INTRODUCTION: Histoplasmosis is the most common mycotic infection in the United States and is especially prevalent in the Ohio and Mississippi river valleys. Most cases are asymptomatic benign pulmonary infections incidentally found in immune competent hosts. Disseminated histoplasmosis infections are usually found in patients that are immune compromised by medications, cancer, and HIV infection. Fungal particles found in dust and soil are in inhaled. As a result the lower respiratory tract the most common site for infection with extrapulmonary histoplasmosis often affecting the tongue and buccal mucosa. This case illustrates an unusual presentation of disseminated histoplasmosis initially presenting as oral bleeding from raised lesion of the epiglottis.
CASE PRESENTATION: This is a 72 year old male with history of coronary artery disease, valvular heart disease and chronic kidney disease stage III initially presented to his local emergency department after spontaneously developing painless oral bleeding. He denied any trauma, constitutional abnormalities, cough or dyspnea. No lesions or source of bleeding were found at that time. He did have an elevated INR that was corrected and the bleeding subsided. During the following week he redeveloped intermittent oral bleeding and returned for evaluation. His INR was 2 and again oropharyngeal examination did not reveal the source of bleeding. A nasopharyngoscopy was performed that showed epiglottic thickened with ulceration and a right true vocal fold with a mounded soft tissue mass concerning for malignancy. The patient was taken to the operating room for biopsies of these lesions and surrounding regions of the posterior pharynx. All of the biopsies revealed extensive infiltration of histoplasmosis. The patient continued to have oral bleeding post-operatively and remained intubated for airway protection. Over the course of the next three days he continued to have extensive oral bleeding requiring repeated packing despite a normal coagulation profile. He was alert and oriented during this time and tolerated his antifungal therapy. On his fourth post-operative day his mental status declined and eventually became obtunded off of any sedative medications. A head CT and brain MRI did not show any abnormalties responsible for the patient's condition. A lumbar puncture was performed for further evaluation of his mental status change. The cerebral spinal fluid (CSF) had no organisms seen and the white blood cell count was 3, but his histoplasmosis antigen was positive. Urine, serum, and bronchoalveolar lavage specimens were negative for histoplasmosis and its antigen. HIV testing was also negative. The patient’s mental status did not improve and a repeat lumbar puncture was performed to confirm the diagnosis of central nervous system (CNS) histoplasmosis. The CSF now revealed acute inflammation with leukocytosis and the histoplasmosis antigen was again positive. Despite treatment, his clinical condition worsened and the patient expired.
DISCUSSION: Histoplasmosis is a common organism in the United States with positive histoplasmin skin testing in as many as 90 percent of the people living endemic areas. It usually does not cause disseminated infection in immune competent patients and in those that do develop active infection, presenting symptoms often include malaise, low grade fevers, and dyspnea. The respiratory tract is the most common site for lesions since the organism enters the body by inhalation. Non bleeding oral lesions are more common the HIV positive patients, but can occur in any patient. Usually the tongue and buccal mucosa are affected and the lesions can be misdiagnosed as malignant on first appearance. The organism migrates through the bloodstream to involved many organ systems. Central nervous system infection occurs in approximately 5 percent of cases with disseminated disease and yields a poor prognosis. Histoplasmosis needs to be considered in endemic regions since special testing is often required. Antigen testing from urine and serum as well as other body fluids can support to the diagnosis of disseminated infection. Early and aggressive antifungal therapy with prolonged duration is necessary in disseminated infection, especially with CNS involvement.
CONCLUSIONS: The diagnosis of disseminated histoplasmosis can be difficult in an immune competent patient since symptoms may be vague and mimic many disease processes. The organism is not often reveal itself on standard cultures. A hightened index of suspicion for infection with histoplasmosis is necessary in endemic regions, since aggressive antifungal therapy is required.
Reference #1 Oropharyngeal histoplasmosis: report of eleven cases and review of the literature. Rev Soc Bras Med Trop. 2011 Jan-Feb;44(1):26-9.
Reference #2 L. J. Wheat, et al. Diagnosis and Management of Central Nervous System Histoplasmosis. Clin Infect Dis. (2005) 40 (6): 844-52.
DISCLOSURE: The following authors have nothing to disclose: Brian Mieczkowski, Matthew Exline
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