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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 6  |  Issue : 1  |  Page : 72-74

Adrenal histoplasmosis with disseminated cutaneous manifestations in an immunocompetent patient: A case report


1 Department of Microbiology, Institute of Postgraduate Medical Education and Research, Kolkata, West Bengal, India
2 Department of Microbiology, National Medical College, Kolkata, West Bengal, India

Date of Web Publication8-Dec-2014

Correspondence Address:
Kalidas Rit
70B TC Mukherjee Street, PO - Rishra, Hooghly - 712 248, West Bengal
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0975-9727.146470

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  Abstract 

This report describes a case of adrenal histoplasmosis along with disseminated cutaneous involvement in a 48-year-old immunocompetent male. The patient presented with multiple erythematous nodules on the skin of the scalp, face, back, thigh, abdomen, and soles. Abdominal computed tomography (CT) imaging revealed bilateral adrenomegaly, with hepatosplenomegaly. Histological examination of the biopsy specimen from both the skin lesion and CT-guided fine-needle aspiration cytology (FNAC) aspirates of the adrenal gland showed a granulomatous infiltrate with numerous intracellular Periodic Acid Schiff (PAS)-positive rounded yeast cells within the macrophages. Culturing of these materials, incubated in Sabouraud's agar, showed growth of Histoplasma capsulatum. Treatment with itraconazole showed an excellent response.

Keywords: Adrenal, erythematous nodules, histoplasmosis, itraconazole


How to cite this article:
Rit K, Naha A, Saha R. Adrenal histoplasmosis with disseminated cutaneous manifestations in an immunocompetent patient: A case report. Muller J Med Sci Res 2015;6:72-4

How to cite this URL:
Rit K, Naha A, Saha R. Adrenal histoplasmosis with disseminated cutaneous manifestations in an immunocompetent patient: A case report. Muller J Med Sci Res [serial online] 2015 [cited 2019 Nov 14];6:72-4. Available from: http://www.mjmsr.net/text.asp?2015/6/1/72/146470


  Introduction Top


Histoplasmosis is a deep mycotic infection caused by genus Histoplasma having two varieties namely H. capsulatum var capsulatum found in America and the Tropics and H. capsulatum var duboisii found in Africa. [1] The disease is also endemic in certain parts of the Asia. [1] H. capsulatum usually causes pulmonary involvement resembling tuberculosis and may pass asymptomatically to involve the reticuloendothelial system and the skin. [2] H. duboisii causes cutaneous and osseous involvement, and is restricted to the African continent. [2] The pulmonary and disseminated forms of histoplasmosis are very common in acquired immunodeficiency syndrome (AIDS) patients, with great morbidity and mortality. [3] Primary adrenal histoplasmosis with cutaneous involvement is rare in immunocompetent patients. We report a case of adrenal histoplasmosis with disseminated cutaneous involvement caused by H. capsulatum in an immunocompetent patient, on account of its rarity.


  Case Report Top


A 48-year-old, non-diabetic normotensive male presented with low-grade, intermittent fever, and loss of weight and appetite, of seven months' duration. He had already received antitubercular treatment, with four drugs, for three months, without any remission of fever. A general examination revealed erythematous nodules on the skin of the scalp, face, back, thighs, abdomen, and soles [Figure 1]. Routine blood examination, urine examination, renal function test, and chest X-rays were all normal. The blood culture was sterile. The tests for malaria, tuberculosis, and human immunodeficiency virus (HIV) were negative. The peripheral blood smear examination and dual antigen assay for malaria were negative. The enzyme-linked immunosorbent assay (ELISA) test for detecting the antibody against HIV1 and HIV2 was nonreactive. The Mantoux test result was negative. Contrast-enhanced computed tomography (CECT) of the abdomen showed hepatosplenomegaly with enlarged hypoattenuated bilateral adrenal glands. The adrenal endocrine profile was normal. Transabdominal CT-guided fine-needle aspiration cytology (FNAC) revealed numerous organisms ranging in size from 2-4 um in a necrotic background. Histopathology of the lesion showed a chronic granulomatous infiltrate with PAS-positive intracellular organisms, with a clear halo [Figure 2]. A tissue culture for fungus on Sabouraud's dextrose agar (SDA), at 25˚ C, yielded the typical, cottony white, buff colony in four weeks [Figure 3]. The Lactophenol cotton blue preparation, with growth from the SDA slope, under a high power objective of the microscope showed typical tuberculate macroconidia [Figure 4]. Material from the fungal colony, cultured in the Brain-Heart Infusion Agar, containing 10% sheep blood with added glutamine, at 37˚ C, showed the formation of slowly growing creamy yeast-like colonies. From the growth, narrow-based, ovoid and budding yeast cells were demonstrated micropscopically. Repeat culture from these yeast-like colonies on the SDA slope, at 25˚ C, yielded cottony, buffy, mycelial growth, thus confirming dimorphism. In our study the diagnosis of histoplasmosis was made by radiological examination, histopathological examination, and fungal cultures of the appropriate specimens. The patient was treated with itraconazole 200 mg, twice daily, and clinical improvement was seen within six weeks.
Figure 1: Cutaneous manifestation of disseminated histoplasmosis

