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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.

SUBMITTER: Jayathilake WAPP

PROVIDER: S-EPMC7103037 | biostudies-literature | 2020

REPOSITORIES: biostudies-literature

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

Jayathilake Wanasinghe Arachchilage Praneeth Prabash WAPP Kumarihamy Kulatunga Wijekoon Mudiyanselage Pramitha Prabhashini KWMPP Ralapanawa Dissanayake Mudiyanselage Priyantha Udaya Kumara DMPUK Jayalath Widana Arachchilage Thilak Ananda WATA

Case reports in medicine 20200317

Histoplasmosis is caused by <i>Histoplasma capsulatum</i>, and commonly it causes an asymptomatic illness. Although <i>Histoplasma</i> 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 ...[more]

PMID: 32256604

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Project description:INTRODUCTION:Histoplasmosis is a rare fungal disease caused by dimorphic fungi Histoplasma capsulatum. The causative fungus persists in soil, infects through inhalation and manifests in three main types-acute primary, chronic cavitary and progressive disseminated histoplasmosis. Disseminated Histoplasmosis (DH) is defined as a clinical condition where the fungus is present in more than one location. Among the forms of histoplasmosis, DH is the rarest and mostly found in an immuno-compromised individual. Here we are presenting our experiences of the series of cases of DH in immuno-competent individuals who have been diagnosed in our institute in last 5 years. MATERIALS AND METHODS:This is a single centre retrospective observational study, conducted in Institute of Haematology and Transfusion Medicine, which is a referral centre for Eastern India, from May 2009 to April 2014. Only cases with DH in otherwise healthy immuno-competent individuals were included in the study. The histoplasmosis was confirmed either by presence of Histoplasma in biopsy specimen from an extrapulmonary organ or by positive growth in fungal culture. RESULT:Total seven patients met the inclusion criteria. Five out of 7 patients were male. The mean age was 35 years. Five of the 7 patients presented with fever for a long duration. Six patients complained of significant weight loss before diagnosis. On examination, one patient had skin nodules, five patients had hepato-splenomegaly, and two patients had lymphadenopathy. The laboratory investigations revealed anaemia in six out of 7 patients, and pancytopenia in 3 patients. Two patients had features of the hemophagocytic syndrome in the bone marrow. All patients were treated with conventional amphotericin B deoxycholate and azole antifungal. One patient with adrenal involvement died in hospital. The patient with skin nodule had recurrent relapses. The other patients had resolution of symptoms and were clinically cured. CONCLUSION:DH is not an uncommon aetiology of fever of prolonged duration even in immuno-competent individual and should be kept as a differential diagnosis. Targeted investigation through early bone marrow biopsy and fungal culture may help in the diagnosis of DH. Imaging study to exclude adrenal involvement prevents case fatality. Cytopenia may be due to a secondary hemophagocytic syndrome, which improves with anti-fungal therapy. Treatment with either amphotericin B or itraconazole gives excellent outcome though therapy may have to be given for a prolonged period in case of relapses.

Project description:BackgroundIn patients with adrenal insufficiency (AI), adrenal crisis (AC) represents a clinical emergency. Early recognition and prompt management of AC or AC-risk conditions in the Emergency Department (ED) can reduce critical episodes and AC-related outcomes. The aim of the study is to report the clinical and biochemical characteristics of AC presentation to improve their timely recognition and proper management in a ED setting.MethodsSingle-centre, retrospective, observational study on pediatric patients followed at the Department of Pediatric Endocrinology of Regina Margherita Children's Hospital of Turin for primary AI (PAI) and central AI (CAI).ResultsAmong the 89 children followed for AI (44 PAI, 45 CAI), 35 patients (21 PAI, 14 CAI) referred to the PED, for a total of 77 accesses (44 in patients with PAI and 33 with CAI). The main causes of admission to the PED were gastroenteritis (59.7%), fever, hyporexia or asthenia (45.5%), neurological signs and respiratory disorders (33.8%). The mean sodium value at PED admission was 137.2 ± 1.23 mmol/l and 133.3 ± 1.46 mmol/l in PAI and CAI, respectively (p = 0.05). Steroids administration in PED was faster in patients with CAI than in those with PAI (2.75 ± 0.61 and 3.09 ± 1.47 h from PED access, p = 0.83). Significant factors related to the development of AC were signs of dehydration at admission (p = 0.027) and lack of intake or increase of usual steroid therapy at home (p = 0.059). Endocrinological consulting was requested in 69.2% of patients with AC and 48.4% of subjects without AC (p = 0.032).Conclusionchildren with AI may refer to the PED with an acute life-threatening condition that needs prompt recognition and management. These preliminary data indicate how critical the education of children and families with AI is to improve the management at home, and how fundamental the collaboration of the pediatric endocrinologist with all PED personnel is in raising awareness of early symptoms and signs of AC to anticipate the proper treatment and prevent or reduce the correlated serious events.

Dataset Information

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

Publications

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

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