involvement. Clinically apparent oral lesions were confirmed via histologic studies. Additional reports have described palatal paracoccidioidomycosis in United States residents at least 1 decade following residence in Venezuela or Brazil (23, 24). Reder and Neel (25) reviewed 102 cases of blastomycosis at a United States referral hospital over a 10-year period that were confirmed by culture or histologic studies. Twenty-three cases had otolaryngologic manifestations; among these, nearly 70% had skin and mucosal involvement (primarily in the head and neck). Of the subset, 22% had laryngeal involvement. Histopathologic and gross features of laryngeal lesions resembled those of welldifferentiated squamous cell carcinoma. However, no data regarding palatal B. dermatitidis disease were described. Within the 23-patient subset, 17 (74%) had concomitant pulmonary involvement. A total of 83% of patients received either amphotericin (15 patients) or ketoconazole (4 patients) as a primary antifungal regimen. The male/female ratio for otolaryngeal blastomycosis was 2.8:1 in this series. In conclusion, we present a case of erosive palatal blastomycosis in the context of disseminated disease without acute respiratory distress. This presentation is unusual for a number of reasons. First, extrapulmonary blastomycosis is typically observed with active pulmonary infection (8). Second, B. dermatitidis currently has a lesser predilection for skeletal sites than has been described in past literature. Moreover, no specific reports of palatal blastomycosis have been documented. Finally, fungal etiologies of sinus tract disease in diabetic patients commonly include Aspergillus spp. and those involved in rhinocerebral zygomycosis/mucormycosis (26, 27). Clinicians practicing in areas of B. dermatitidis en-
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demicity should include blastomycosis in the differential diagnosis of oral/palatal soft-tissue masses. REFERENCES 1. Chapman SW, Dismukes WE, Prola LA, Bradsher RW, Pappas PG, Threlkeld MG, Kauffman CA. 2008. Clinical practice guidelines for the management of blastomycosis: 2008 update by the Infectious Diseases Society of America. Clin. Infect. Dis. 46:1801–1812. http://dx.doi.org/10 .1086/588300. 2. Pappas PG, Bradsher RW, Kauffman CA, Cloud GA, Thomas CJ, Campbell GD, Jr, Chapman SW, Newman C, Dismukes WE. 1997. Treatment of blastomycosis with higher doses of fluconazole. The National Institute of Allergy and Infectious Diseases Mycoses Study Group. Clin. Infect. Dis. 25:200 –205. 3. Chapman SW, Bradsher RW, Jr, Campbell GD, Jr, Pappas PG, Kauffman CA. 2000. Practice guidelines of the management of patients with blastomycosis. Clin. Infect. Dis. 30:679 – 683. http://dx.doi.org/10.1086 /313750. 4. Dixon DM, Shadomy HJ, Shadomy S. 1977. In vitro growth and sporulation of Blastomyces dermatitidis on woody plant material. Mycologia 69:1193–1195. http://dx.doi.org/10.2307/3758943. 5. Klein BS, Vergeront JM, Weeks RJ, Kumar UN, Mathai G, Varkey B, Kaufman L, Bradsher RW, Stoebig JF, Davis JP. 1986. Isolation of Blastomyces dermatitidis in soil associated with a large outbreak of blastomycosis in Wisconsin. N. Engl. J. Med. 314:529 –534. http://dx.doi.org/10 .1056/NEJM198602273140901. 6. Klein BS, Vergeront JM, DiSalvo AF, Kaufman L, Davis JP. 1987. Two outbreaks of blastomycosis along rivers in Wisconsin. Isolation of Blastomyces dermatitidis from riverbank soil and evidence of its transmission along waterways. Am. Rev. Respir. Dis. 136:1333–1338. 7. Baumgardner DJ, Laundre B. 2001. Studies on the molecular ecology of Blastomyces dermatitidis. Mycopathologia 152:51–58. http://dx.doi.org /10.1023/A:1012438029997. 8. Chapman SW, Sullivan DC. 2010. Blastomyces dermatitidis, p 3319 – 3332. In Mandell GL, Bennett JE, Dolin R (ed), Principles and practice of infectious diseases, 7th ed. Churchill Livingstone, Philadelphia, PA. 9. Rippon JW. 1988. Medical mycology: the pathogenic fungi and pathogenic actinomycetes, 3rd ed, p 478. W. B. Saunders, Philadelphia, PA.
Journal of Clinical Microbiology
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FIG 2 Hematoxylin and eosin stain, hard-palate biopsy specimen—⫻1,000 total magnification.
