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| CASE REPORT |
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| Year : 2017 | Volume
: 7
| Issue : 1 | Page : 48-52 |
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Disseminated histoplasmosis in an immunocompetent individual presenting as oropharyngeal mass
Rateesh Sareen1, Menka Kapil1, GN Gupta1, Anurag Govil2
1 Department of Pathology and Transfusion Medicine, Santokba Durlabhji Memorial Hospital, Jaipur, Rajasthan, India
2 Department of Gastroenterology, Santokba Durlabhji Memorial Hospital, Jaipur, Rajasthan, India
| Date of Web Publication | 3-Jul-2017 |
Correspondence Address:
Rateesh Sareen
Department of Pathology and Transfusion Medicine, Santokba Durlabhji Memorial Hospital, Jaipur, Rajasthan
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/ijohs.ijohs_6_17
We report a case of disseminated histoplasmosis in a 70 year old immunocompetent male patient. Generally most of the cases of Histoplasmosis in India present with skin lesions and oral ulcers, mass lesions are uncommon presentation. The close masquerade with malignancy makes it an interesting rare case with lot of academic learning for considering Histoplasmosis as one of the differential diagnosis in oral mass lesions.
Keywords: Histoplasmosis, immunocompetent, oropharyngeal mass
How to cite this article:
Sareen R, Kapil M, Gupta G N, Govil A. Disseminated histoplasmosis in an immunocompetent individual presenting as oropharyngeal mass. Int J Oral Health Sci 2017;7:48-52 |
How to cite this URL:
Sareen R, Kapil M, Gupta G N, Govil A. Disseminated histoplasmosis in an immunocompetent individual presenting as oropharyngeal mass. Int J Oral Health Sci [serial online] 2017 [cited 2023 Feb 1];7:48-52. Available from: https://www.ijohsjournal.org/text.asp?2017/7/1/48/209354 |
| Introduction | |  |
Histoplasmosis is a systemic mycotic infection caused by Histoplasma capsulatum, a dimorphic fungus.[1] In soil, it develops as branching forms and assumes yeast-like form in host. It most commonly presents as a pulmonary and progressive pulmonary disseminated (PPD) forms.[2] PPD is mainly seen in immunocompromised form in HIV-infected individuals. Other manifestations of PPD include skin lesions, fever, malaise, hepatosplenomegaly, lymphadenopathy, disseminated intravascular coagulation, renal failure, pancytopenia, gastrointestinal (GI) disturbances such as vomiting, diarrhea, neurological manifestations such as encephalopathy, focal parenchymal lesions, and adrenal insufficiency.[3] We report a case of disseminated histoplasmosis in an immunocompetent host with unusual presentation as an oropharyngeal mass lesion. We report a case of histoplasmosis presenting in an unusual manner as an oropharyngeal mass; the presentation itself makes the case interesting as it is rare to find an infectious agent as a cause of mass lesion.
| Case Report | | |
A 70-year-old male farmer from Churu district in Rajasthan was admitted in a tertiary care hospital in Jaipur, Rajasthan, with complaints of generalized weakness and loss of appetite since a month, difficulty in deglutination, initially to solids and progressively to both solids and liquids and constipation for the past 15 days. He was on antihypertensive for the past 8 years. Personal history was positive for beedi smoking for the past 30 years with an average of one pack per day (one pack containing forty beedis).
Examination
At the time of admission on day 0, general physical examination revealed pallor. The vitals were as follows: Blood pressure - 130–70 mmHg, pulse - 91/min, respiratory rate - 24/min, and temperature - 98.6°F. Rest of the general and physical examination was normal without any hepatosplenomegaly or lymphadenopathy. The chest examination revealed hyperinflation, diffusely decreased breath sounds, and prolonged expiration. Occasionally, coarse crackles at the beginning of inspiration were heard.
