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Objective: Increased visceral fat and sarcopenia are cardiovascular risk factors that may explain increased cardiovascular morbidity and frailty in patients with adrenal adenomas. Our objective was to compare body composition measurement of patients with adrenal adenomas to referent subjects without adrenal disease Design: Cross-sectional study, 2014-2018 Methods: Participants were adults with nonfunctioning adrenal tumor (NFAT), mild autonomous cortisol secretion (MACS) and Cushing syndrome (CS), and age, sex and body mass index 1:1 matched referent subjects without adrenal disorders. Main outcome measures were body composition measurements calculated from abdominal computed tomography imaging. Intraabdominal adipose tissue and muscle mass measurements were performed at the 3rd lumbar spine level. Results: Of 227 patients with adrenal adenomas, 20 were diagnosed with CS, 76 with MACS and 131 with NFAT. Median age was 56 years (range, 18-89), and 67% were women. When compared to referent subjects, patients with CS, MACS, and NFAT demonstrated a higher visceral fat (odds ratio (OR) of 2.2 [95% CI 0.9-6.5], 2.0 [1.3-3.2], and 1.8 [1.2-2.7] and a lower skeletal muscle area (OR of 0.01 [95% CI 0-0.09], 0.31 [0.18-0.49], and 0.3 [1.2-2.7]), respectively. For every 1 mcg/dL cortisol increase after overnight dexamethasone, visceral fat/muscle area ratio increased by 2.3 (P=0.02) and mean total skeletal muscle area decreased by 2.2cm2 (P=0.03). Conclusion: Patients with adrenal adenomas demonstrate a lower muscle mass and a higher proportion of visceral fat when compared to referent subjects, including patients with NFAT. Even a subtle abnormality in cortisol secretion may impact health of patients with adenomas.

SummaryTo assess continuous subcutaneous hydrocortisone infusion (CSHI) in patients with adrenocortical insufficiency (AI) and difficulties with oral replacement. Three patients with AI and frequent hospital admissions attributed to adrenal crises were treated with CSHI, which was delivered via a continuous subcutaneous infusion. All three patients preferred CSHI and remained on it long term, which permitted prolonged follow-up analysis. All three patients reported symptomatic improvement, and in two cases, reduced hospital admission rates and inpatient stay lengths were observed. The cost of hospital admissions and overall treatment was reduced in all cases. CSHI offers a practical and acceptable alternative to oral replacement in a subset of patients with AI. The cost of initiating and maintaining the pump is offset in the long term by reduced frequency and duration of emergency admissions. CSHI can therefore be considered in a select group of patients who are resistant to treatment with conventional oral glucocorticoids.Learning points Continuous subcutaneous infusion of cortisol is a viable alternative in patients unable to take oral steroids.Patient acceptability was high, with three out of three patients preferring to remain on pump treatment.Hospital admissions were reduced in response to pump therapy, which compensated for the increased treatment cost.The daily dosage of hydrocortisone can be reduced by using pump therapy.

Rationale: Sclerosing pneumocytoma is a rare benign lung neoplasm seen in middle aged adults with a female predominance. Originally thought to be vascular in origin, this rare entity is now understood to be epithelial in nature. On imaging, sclerosing pneumocytoma manifests as a well circumscribed nodule or mass, often juxtapleural in location. On histopathology, sclerosing pneumocytoma is composed of cuboidal “surface cells” and round “stromal cells,” both of which show nuclear staining for thyroid transcription factor-1 (TTF-1). Here we review the existing literature on sclerosing pneumocytoma and present a case of sclerosing pneumocytoma in a highly unusual endobronchial location. Patient concerns: This case is a 43 year old woman who presented with chronic cough. Diagnosis: Imaging revealed a right upper lobe nodule with an endobronchial component. Interventions and outcomes: Endoscopic biopsy was performed, and pathologic diagnosis was confirmed. Lessons: Although extremely rare, endobronchial presentation of sclerosing pneumocytoma is possible, and should remain on the differential for patients with endobronchial pulmonary lesions. Pathologic tissue analysis is necessary to confirm this uncommon diagnosis.

Background: Adrenal incidentalomas are mostly non-functioning adrenal tumors (NFAT) or adenomas with mild autonomous cortisol excess (MACE), of which the natural history is unclear. We conducted a systematic review and meta-analysis to study the long-term follow-up of NFAT and MACE to determine: (i) proportion of tumor growth and malignant transformation, (ii) incident change of hormone function, and (iii) incident cardio-metabolic comorbidities and mortality. Methods: Literature search of multiple databases including MEDLINE, EMBASE, Cochrane, and Scopus (January 1990 to February 2018). We included studies of adults with NFAT or MACE (as defined by authors), with ≥20 patients undergoing conservative management, and reported outcomes of interest at baseline and after ≥12 months follow-up. Results: We included 31 studies reporting on 4138 patients with incidental NFAT and MACE; 61.9% females, mean age 60.1 years, and mean follow-up 48.9 months. Overall, the data quality was medium-high, but there was significant heterogeneity in MACE and comorbidities definitions. Mean tumor growth in NFAT and MACE was 1.8mm over 60.6 months, which was similar between both groups. Adenomas were less likely to grow when the baseline mean size was ≥2.5cm, compared to smaller adenomas (<2.5 cm); mean size difference 0.8mm and 2.3mm, respectively. While 10% of patients demonstrated tumor enlargement, growth of ≥1cm occurred in only 4.5% over 62.0 months, which was relatively higher in MACE (MACE 4.5% vs. NFAT 2.3%). None of 2690 NFAT and MACE patients developed adrenal malignancy over 42.4 months. Development of overt Cushing’s syndrome and pheochromocytoma was very rare (0.4%). None of 2690 patients developed primary hyperaldosteronism. Of 2088 NFAT patients, 5.6% developed MACE, while pre-existing MACE resolved in 1.9% of 780 patients during 50.6 months. Hypertension, obesity, dyslipidemia, and type 2 diabetes were highly prevalent, 59.8%, 46.6%, 34.2%, and 19.3% of patients, respectively, and more likely to develop and worsen in MACE compared to NFAT. Cardiovascular events were twice as common in MACE compared to NFAT, and cardiovascular mortality occurred more in MACE (6.6%) than NFAT (5.0%) during 58.7 months follow-up. Conclusions: Patients with NFAT and MACE do not require radiological or hormonal follow-up. However, they carry an increased risk of cardio-metabolic comorbidities, warranting evaluation and appropriate therapy.