Case Report
August 2014 Volume 52 Number 8
19. MacDonald PB, Black GB, MacKenzie R. 1990. Orthopaedic manifestations of blastomycosis. J. Bone Joint Surg. Am. 72:860 – 864. 20. Bassett FH, III, Tindall JP. 1972. Blastomycosis of bone. South. Med. J. 65:547–555. http://dx.doi.org/10.1097/00007611-197205000-00009. 21. de Almeida OP, Jorge J, Scully C, Bozzo L. 1991. Oral manifestations of paracoccidioidomycosis (South American blastomycosis). Oral Surg. Oral Med. Oral Pathol. 72:430 – 435. http://dx.doi.org/10.1016 /0030-4220(91)90554-P. 22. Sposto MR, Scully C, de Almeida OP, Jorge J, Graner E, Bozzo L. 1993. Oral paracoccidioidomycosis. A study of 36 South American patients. Oral Surg. Oral Med. Oral Pathol. 75:461– 465. http://dx.doi.org/10.1016 /0030-4220(93)90171-Y. 23. Salman L, Sheppard SM. 1962. South American blastomycosis. Oral Surg. Oral Med. Oral Pathol. 15:671– 676. http://dx.doi.org/10.1016/0030 -4220(62)90247-5. 24. Limongelli WA, Rothstein SS, Smith LG, Clark MS. 1978. Disseminated South American blastomycosis (paracoccidioidomycosis): report of a case. J. Oral Surg. 36:625– 630. 25. Reder PA, Neel HB, III. 1993. Blastomycosis in otolaryngology: review of a large series. Laryngoscope 103(Pt 1):53–58. 26. Latgé J-P. 1999. Aspergillus fumigatus and aspergillosis. Clin. Microbiol. Rev. 12:310 –350. 27. Roden MM, Zaoutis TE, Buchanan WL, Knudsen TA, Sarkisova TA, Schaufele RL, Sein M, Sein T, Chiou CC, Chu JH, Kontoyiannis DP, Walsh TJ. 2005. Epidemiology and outcome of zygomycosis: a review of 929 reported cases. Clin. Infect. Dis. 41:634 – 653. http://dx.doi.org/10 .1086/432579.
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10. Cherniss EI, Waisbren BA. 1956. North American blastomycosis: a clinical study of 40 cases. Ann. Intern. Med. 44:105–123. http://dx.doi.org/10 .7326/0003-4819-44-1-105. 11. Abernathy RS. 1959. Clinical manifestations of pulmonary blastomycosis. Ann. Intern. Med. 51:707–727. http://dx.doi.org/10.7326/0003-4819 -51-4-707. 12. Witorsch P, Utz JP. 1968. North American blastomycosis: a study of 40 patients. Medicine (Baltimore) 47:169 –200. 13. Lockwood WR, Allison F, Jr, Batson BE, Busey JF. 1969. The treatment of North American blastomycosis. Ten years’ experience. Am. Rev. Respir. Dis. 100:314 –320. 14. Duttera MJ, Jr, Osterhout S. 1969. North American blastomycosis: a survey of 63 cases. South. Med. J. 62:295–301. 15. Busey JF. 1972. Blastomycosis. 3. A comparative study of 2-hydroxystilbamidine and amphotericin B therapy. Am. Rev. Respir. Dis. 105:812– 818. 16. Crampton TL, Light RB, Berg GM, Meyers MP, Schroeder GC, Hershfield ES, Embil JM. 2002. Epidemiology and clinical spectrum of blastomycosis diagnosed at Manitoba hospitals. Clin. Infect. Dis. 34:1310 –1316. http://dx.doi.org/10.1086/340049. 17. Baumgardner DJ, Buggy BP, Mattson BJ, Burdick JS, Ludwig D. 1992. Epidemiology of blastomycosis in a region of high endemicity in north central Wisconsin. Clin. Infect. Dis. 15:629 – 635. http://dx.doi.org/10 .1093/clind/15.4.629. 18. Chapman SW, Lin AC, Hendricks KA, Nolan RL, Currier MM, Morris KR, Turner HR. 1997. Endemic blastomycosis in Mississippi: epidemiological and clinical studies. Semin. Respir. Infect. 12:219 –228.
Unexpected Blastomyces dermatitidis Etiology of Fungal Sinusitis and Erosive Palatal Infection in a Diabetic Patient Wheaton Franciscan Laboratory, Milwaukee, Wisconsin, USAa; College of Health Sciences, University of Wisconsin—Milwaukee, Milwaukee, Wisconsin, USAb; Midwest Infectious Disease Associates, S.C., New Berlin, Wisconsin, USAc
We present what is believed to be the initial report of hard-palate infection caused by Blastomyces dermatitidis. The organism was cultivated from biopsy material obtained from a diabetic patient presenting with complaints of headache and malaise. Radiologic findings revealed a malignant-appearing soft-tissue mass with paranasal sinus base destruction.