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Figure 2: PAS-stained smear showing degenerating H. capsulatum organisms on a necrotic background

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Figure 3: Colony of H. capsulatum on an SDA slope

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Figure 4: Lactophenol cotton blue preparation showing typical tuberculate macroconidia

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  Discussion Top


Histoplasmosis is widely distributed throughout the world, occurring in more than 60 temperate and tropical countries in USA, Africa, and Australia. [1] In India, histoplasmosis seems to be prevalent in the Gangetic delta. Panja and Sen reported the first case of disseminated histoplasmosis from Calcutta, in 1954. [4] Since then, individual cases have been reported from various states, mostly from West Bengal. [5] There are sporadic reports from Andhra Pradesh and Bihar as well. [6],[7] In West Bengal a previous study showed skin positivity to the histoplasmin antigen in 9.4% of the persons studied. [8] Among the forms of histoplasmosis reported from India, the disseminated one is the rarest. Cases of adrenal histoplasmosis with disseminated cutaneous manifestations in immunocompetent patients are only occasionally reported from India. [1],[5] There have also been reports of disseminated mucocutaneous histoplasmosis in South India. [7]

Histoplasmosis is usually a self-limiting pulmonary infection, which is usually asymptomatic and can be associated with mild influenza-like symptoms. There are three major clinical presentations: Pulmonary, progressive disseminated, and primary cutaneous histoplasmosis. [9] The progressive disseminated form of the disease is rare and occurs in immunocompromised patients in the endemic areas. [3] Primary cutaneous histoplasmosis is very rare and occurs due to local trauma or inoculation and can present with nodules, ulcers, abscesses or Molluscum contagiosum-like lesions. Disseminated histoplasmosis can present with maculopapular rash. The interesting feature in our patient is that there is a presence of disseminated cutaneous nodules along with bilateral adrenal involvement. We are reporting this case because of its rarity and clinical significance.

The diagnostic modalities include cultures, fungal stains of tissue or body fluids, and tests for antigens and antibodies. Serological tests for the detection of antibodies are nonspecific in histoplasmosis and require at best a month to appear after the initial exposure. The facilities to perform serological tests for diagnosis of H. capsulatum infection are currently not available in our institution. On CT scan imaging, both adrenals usually show uniformly bilateral symmetrical enlargement, with preservation of the contour of the gland and central hypodensity with peripheral enhancement. [10] The differential diagnoses of bilateral adrenomegaly are metastasis, lymphoma, adrenal hemorrhage, sarcoidosis, and infections, which include histoplasmosis, tuberculosis, cryptococcosis, coccidioidomycosis, and blastomycosis. [11] However, central hypodensity and peripheral rim enhancement of the adrenals narrow down the differentials only to tuberculosis and histoplasmosis. As H. capsulatum is an intracellular dimorphic fungus, it is commonly seen within the cytoplasm of the macrophages, exhibiting a narrow-based budding. In the fine-needle aspiration smears, these organisms can also be seen extracellularly, as the fragile cytoplasm of the macrophages may get disrupted at the time of making the smears. In our case, diagnosis was established by positive histopathological findings on both the skin biopsy material and examination of the CT-guided FNAC-aspirated material and growth of H. capsulatum on the SDA agar slope, thus excluding the tubercular etiology. Lactophenol cotton blue preparation of growth under the high power objective of microscope from the SDA slope showed typical tuberculate macroconidia [Figure 4]. Also the patient was unresponsive to anti-TB chemotherapy.

