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Table of Contents   
CASE REPORT  
Year : 2013  |  Volume : 6  |  Issue : 4  |  Page : 479-481
A case of clinically inapparent bilateral adrenal histoplasmosis in an Immunocompetent person


1 Department of Urology, Apollo Hospitals, Chennai, Tamil Nadu, India
2 Department of Infectious disease, Apollo Hospitals, Chennai, Tamil Nadu, India
3 Department of Pathology, Apollo Hospitals, Chennai, Tamil Nadu, India

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Date of Web Publication26-Feb-2014
 

   Abstract 

Histoplasmosis presenting as bilateral large adrenal masses is rare. We describe the case of a 35-year-old male with bilateral adrenal masses which turned out to be histoplasmosis on biopsy, so the masses were treated with antifungal agents. We describe the case for its rarity.

Keywords: Adrenal, bilateral, histoplasmosis, immunocompetent, itraconazole

How to cite this article:
Shringarpure S, Thachil JV, Gaffur A, Geetha. A case of clinically inapparent bilateral adrenal histoplasmosis in an Immunocompetent person. Ann Trop Med Public Health 2013;6:479-81

How to cite this URL:
Shringarpure S, Thachil JV, Gaffur A, Geetha. A case of clinically inapparent bilateral adrenal histoplasmosis in an Immunocompetent person. Ann Trop Med Public Health [serial online] 2013 [cited 2020 Sep 23];6:479-81. Available from: http://www.atmph.org/text.asp?2013/6/4/479/127805

   Introduction Top


Histoplasmosis is a fungal infection caused by Histoplasma capsulatum, which commonly presents as transient pulmonary infection that usually subsides without treatment. Disseminated histoplasmosis may affect almost all systems, including the reticuloendothelial system, lungs, gastrointestinal tract, renal tract, central nervous system, bone marrow, and adrenal glands. Adrenal gland is frequently involved in disseminated histoplamosis but commonly present as unilateral mass; bilateral involvement is rare. It is also rare in immunocompetent hosts and only few cases have been reported in past. We report a rare case of bilateral adrenal masses which turned out to be H. capsulatum in an immunocompetent patient. The masses resolved completely on treatment with antifungal agents.


   Case Report Top


A 35-year non-diabetic, normotensive male who was a farmer by occupation from north-eastern part of India, presented with epigastric discomfort of 6-months duration. There was no past history of tuberculosis. On examination, there was no hepatosplenomegaly or generalized lymphadenopathy. Blood count reports were within normal limits. The workup for tuberculosis and HIV was negative. Ultrasound abdomen and CT scan Abdomen showed bilateral adrenal masses with central hypodensities [Figure 1]. Serum Calcium, DHEA, Cortisol, thyroid functions and 24-hr urinary metanephrines were within normal limits. His HIV serology was negative. CT chest and echocardiogram did not reveal any primary source of infection. CT-guided biopsy of the adrenal masses showed cluster of macrophages with abundant cytoplasm containing yeast form of H. capsulatum [Figure 2]. Gomori Methanamine silver staining was positive for histoplasmosis [Figure 3]. Culture-confirmed H. capsulatum [Figure 4] Investigating retrograde for the other sites - there was no skin, oropharyngeal, or lung involvement (CT chest was normal). The patient also had no Addisonian features despite bilateral involvement. The patient was treated with Itraconazole (200 mg daily) for 6 months and he responded well with resolution of the masses on repeat CT scan.
Figure 1: CT scan Abdomen showed bilateral adrenal masses with central hypodensities

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Figure 2: CT guided biopsy of the adrenal masses showed cluster of macrophages with abundant cytoplasm containing yeast form of histoplasma capsulatum. (H& E × 400)

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Figure 3: Gomori Methanamine silver staining positive for histoplasmosis

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Figure 4: Culture showing spores of Histoplasma capsulatum

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   Discussion Top


Histoplasmosis presenting as bilateral adrenal enlargement has been previously described. [1],[2] Also, this patient had selective involvement of both the adrenals without involvement of the lungs, oropharngeal or skin.