CASE REPORT
A
79-year-old female presented for evaluation of a 4- to 5-day history of malaise and bilateral frontal lobe headache. Associated pain was characterized as moderate to severe. The patient was withdrawn and uncooperative during the examination. Weakness, dizziness, loss of appetite, and dysphagia also characterized the course of illness. Family members intimated that the patient had lost 30 to 40 pounds over the previous month. Past medical history was significant for type 2 diabetes mellitus, aortic valve replacement, depression, osteoarthritis, and hyperlipidemia. The patient was a nonsmoker and did not consume alcohol. No history of chronic headaches, cerebrovascular accident, transient ischemic attack, and psychiatric or neurologic maladies was revealed. At the time of presentation, the patient resided in a Wisconsin assisted-living facility. The patient was also a past resident of Tennessee, though the timing of this was unclear. Vital signs were stable upon presentation (temperature, 98.3°F; heart rate, 67 beats per min; blood pressure, 143/66), with the exception of slight tachypnea (20 inhalations per min). Pulse oximetry was 98% on 2 liters of supplemental oxygen. A chest X-ray indicated mild chronic obstructive pulmonary disease without acute disease. The patient had elevated laboratory values for peripheral leukocytes (15,800/l, with increased neutrophils [79.0%] and decreased lymphocytes [11.0%] upon differential), serum glucose (226 mg/dl; upper limit of normal, 99 mg/dl), serum C-reactive protein (135.4 mg/liter; upper limit of normal, 8 mg/liter), and Westergren sedimentation rate (92 mm/h; upper limit of normal, 20 mm/h). A decreased serum albumin level (2.8 g/dl; lower limit of normal, 3.2 g/dl) was also documented. Two sets of blood cultures yielded no growth. Due to leukocyte esterase and microscopic leukocyte (5 to 10 per high-power field) findings from a urinalysis, diagnosis of a urinary tract infection was entertained, but culture was not pursued. The patient was treated with empirical vancomycin at 1,250 mg administered intravenously (i.v.) every 12 h (q12h) and levofloxacin at 750 mg i.v. q48h and subsequently discharged. During the hospitalization, the physical finding of an approximately 4-cm-diameter soft granulomatous mass on the hard palate of the posterior pharynx with mild anterior cervical lymphadenopathy prompted outpatient follow-up. An initial maxillofacial computed-tomography (CT) scan exhibited findings most consistent with chronic parasinusitis with extensive opacification of the ethmoid and sphenoid sinuses. Chest radiology was noncontrib-
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utory. Follow-up maxillofacial radiology revealed a malignantappearing soft-tissue mass in the posterior aspect of the hard bony palate, with slight infiltration and destruction of the base of the paranasal sinuses (Fig. 1). At the time of surgical intervention, differential diagnosis included squamous cell carcinoma, metastatic disease, and adenoid cystic carcinoma of the palate. A biopsy specimen excised from the hard palate (Fig. 2) revealed an abundance of granulation tissue, an abundance of inflammatory cells (primarily neutrophils), and rare multinucleated giant cells. Observation of innumerable broad-based budding yeast forms (Fig. 2), subsequently demonstrated to be Gomori methenamine silver stain positive and mucicarmine stain negative, narrowed the preliminary differential diagnosis to fungal disease caused by yeasts (including blastomycosis). This impression was supported by negative results for Cryptococcus neoformans serum antigen testing (1 day postbiopsy) and reactive complement fixation and Blastomyces dermatitidisspecific antigen results available 7 and 10 days postbiopsy, respectively, from reference laboratories. Antigen was quantitated at 12.49 ng/ml from urine (moderately positive reference range, 2.0 to 14.7 ng/ml; MiraVista Diagnostics, Indianapolis, IN). Laboratory diagnosis was confirmed by cultivation of characteristic B. dermatitidis mycelial growth from the biopsy specimen following 13 days of incubation on mycobiotic agar (Remel, Lenexa, KS) in 30°C ambient air and subsequent oligonucleotide hybridization with B. dermatitidis-specific rRNA (Accuprobe Blastomyces Dermatitidis Culture Identification Test; Gen-Probe, Incorporated, San Diego, CA). No fungal studies of respiratory secretions were undertaken. The patient received intravenous liposomal amphotericin B at 5 mg/kg of body weight daily for 6 weeks; step-down fluconazole (1) was administered through a percutaneous gastrostomy tube (400 mg q24h), as additional lesions on her scalp and right foot were also culture positive for B. dermatitidis. Fluconazole was sub-
Received 14 May 2014 Returned for modification 24 May 2014 Accepted 30 May 2014 Published ahead of print 4 June 2014 Editor: P. Bourbeau Address correspondence to Erik Munson, [email protected]. Copyright © 2014, American Society for Microbiology. All Rights Reserved. doi:10.1128/JCM.01392-14
p. 3130 –3133
August 2014 Volume 52 Number 8
Downloaded from http://jcm.asm.org/ on March 28, 2015 by SUNY HSCB MEDICAL RESEARCH LIBRARY
Jane Thomas,a Erik Munson,a,b John C. Christiansonc
Case Report
FIG 1 Sagittal (A), coronal (B), and transverse (C) sections of maxillofacial CT scan with contrast; thick arrows indicate the site of soft-tissue mass, and narrow arrows indicate evidence of bone erosion.
