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Table of Contents
CASE REPORT
Year : 2019  |  Volume : 16  |  Issue : 2  |  Page : 109-113

Bilateral adrenal histoplasmosis in a diabetic patient: A case report and review of cases in India


1 Department of Microbiology, Indraprastha Apollo Hospitals, New Delhi, India
2 Department of Internal Medicine, Indraprastha Apollo Hospitals, New Delhi, India
3 Department of Pathology, Indraprastha Apollo Hospitals, New Delhi, India
4 Department of Radiodiagnosis, Indraprastha Apollo Hospitals, New Delhi, India

Date of Submission06-Mar-2019
Date of Acceptance29-Apr-2019
Date of Web Publication19-Jun-2019

Correspondence Address:
Hena Butta
Department of Microbiology, Indraprastha Apollo Hospitals, New Delhi - 110 076
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/am.am_8_19

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  Abstract 


Disseminated histoplasmosis usually affects immunocompromised and immunosuppressed individuals and may involve various organ systems of our body. The definitive diagnosis of histoplasmosis is very challenging because it usually presents with nonspecific symptoms such as malignancies, tuberculosis, sarcoidosis, and other chronic infections. Hereby, we report a culture-proven case of bilateral adrenal histoplasmosis in a diabetic but immunocompetent patient. The patient showed good response to treatment with antifungal drugs.

Keywords: Adrenal, diabetic, Histoplasma, immunocompetent, India


How to cite this article:
Butta H, Chatterjee S, Mendiratta L, Gilotra K, Jadhav L, Rawat V, Sardana R. Bilateral adrenal histoplasmosis in a diabetic patient: A case report and review of cases in India. Apollo Med 2019;16:109-13

How to cite this URL:
Butta H, Chatterjee S, Mendiratta L, Gilotra K, Jadhav L, Rawat V, Sardana R. Bilateral adrenal histoplasmosis in a diabetic patient: A case report and review of cases in India. Apollo Med [serial online] 2019 [cited 2019 Aug 18];16:109-13. Available from: http://www.apollomedicine.org/text.asp?2019/16/2/109/260691




  Introduction Top


Histoplasmosis is a systemic type of endemic mycosis caused by Histoplasma capsulatum which has a worldwide distribution. It is a thermally dimorphic fungus which exists in yeast form inside the human body and at 37°C and mold form in the environment and at 25°C–30°C. Usually, infection by Histoplasma occurs by the inhalation of microconidia into alveoli, but infection may also occur by other routes such as fomites, direct inoculation, solid organ transplant, and sexual contact. In the warm and moist environment in alveoli, the spores germinate and convert to yeast forms. The yeasts are then taken up by alveolar macrophages where replication of yeasts occurs. After replication within the macrophages, dissemination occurs using the reticuloendothelial system to regional lymph nodes and other organs of the body.[1] However, as a result of host immune response, the growth of yeasts is stopped within 1–2 weeks of exposure, and delayed type of hypersensitivity to Histoplasma antigens develops after 3–4 weeks of exposure.[2] Disseminated histoplasmosis is the extrapulmonary form of histoplasmosis and usually affects immunocompromised and immunosuppressed individuals such as patients with acquired immunodeficiency syndrome (AIDS), posttransplant patients, and patients with serious underlying disorders. Disseminated histoplasmosis may involve various organ systems of our body such as central nervous system (CNS), ocular system, gastrointestinal tract, and genitourinary tract. However, most of the patients with disseminated histoplasmosis presents with bilateral adrenal masses, and adrenal gland is involved in 80% of the cases.[3] A patient with disseminated histoplasmosis usually presents with nonspecific symptoms such as malignancies, tuberculosis, sarcoidosis, and other chronic infections.[4] The clinical manifestations include fever, anorexia, weight loss, hepatosplenomegaly, and lymphadenopathy. Hereby, we report a case of adrenal form of disseminated histoplasmosis in a diabetic but immunocompetent patient.