Histoplasmosis usually responds to itraconazole, fluconazole or amphotericin B. [12] Our patient showed an excellent response to itraconazone 200 mg BD. The treatment was well-tolerated, without any serious adverse effects.

In endemic regions, the possibility of histoplasmosis must always kept in mind when a patient presents with enlarged, hypoattenuating bilateral adrenal glands, with peripheral rim enhancement and enhancing internal septations. Image-guided FNAC will demonstrate the disease and it must be performed without delay, as a great number of patients with adrenal histoplasmosis may develop life-threatening adrenal insufficiency, if untreated.


  Acknowledgment Top


The authors would like to thank Professor Prasanta Kumar Maiti, Professor and Head, Department of Microbiology, Institute of Postgraduate Medical Education and Research. 244 AJC Bose Road. Kolkata - 700 020.

 
  References Top

1.
Kumar N, Singh S, Govil S. Adrenal histoplasmosis: Clinical presentation and imaging features in nine cases. Abdom Imaging 2003;28:703-8.   Back to cited text no. 1
    
2.
Hay RJ, Moore M. Mycology. In: Champion RH, Burton JL, Burns DA, Breathnach SM, editors. Rook/Wilkinson/Ebling's Textbook of Dermatology. 6th ed. Oxford: Blackwell Science; 1998. p. 1363-6.  Back to cited text no. 2
    
3.
Kalter DC, Tschen JA, Klima M. Maculopapular rash in a patient with acquired immunodeficiency syndrome. Disseminated histoplasmosis in acquired immunodeficiency syndrome (AIDS). Arch Dermatol 1985;121:1455-6, 1458-9.   Back to cited text no. 3
    
4.
Panja G, Sen S. A unique case of histoplasmosis. J Indian Med Assoc 1954;23:257-8.  Back to cited text no. 4
    
5.
Dhar S, Roy RK, Todi SK, Roy S, Dhar S. Seven cases of histoplasmosis: Cutaneous and extracutaneous involvements. Indian J Dermatol 2006;51:137-9.   Back to cited text no. 5
  Medknow Journal  
6.
Saoji AM, Grover S, Mene AR, Patoria NK, Sharma KD. Histoplasmosis. J Indian Med Assoc 1976;66:37-8.  Back to cited text no. 6
    
7.
Thankappan TP, Vijayalekshmi A, Nair R. Chronic progressive desseminated mucocutaneous histoplasmosis. Indian J Dermatol Venereol Leprol 1983;49:27-9.   Back to cited text no. 7
    
8.
Sanyal M, Thammayya A. Skin sensitivity to histoplasmin in Calcutta and its neighbourhood. Indian J Dermatol Venereol Leprol 1980;46:94-8.   Back to cited text no. 8
  Medknow Journal  
9.
Hay RJ, Robert SO, Mackenzie DW. Mycology. In: Champion RH, Burton JL, Ebling FJ, editors Textbook of Dermatology. 5 th ed. London: Blackwell Scientific Publications; 1992. p. 1200-2.  Back to cited text no. 9
    
10.
Rozenblit AM, Kim A, Tuvia J, Wenig BM. Adrenal histoplasmosis manifested as Addison's disease: Unusual CT features with magnetic resonance imaging correlation. Clin Radiol 2001;56:682-4.  Back to cited text no. 10
    
11.
Schonfield AD, Jackson JA, Smith DJ, Hurley DL. Disseminated histoplasmosis with bilateral adrenal enlargement: Diagnosis by computed tomography-directed needle biopsy. Tex Med 1991;87:88-90.   Back to cited text no. 11
    
12.
Negroni R, Palmieri O, Koren F, Tiraboschi IN, Galimberti RL. Oral treatment of paracoccidiodomycosis and histoplasmosis with itraconazole in humans. Rev Infect Dis 1987;9 (Suppl 1):S47-50.  Back to cited text no. 12
    


    Figures

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



 

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