August 2014 Volume 52 Number 8
The dimorphic fungus B. dermatitidis typically exists in warm, moist soil of wooded areas that is rich in organic debris (4–7). While specific gender, season, age, race, or vocation is not typically predictive of the onset of blastomycosis, exposure to soil often provides a common link in reports of sporadic disease and outbreaks (8). Regions of Wisconsin and Tennessee have been classified as areas of high endemicity for blastomycosis (9). Up to half of individuals infected with B. dermatitidis develop an asymptomatic illness. The organism has the potential to affect nearly every organ system during chronic disease (8). Studies of clinical blastomycosis published between 1956 and 1972 reported pulmonary, cutaneous, bone, and genitourinary involvement rates of 52% to 90%, 38% to 80%, 7% to 48%, and 10% to 33%, respectively (10–15). A paradigm shift toward mostly pulmonary disease has occurred in more-recent assessments. A total of 70% of blastomycosis cases in a Canadian survey revealed isolated pulmonary involvement (16). A total of 77% to 91% of single-manifestation blastomycosis in two United States regions of endemicity (17, 18) had pulmonary involvement, while between 0% to 3% and 4% to 6% had exclusive bone and cutaneous involvement, respectively. In their analysis of 326 blastomycosis cases, Chapman et al. (18) reported overall cutaneous and bone involvement rates of only 18% and 4%, respectively. Blastomycosis has the potential to affect most bones, though skeletal disease is typically noted in long bones, vertebrae, and ribs (19). The organism preferentially infects metaphyseal and epiphyseal portions of the bone (20). An approximately 3-decade survey of skeletal blastomycosis at a United States medical center revealed 31 cases; of those cases, 7 (23%) showed skull and facial bone involvement. However, no specific mention was made of palatal involvement (20). With respect to the current case report, a PubMed (United States National Library of Medicine/National Institutes of Health) search utilizing permutations of the terms blastomycosis, Blastomyces dermatitidis, palatal, soft palate, and hard palate uncovered no cases of palatal blastomycosis with a B. dermatitidis etiology. This search found two case series of oral South American blastomycosis (Paracoccidioides brasiliensis etiology) in the dental literature (21, 22). The two papers shared the common theme of chronic, ulcerative P. brasiliensis lesions in Brazilian patients presenting as the first signs and symptoms of South American blastomycosis. de Almeida et al. (21) reported on the clinical course of a patient who presented with oral pain from the maxillary left quadrant and of a second patient with persistent mouth ulcers for several months. Oral ketoconazole therapy brought about disease regression and subsequent resolution within 2 to 4 months. Of 36 patients with
jcm.asm.org 3131
Downloaded from http://jcm.asm.org/ on March 28, 2015 by SUNY HSCB MEDICAL RESEARCH LIBRARY
stituted for itraconazole within the step-down therapeutic component because the patient had poor oral intake and was less likely to tolerate and/or absorb itraconazole liquid formulation. Moreover, high-dose fluconazole regimens have been shown to be effective in the management of non-life-threatening blastomycosis (2, 3). The patient exhibited significant clinical resolution of palatal findings following approximately 4 weeks of amphotericin B therapy. However, final efficacy of the combination antifungal therapy for the palatal blastomycosis could not be determined, as the patient expired less than 3 months later due to myocardial infarction.
