Nocardiosis in ectopic ACTH syndrome: A case report and review of 11 cases from the literature

Abstract

Ectopic adrenocorticotropic hormone (ACTH) syndrome (EAS) associated with nocardiosis is rare, and little information is available regarding its clinical characteristics. In this study, the case of a 35-year-old male patient who showed significant cushingoid features and had a cough with yellow phlegm for 1 month is described. Pulmonary computed tomography (CT) scanning and 18F-fluorodeoxyglucose positron emission tomography combined with CT identified two different lesions in the mediastinum and pulmonary region, respectively. The lesion in the mediastinum was finally diagnosed as an ACTH-secreting mediastinal paraganglioma via biopsy. The sputum culture confirmed pulmonary nocardiosis. The patient was effectively treated with complete tumor resection following the treatment of nocardiosis using trimethoprim-sulfamethoxazole. Following the present case, 11 additional cases of nocardiosis in EAS were identified in the literature and their clinical characteristics were compared and evaluated. It may be concluded that, although Nocardia remains a rare opportunistic infection pathogen in EAS, it is necessary to consider nocardiosis as a diagnosis for patients with pulmonary imaging findings of cavity, consolidation or nodule, particularly when there are brain and extra-pulmonary lesions as well as a poor response to regular treatment.

Cushing’s Syndrome With Nocardiosis: A Case Report and a Systematic Review of the Literature

“…In our CS review, 11 of 18 patients (61.1%) died. The mortality rate was similar with that reported in EAS patients of 66.7% ( 8 ). It is seemed that the mortality rate of nocardiosis in CS patients is higher than that in other immunocompromised patients of 34.5–40% ( 3 , 4 ).…”

“…Seventeen CS with nocardiosis patients from 15 published reports ( 8 , 10 – 23 ) were reviewed. Together with our case, 18 CS patients (11 male, 7 female) were identified.…”

“…Nodules, masses, cavitations, infiltration, consolidation, and pleural effusion could be radiographic presentations of pulmonary nocardiosis. Xu L. et al ( 8 ) proposed cavity lesions, consolidation/infiltration, and nodule/mass were the major findings for EAS patients. The most common findings were cavitary lesion and nodules in our review.…”

“…Nocardiosis is regarded as an opportunistic infection, with the majority of infections occurring in immunocompromised patients, including those with long-term corticosteroid exposure, malignancy, human immunodeficiency virus (HIV) infection, and history of transplantation (3)(4)(5)(6)(7), associated with high mortality of 34.5-40% (3,4). Xu L. et al (8) reviewed 12 patients of nocardiosis in EAS patients. However, reports of nocardiosis in patients with other forms of CS were not included.…”

The Role of 18-Fluorodeoxyglucose Positron Emission Tomography/Computed Tomography (FDG-PET/CT) in Management of Nocardiosis: A Retrospective Study and Review of the Literature

“…All publications were case reports of individual patients. Nine were published between 2004 and 2014 [19][20][21][22][23][24][25][26][27], five were published in 2015 [28][29][30][31][32] and six were published between 2016 and 2020 [33][34][35][36][37][38]. These 20 case reports are summarized in Table 2.…”