A Rare Presentation of Possible Disseminated Histoplasmosis with Adrenal Insufficiency Leading to Adrenal Crisis in an Immunocompetent Adult: A Case Report

Abstract

Histoplasmosis is caused by Histoplasma capsulatum, and commonly it causes an asymptomatic illness. Although Histoplasma is the commonest organism to infect adrenal glands, disseminated histoplasmosis in an immune-competent host leading to adrenal insufficiency is rare in current literature. Here, we report a case of possible disseminated histoplasmosis leading to adrenal crisis in a young Asian immunocompetent male. A 42-year-old Sri Lankan male with noninsulin-dependent diabetes mellitus presented with constitutional symptoms and feverishness for three weeks’ duration. He was found to have hepatosplenomegaly with bilateral adrenal masses and pancytopenia. One week later, he developed severe vomiting and hemodynamic collapse and was found to have suppressed adrenal functions. Ultrasound-guided biopsy of adrenals showed fungal spores morphologically similar to Histoplasma. He was started on oral itraconazole and adrenal replacement therapy. He improved symptomatically with treatment and currently is on regular clinic follow-up with a plan to continue antifungal therapy for at least one year to prevent a relapse. Fungal infections, especially histoplasmosis, need to be considered in all immunocompetent patients with compatible history and bilateral adrenal masses. Adrenal insufficiency needs to be promptly diagnosed and treated to prevent Addisonian crisis in these patients.

Disseminated histoplasmosis with adrenal insufficiency in an immunocompetent adult – A case report

“…A similar case of adrenal histoplasmosis in an immunocompetent Srilankan male has been previously described who also improved with antifungals [9]. Another case series described four cases of Addison's disease due to adrenal histoplasmosis, of which one person died [10].…”

Adrenal Histoplasmosis: An Uncommon Cause of Adrenal Crisis in an Immunocompetent Man

“…Treatment depends on disease severity and condition of the patient with amphotericin used initially in severe infections, followed by maintenance prolonged (6-24 months) itraconazole therapy. 13 Our patient had localized disease and therefore was started on itraconazole as the first-line treatment. While high remission rate can be achieved with antifungal therapy, overall mortality remains significant.…”

“…[10][11][12] Diagnosis of adrenal histoplasmosis could be achieved by multiple methods including histopathology, specific fungal stains such as GMS, cultures, antigen detection, serological tests, and molecular biology-based tests. 13,14 The sensitivity and specificity of these methods are dependent on the patient's immune status, disease manifestation, and duration of symptoms prior to presentation. The sensitivity of Histoplasma urine antigen test increases in immunocompromised patients and in those with disseminated histoplasmosis.…”

Case Report of Disseminated Adrenal Histoplasmosis and Secondary Adrenal Insufficiency

“…Though if symptoms do manifest, patients with adrenal histoplasmosis may present with non-specific symptoms of fatigue, weight loss, nausea, and orthostasis for months. To differentiate fungal infection from the malignancy of the adrenal gland, the adrenal biopsy gland may be particularly useful, especially in immunocompetent individuals [ 8 ]. Typical imaging findings of adrenal histoplasmosis on CT or MRI may involve bilateral adrenal enlargement, peripheral enhancement, and central hypodensity due to necrosis and/or hemorrhage [ 5 ].…”