Case Report
painful, chronic proliferative or ulcerative oral lesions in the series described by Sposto et al. (22), 17 (47%) exhibited disease in the palate and 23 (64%) had subsequent pulmonary involvement. Clinically apparent oral lesions were confirmed via histologic studies. Additional reports have described palatal paracoccidioidomycosis in United States residents at least 1 decade following residence in Venezuela or Brazil (23, 24). Reder and Neel (25) reviewed 102 cases of blastomycosis at a United States referral hospital over a 10-year period that were confirmed by culture or histologic studies. Twenty-three cases had otolaryngologic manifestations; among these, nearly 70% had skin and mucosal involvement (primarily in the head and neck). Of the subset, 22% had laryngeal involvement. Histopathologic and gross features of laryngeal lesions resembled those of welldifferentiated squamous cell carcinoma. However, no data regarding palatal B. dermatitidis disease were described. Within the 23-patient subset, 17 (74%) had concomitant pulmonary involvement. A total of 83% of patients received either amphotericin (15 patients) or ketoconazole (4 patients) as a primary antifungal regimen. The male/female ratio for otolaryngeal blastomycosis was 2.8:1 in this series. In conclusion, we present a case of erosive palatal blastomycosis in the context of disseminated disease without acute respiratory distress. This presentation is unusual for a number of reasons. First, extrapulmonary blastomycosis is typically observed with active pulmonary infection (8). Second, B. dermatitidis currently has a lesser predilection for skeletal sites than has been described in past literature. Moreover, no specific reports of palatal blastomycosis have been documented. Finally, fungal etiologies of sinus tract disease in diabetic patients commonly include Aspergillus spp. and those involved in rhinocerebral zygomycosis/mucormycosis (26, 27). Clinicians practicing in areas of B. dermatitidis en-
3132
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demicity should include blastomycosis in the differential diagnosis of oral/palatal soft-tissue masses. REFERENCES 1. Chapman SW, Dismukes WE, Prola LA, Bradsher RW, Pappas PG, Threlkeld MG, Kauffman CA. 2008. Clinical practice guidelines for the management of blastomycosis: 2008 update by the Infectious Diseases Society of America. Clin. Infect. Dis. 46:1801–1812. http://dx.doi.org/10 .1086/588300. 2. Pappas PG, Bradsher RW, Kauffman CA, Cloud GA, Thomas CJ, Campbell GD, Jr, Chapman SW, Newman C, Dismukes WE. 1997. Treatment of blastomycosis with higher doses of fluconazole. The National Institute of Allergy and Infectious Diseases Mycoses Study Group. Clin. Infect. Dis. 25:200 –205. 3. Chapman SW, Bradsher RW, Jr, Campbell GD, Jr, Pappas PG, Kauffman CA. 2000. Practice guidelines of the management of patients with blastomycosis. Clin. Infect. Dis. 30:679 – 683. http://dx.doi.org/10.1086 /313750. 4. Dixon DM, Shadomy HJ, Shadomy S. 1977. In vitro growth and sporulation of Blastomyces dermatitidis on woody plant material. Mycologia 69:1193–1195. http://dx.doi.org/10.2307/3758943. 5. Klein BS, Vergeront JM, Weeks RJ, Kumar UN, Mathai G, Varkey B, Kaufman L, Bradsher RW, Stoebig JF, Davis JP. 1986. Isolation of Blastomyces dermatitidis in soil associated with a large outbreak of blastomycosis in Wisconsin. N. Engl. J. Med. 314:529 –534. http://dx.doi.org/10 .1056/NEJM198602273140901. 6. Klein BS, Vergeront JM, DiSalvo AF, Kaufman L, Davis JP. 1987. Two outbreaks of blastomycosis along rivers in Wisconsin. Isolation of Blastomyces dermatitidis from riverbank soil and evidence of its transmission along waterways. Am. Rev. Respir. Dis. 136:1333–1338. 7. Baumgardner DJ, Laundre B. 2001. Studies on the molecular ecology of Blastomyces dermatitidis. Mycopathologia 152:51–58. http://dx.doi.org /10.1023/A:1012438029997. 8. Chapman SW, Sullivan DC. 2010. Blastomyces dermatitidis, p 3319 – 3332. In Mandell GL, Bennett JE, Dolin R (ed), Principles and practice of infectious diseases, 7th ed. Churchill Livingstone, Philadelphia, PA. 9. Rippon JW. 1988. Medical mycology: the pathogenic fungi and pathogenic actinomycetes, 3rd ed, p 478. W. B. Saunders, Philadelphia, PA.
Journal of Clinical Microbiology
Downloaded from http://jcm.asm.org/ on March 28, 2015 by SUNY HSCB MEDICAL RESEARCH LIBRARY
FIG 2 Hematoxylin and eosin stain, hard-palate biopsy specimen—⫻1,000 total magnification.
Case Report
August 2014 Volume 52 Number 8
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