In our case, the presentation of bilateral adrenal masses and abdominal pain, made us consider the differential diagnoses of adrenal tumors-benign or malignant, pheochromocytoma, adrenal hyperplasia, disseminated infection as tuberculosis, fungal infections.

Regarding endemicity, the patient was a farmer and was from north-eastern part of India, where histoplasmosis has been found to be endemic in West Bengal and the organism (H. capsulatum) has been isolated from the soil in Gangetic plain. [3]

The CT scan features of adrenal histoplasmosis include preserved, enlarged adrenals with central hypodensities, and peripheral enhancement. [4] Although CT scan cannot rule out other infective causes.

FDG-PET is a useful modality to distinguish benign from malignant adrenal lesions in patients with incidentally detected adrenal masses on CT scan or MRI with sensitivity and specificity for malignant adrenal lesions being 100% and 80-100%, respectively. Increased FDG uptake in the adrenals is also seen in inflammatory or infective conditions of the adrenals. Umeoka et al., reported a case of bilateral adrenal histoplasmosis with increased FDG uptake. [5] In our case, 18-FDG PET study was not done due to the unaffordability of the patient.

Percutaneous image-guided biopsy of the adrenals remains a safe and useful procedure in diagnosing metastasis, tumors, and infections in patients with nonspecific features on imaging. [6] One has to rule out pheochromocytoma before doing a biopsy. Sometimes FNAB may be inconclusive and may require an open or laparoscopic adrenal biopsy. The potential disadvantages of FNAB include high false negative rate, bleeding, and hemodynamic catastrophe if the lesion is a pheochromocytoma. Histopathological examination shows that the intracellular forms are situated within the cytoplasm of histiocytes, where they appear as numerous small spherical or oval yeast forms surrounded by a clear ring of space that resembles a capsule, hence the misnomer, H. capsulatum.

According to the latest Infectious Diseases Society of America/American Thoracic Society (IDSA/ ATS 2007) guidelines, Itraconazole is the reasonable first choice of antifungal agent against disseminated histoplasmosis. There have been reports of complete resolution of adrenal masses due to histoplasmosis with itraconazole. [7]

This case emphasizes the fact that adrenal histoplasmosis does occur in immunocompetent patients and has to be considered in the differential diagnosis of bilateral adrenal masses.


   Conclusion Top


Bilateral adrenal masses in an immunocompetent and nontubercular patient with variable clinical presentation should raise the possibility of adrenal histoplasmosis and should be confirmed with a FNAB of the adrenals.

 
   References Top

1.Mukherjee JJ, Villa ML, Tan L, Lee KO. Bilateral adrenal masses due to histoplasmosis. J Clin Endocrinol Metab 2005;90:6725-6.  Back to cited text no. 1
    
2.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. 2
    
3.Sanyal M, Thanmayya A. Histoplasma capsulatum in the soil of Gangetic plain in India. Indian J Med Res 1975;63:1020-8.  Back to cited text no. 3
    
4.Levine E. CT evaluation of active adrenal histoplasmosis. Urol Radiol 1991;13:103-6.  Back to cited text no. 4
    
5.Umeoka S, Koyama T, Saga T, Higashi T, Ito N, Kamoto T, et al. High 18F-fluorodeoxyglocoseuptake in adrenal histoplasmosis: A case report. Eur Radiol 2005;15:2483-6.  Back to cited text no. 5
    
6.Paulsen SD, Nghiem HV, Korobkin M, Caoili EM, Higgins EJ. Changing role of imaging-guided percutaneous biopsy of adrenal masses: Evaluation of 50 adrenal biopsies. AJR Am J Roentgenol 2004;182:1033-7.  Back to cited text no. 6
    
7.Mandell LA, Wunderink RG, Anzueto A, Bartlett JG, Campbell GD, Dean NC, et al. Infectious Diseases Society of America, American Thoracic Society. Infectious diseases society of America/American thoracic society consensus guidelines on the management of community-acquired pneumonia in adults. Clinil Infect Dis 2007;44:S27-72.  Back to cited text no. 7
    

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Correspondence Address:
Sanish Shringarpure
27 Vishal Nagar, Bijapur road, Solapur-413 004, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1755-6783.127805

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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