Bilateral adrenal histoplasmosis in a Malaysian tertiary hospital: Report of four cases

Abstract

Rationale: Adrenal histoplasmosis can present as bilateral adrenal enlargement with constitutional symptoms and/or adrenal insufficiency. Often these patients are initially investigated as secondary adrenal metastases before eventually their diagnosis established by histopathological examination of the adrenal tissues.

Patients concerns: We report 4 cases of patients aged 55-78 who presented with hypocortisolism and bilateral adrenal masses.

Diagnoses: Bilateral adrenal histoplasmosis.

Interventions: Three patients received antifungal treatment for at least one year while 1 patient passed away before the initiation of antifungal treatment.

Outcomes: Two patients’ adrenal function recovered not requiring any steroid replacement, while 1 patient still requires long-term steroid replacement.

Lessons: Adrenal histoplasmosis should be excluded in patients from endemic areas presenting with bilateral adrenal masses and adrenal insufficiency. Usually patients have an exposure to bird droppings, bat guano or soil contaminated with histoplasmosis fungi. Histopathological examination of biopsied adrenal tissues is still the gold standard investigation as serology and molecular diagnostic methods are still not widely available in most centers in Malaysia.

Keywords

Adrenal histoplasmosis / Histoplasmosis / Bilateral adrenal masses

References

[1] Baker J, Setianingrum F, Wahyuningsih R, Denning DW. Mapping histoplasmosis in South East Asia-implications for diagnosis in AIDS. Emerg Microbes Infect 2019; 1(1): 1139-1145.

[2] Bahr NC, Antinori S, Wheat LJ, Sarosi GA. Histoplasmosis infections worldwide: Thinking outside of the Ohio River Valley. Curr Trop Med Rep 2015; 2(2): 70-80.

[3] Ewa AU, Ekeng B, Bassey G, Akpah E, Asemota O, Nweke L. Disseminated histoplasmosis in a 17-year-old Nigerian male patient: A case report. Asian Pac J Trop Med 2022; 6(6): 283-286.

[4] Lenhart WL, Schafer MP, Singal M, Hajjeh R. Histoplasmosis: Protecting workers at risk. [Online]. Available from: htps:/stacksc.gov/ivw/cl.65686dc_6568DS1.pdf. [Accessed on 20 May 2023].

[5] Gajendra S, Sharma R, Goel S, Goel R, Lipi L, Sarin H, et al. Adrenal histoplasmosis in immunocompetent patients presenting as adrenal insufficiency. Turk Patoloji Derg 2016; 2(2): 105-111.

[6] Gupta S, Ansari MAM, Gupta AK, Chaudhary P, Bansal LK. Current approach for diagnosis and treatment of adrenal tuberculosis-our experience and review of literature. Surg J (N Y) 2022; 1(1): e92-e97.

[7] Altinmakas E, Guo M, Kundu UR, Habra MA, Ng C. Computed tomography and 18F-fluorodeoxyglucose positron emission tomography/ computed tomography findings in adrenal candidiasis and histoplasmosis: Two cases. Clin Imaging 2015; 6(6): 1115-1118.

[8] Tsai YJ, Lin YH, Hsu CH, Yeh S Der. 18F-fluorodeoxyglucose positron emission tomography for the initial evaluation and monitoring of therapeutic response in bilateral adrenal histoplasmosis. Clin Imaging 2013; 4(4): 791-793.

[9] Azar MM, Hage CA. Laboratory diagnostics for histoplasmosis. J Clin Microbiol 2017; 6(6): 1612-1620.

[10] Kulshreshtha B, Kothari D, Chopra S, Bhardwaj M, Ajmani A. Persistence of histoplasma in adrenals 7 years after antifungal therapy. Indian J Endocrinol Metab 2013; 17(3): 529.

[11] Singh M, Chandy DD, Bharani T, Marak RSK, Yadav S, Dabadghao P, et al. Clinical outcomes and cortical reserve in adrenal histoplasmosis-A retrospective follow-up study of 40 patients. Clin Endocrinol 2019; 4(4): 534-541.