  Case Report Top


A 56-year-old male, resident of Dehradun, Uttarakhand, India, presented with complaints of weight loss, decreased appetite, and malaise for the past 6 months. The patient gave a history of 6–7 kg weight loss in the past 6 months. He also complained of on and off cough with sputum for 3 months which gets aggravated at night. He was a known case of type II diabetes for the past 15 years and hypertension for the past 10 years. There was no history of fever and hemoptysis. He was an engineer by profession and was not involved in fieldwork. On physical examination at our institute, he was conscious, pale, febrile, and normotensive. Laboratory investigations showed normocytic normochromic anemia, total leukocyte count, differential leukocyte count, liver function test, and kidney function test within normal limits. Erythrocyte sedimentation rate was 97 mm/1st h, and glycosylated Hemoglobin (HbA1c) was 7.5%. Blood culture was negative for bacterial and fungal growth. Positron-emission tomography–computed tomography (CT) of the whole body showed hepatomegaly, peripherally fluorodeoxyglucose-avid bilateral adrenal mass lesion and bilateral lung nodules [Figure 1]. Chest findings showed few centrilobular and endarterial lesions in the right lower lobe, left upper lobe and lingula, and few subcentimetric mediastinal lymph nodes.
Figure 1: Positron-emission tomography–computed tomography showing hepatomegaly, peripherally fluorodeoxyglucose-avid bilateral adrenal mass lesion and bilateral lung nodules

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CT-guided fine-needle aspiration cytology of the left adrenal mass was consistent with inflammatory smear of fungal etiology with morphology closest to histoplasmosis [Figure 2]. CT-guided left adrenal mass core biopsy tissue was sent for microbiological examination. Fungal smear examination showed small yeast cells. Fungal culture was done on Sabouraud dextrose agar (SDA) and brain heart infusion blood agar (BHI-BA) and kept at 25°C and 37°C. After 15 days of incubation, mycelial growth was observed at 25°C SDA tube. The tube was further reincubated. The growth was white in color, suede-like, and showed slightly yellow reverse [Figure 3]. After 20 days of incubation, Lactophenol Cotton Blue (LPCB) mount was made, and subculture was done on BHI-BA in duplicate and kept at both 25°C and 37°C. LPCB mount of mycelial growth showed large, rounded, single-celled, tuberculate macroconidia and few small round microconidia [Figure 4]. Subculture done on BHI-BA at 37°C showed growth of smooth cream-colored pasty colonies after 8 days of incubation which were microscopically seen as yeast cells in LPCB mount [Figure 5]. Based on the dimorphic nature of the fungi and its macroscopic and microscopic morphological characteristics, the fungus was identified as H. capsulatum. It is noteworthy to mention that all the inoculation procedures were done with extreme caution in class II biological safety cabinet since the initial processing of specimen.
Figure 2: Periodic acid–Schiff staining showing intracellular round yeast cells in clusters (×1000)

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Figure 3: Growth on Sabouraud dextrose agar tubes

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Figure 4: Lactophenol Cotton Blue mount showing tuberculate macroconidia (×400)

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Figure 5: Lactophenol Cotton Blue mount showing yeast cells (×400)

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The patient was tested for immunodeficiency. HIV was nonreactive, and CD3, CD4, and CD8 cell counts were found to be within normal limits.

The patient was initially treated with amphotericin B, but due to the development of progressive acute kidney injury, amphotericin B was stopped and oral itraconazole was started. The patient was given oral itraconazole 200 mg twice a day. The patient's kidney functions were improved, and the patient showed good clinical response to treatment. The patient was discharged in hemodynamically stable and afebrile condition. The patient was discharged on itraconazole 200 mg twice a day with proper monitoring. His treatment continued for 1 year, and the patient is on regular follow-up till date.


  Discussion and Review of Cases in India Top


The natural habitat of the mycelial form of H. capsulatum is soil with high nitrogen content, such as areas contaminated with bird and bat guano, and is associated with bird roosts, caves, and dilapidated buildings. There are three varieties of H. capsulatum, namely var. capsulatum, var. duboisii, and var. farciminosum, of which var. farciminosum is pathogenic to animals. Histoplasma capsulatum var. capsulatum is endemic in the Eastern United States and Latin America, and H. capsulatum var. duboisii is endemic in Central and Western Africa; the majority of cases have been reported from Nigeria. Globally, histoplasmosis is endemic in central and eastern states of the United States, South America, Africa, and Asia. In India, most of the cases have been reported from the north and north-east states along the rivers Ganges, Yamuna, and Brahmaputra.[5] There are three major clinical presentations: pulmonary, progressive disseminated, and cutaneous histoplasmosis. Pulmonary histoplasmosis is the most common form of histoplasmosis and is usually self-limiting. Patients are mostly asymptomatic and may present with mild influenza-like illness.[6] Progressive disseminated form of the disease is rare and may manifest as chronic disease in immunocompetent host or acute progressive disease in immunosuppressed hosts.