Investigations
Laboratory workup done revealed red blood cell - 3.09 × 106/Ul, hemoglobin - 8.7 g/dl, hematocrit - 24.4%, total leukocyte count - 3970/mm 3, normal differential count, and platelet count - 1,50,000/mm 3. ESR was within normal limits. Complete urine examination showed albuminuria Grade + 1, with 24 h protein excretion 950 mg/dl. Renal function tests, liver function tests, random sugar, and coagulation profile were all within normal limits. HIV antibody, hepatitis B antigen, and hepatitis C antibody test were all negative. There were no acid-fast bacilli on sputum examination, and blood culture was negative on culture.
An upper GI endoscopy performed revealed a friable growth in oropharynx was seen measuring 2.8 cm × 1.2 cm × 1 cm in size. The growth was rising above the epiglottis. Buccal mucosa was normal. The proximal portion of esophagus was unremarkable [Figure 1]. Thick whitish slough was seen throughout esophagus. Stomach and duodenum were normal. Ultrasonography abdomen showed age-related early medical renal disease. Computed tomography chest examination revealed multiple lytic lesions in right anterior, second, fifth, and sixth rib and proximal sternum, fibrotic lesions in both upper lobes, and superior segment of left lower lobe - Koch's sequel. Mild centriacinar emphysematous changes in both upper lobes were seen. There was mild bilateral pleural effusion along with Micronodularity in right middle lobe and both lower lobes which were suggestive of chronic bronchitis or small airway disease. Two-dimensional echocardiography showed aortic cusps sclerosed with intact intra-auricular and interventricular septum. Left ventricular ejection fraction was 60% and pulmonary artery systolic pressure was 40 mm of Hg. There was grade I Left Ventricular (LV) dysfunction with mild Tricuspid regurgitation. No thrombus, vegetation, or pericardial effusion or regional wall motion was noted. | Figure 1: Upper gastrointestinal endoscopy showing growth above epiglottis
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The biopsy from oropharyngeal mass reveals an inflammatory granulomatous tissue composed of lymphocytes, plasma cells, neutrophils, and histiocytes. In few foci, cytoplasm of macrophages shows rounded structures with clear capsule [Figure 2],[Figure 3],[Figure 4]. These rounded structures stain positive with MSN stain and periodic acid-Schiff (PAS). Thus, a diagnosis of histoplasmosis was made [Figure 5]. The patient was given intravenous antifungal treatment. | Figure 2: H and E stained section area in circle shows intracellular organisms (×40)
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 | Figure 3: H and E stained section arrowhead shows round microorganisms (×40)
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 | Figure 4: Intracellular location of microorganisms on Hand E section (x40)
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Fungus epidemiology
Systemic histoplasmosis also known as Darling's disease was first described by Samuel Taylor Darling, a pathologist, in 1905. He examined viscera and bone marrow of a young man whose death was attributed to tuberculosis. On microscopy, small capsulated intracellular organisms were noted whom he named Histoplasma capsulatum.[4] Histoplasmosis is a dimorphic fungus which has two distinct pathogenic forms H. capsulatum var Capsulatum, endemic in Eastern US and Latin America [5] and H. capsulatum var Duboisii, prevalent in Africa and South East Asia.[6] In India, H. capsulatum is known to be endemic and most cases were reported from West Bengal and Assam.[7] The low awareness of the disease and misdiagnosis as tuberculosis or leishmaniasis makes the disease go unrecognized making the true prevalence of this mycosis underreported.[8],[9] The first case of histoplasmosis in our country was reported in 1954.[10] Disseminated histoplasmosis is a rare finding in immunocompetent host.[11] In India, histoplasmosis is rare as it is thought to be endemic only in small regions in West Bengal in Western India.[12]
Etiopathogenesis
The fungus grows in warm and humid environments contaminated by bird or bat excreta. Primary mode of infection in histoplasmosis is either inhalation of fungal conidia or mycelia fragment. Once inhaled these fungal elements transform in yeast form and may disseminate systemically. Primary site of infection is usually the lungs. The air-borne spores - microconidia of the fungus are inhaled which enter in terminal bronchioles and are engulfed by alveolar macrophages in alveoli. Microconidia then transform into yeast forms. This subsequently generates an inflammatory response in the host. The yeast-like forms reach the regional lymph nodes gaining access to the hematogenous route eventually harboring reticuloendothelial cells and internal organs. The inflammatory response by the host destroys the infectious organism and arrests infection from progression leading to latency. The immunocompetence of the host results in granuloma formation which undergoes fibrosis and calcification. The progressive disseminated infection may involve central nervous system, GI tract including oral mucosa, liver, spleen, kidney, adrenal gland, and lymph node.[13] Excavation and construction disrupt soil and releases organisms which through inhalational route infest healthy individuals.[14]
Signs and symptoms
Fever, headache, cough, and chest pain are most common symptoms. The disease is often self-limiting pulmonary infection which is often asymptomatic or mild influenza-like illness. Pulmonary involvement varies from mild pneumonia to acute respiratory syndrome.[15] The immune status of the host determines the extent of disease. Oral lesions are seen in 25%–40% of patients with Histoplasma infection.[16] Oral manifestation of histoplasmosis, although usually described associated with the chronic disseminated form of the disease, constitutes a rare event in HIV-negative patient, without subjacent clinical disorder. Disseminated histoplasmosis with oral involvement in an immunocompetent patient has rarely been described worldwide.[17]
Oral lesions in histoplasmosis can involve buccal mucosa, tongue, and palate as chronic nonhealing ulcer or as a mass lesion. The lesion can manifest in a variety of forms such as ulcers, erythematous, or vegetative nodule or wart-like growth. The differential diagnosis includes aphthous or traumatic ulcer, ulcerative necrotic gingivitis, other mycoses, squamous cell carcinoma, and lymphomas. The lesions are initially budding wart like and gradually progress to ulcerated, painful ulcers with rolled, and firm margins. The oral manifestations are considered as localized disease by authors whereas few authors suggest that they arise from hematogenous spread from an unknown infectious focus. The presentation of Histoplasma infection in the form of oral lesions and a mass lesion is very rare. The disease has a male preponderance as supported by studies from Patil et al. with median age incidence from 26 to 65 years of age.[18]
Diagnostic workup
Histoplasmosis can be diagnosed based on clinical signs and symptoms, chest X-ray, blood and bone marrow smear examination, histopathology, fungal culture, serology test such as complement fixation test, immunodiffusion, and histoplasmin skin test.
Histoplasma skin testing was the earliest diagnostic modality as supported in studies in India by Panja and Sen, Taneja et al. (1955), Viswanathan et al. (1960) and Pandalai et al. (1962).[19] Histoplasmin skin test has limited value as no longer reagents available.[20]
Serology test has limited value in HIV patient because of decreased antibody production. Direct immunofluorescence is diagnostic in case of HIV patient.
Histopathology is the prime investigating modality as identification of histoplasmosis in the sections provides conclusive evidence of disease.[21] Studies' histopathological examination of the infected tissue usually demonstrates noncaseating granulomatous inflammation. Numerous small (1–2 μ in diameter) yeast-like microorganisms surrounded by the characteristic clear halo may be seen within macrophage, multinucleate giant cells, or within fibrous tissue.[22] Special stains such as Gomori methenamine silver (GMS) or PAS are required to confirm the organism in tissues. GMS should be done in all suspected cases as PAS may not pick up scant organisms. Fungal culture remains a gold standard diagnostic test. Culture is done on enriched agar base.[23]
Treatment
Treatment modality of histoplasmosis shows that in immunocompetent patient without AIDS, amphotericin B is effective by 68%–72%, itraconazole 100%, and ketoconazole by 56%–70%, whereas in AIDS patient, amphotericin B is effective by 74%–84% and itraconazole by 85%.
| Conclusion | | |
The rare presentation of histoplasmosis as a mass lesion in oral cavity posed a diagnostic challenge. Thus, a complete clinical knowledge about oral histoplasmosis is important in diagnosis and preventing further dissemination and fulmination of fatal disease. People who are immunocompromised or prone to disease should avoid soil excavation sites. High level of awareness on the clinical part to prevent misdiagnosis as tuberculosis is also required.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
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