Disseminated histoplasmosis may affect reticuloendothelial system, lungs, gastrointestinal tract, urinary tract, CNS, bone marrow, and adrenal glands. The adrenal glands are frequently involved through hematogenous route in disseminated histoplasmosis mainly in immunocompromised patients such as AIDS patients, transplant recipients, those with hematologic malignancies, and patients on corticosteroids. Although rare, adrenal histoplasmosis has been reported in immunocompetent patients in India as well as in other countries. [Table 1] depicts the reports of adrenal histoplasmosis in otherwise immunocompetent patients from India.
Table 1: Reports of adrenal histoplasmosis from India

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Cutaneous histoplasmosis occurs mainly in association with pulmonary and disseminated form and rarely as primary cutaneous histoplasmosis.[15]

The differential diagnosis of adrenal histoplasmosis includes primary adrenal malignancy, tuberculosis, adrenal blastomycosis, and adrenal coccidioidomycosis. In a country like India where tuberculosis is very common, it is often misdiagnosed as tuberculosis and the patient is put on antitubercular therapy. We report a culture-proven case of adrenal histoplasmosis in a diabetic immunocompetent patient from Uttarakhand state which is the progenitor of Ganga River. Immunocompetent patients may also present with adrenal histoplasmosis. Diabetes may be a predisposing factor in immunocompetent patients. Niknam et al. had also found diabetes as a risk factor for disseminated histoplasmosis in an immunocompetent patient from nonendemic area.[16]


  Conclusion Top


Adrenal histoplasmosis is not much uncommon in India, and it may affect immunocompetent individuals also. A high degree of clinical suspicion and complete clinicomicrobiological workup of the patient is required for the definitive diagnosis and management of the patients with histoplasmosis. Diabetes may be a risk factor for disseminated histoplasmosis. amphotericin B and itraconazole are the drug of choice for the treatment. If treated timely, it has good prognosis in immunocompetent patients.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Rog CJ, Rosen DG, Gannon FH. Bilateral adrenal histoplasmosis in an immunocompetent man from Texas. Med Mycol Case Rep 2016;14:4-7.  Back to cited text no. 1
    
2.
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Gupta A, Ghosh A, Singh G, Xess I. A twenty-first-century perspective of disseminated histoplasmosis in India: Literature review and retrospective analysis of published and unpublished cases at a tertiary care hospital in North India. Mycopathologia 2017;182:1077-93.  Back to cited text no. 5
    
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Mahajan R, Sharma U, Trivedi N, Prasad M, Kansra U, Bhandari S, et al. Histoplasma capsulatum in adrenal gland aspirate – A case report. Indian J Pathol Microbiol 2000;43:165-8.  Back to cited text no. 7
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Sharma SK, Malav IC, Banga A, Gulati M. Bilateral adrenal abscesses and skin lesions in an immunocompetent patient. Lancet Infect Dis 2005;5:126.  Back to cited text no. 8
    
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Dhar S, Roy RK, Todi SK, Roy S, Dhar S. Seven cases of histoplasmosis: Cutaneous and extracutaneous involvement. Indian J Dermatol 2006;51:137-9.  Back to cited text no. 10
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Narang V, Sinha T, Sandhu AS, Karan SC, Srivastava A, Sethi GS, et al. Clinically inapparent bilateral adrenal masses due to histoplasmosis. Eur Urol 2009;55:518-21.  Back to cited text no. 11
    
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Rana C, Kumari N, Krishnani N. Adrenal histoplasmosis: A diagnosis on fine needle aspiration cytology. Diagn Cytopathol 2011;39:438-42.  Back to cited text no. 12
    
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Ahuja A, Mathur SR, Iyer VK, Sharma SK, Kumar N, Agarwal S. Histoplasmosis presenting as bilateral adrenal masses: Cytomorphological diagnosis of three cases. Diagn Cytopathol 2012;40:729-31.  Back to cited text no. 13
    
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Bhansali A, Das S, Dutta P, Walia R, Nahar U, Singh SK, et al. Adrenal histoplasmosis: Unusual presentations. J Assoc Physicians India 2012;60:54-8.  Back to cited text no. 14
    
15.
Vasudevan B, Ashish B, Amitabh S, Mohanty AP. Primary cutaneous histoplasmosis in a HIV-positive individual. J Glob Infect Dis 2010;2:112-5.  Back to cited text no. 15
    
16.
Niknam N, Malhotra P, Kim A, Koenig S. Disseminated histoplasmosis presenting as diabetic keto-acidosis in an immunocompetent patient. BMJ Case Rep 2017;2017. pii: bcr2016217915.  Back to cited text no. 16
    


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