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Prolonged exposition to supraphysiological doses of exogenous glucocorticoid eventually results in iatrogenic Cushing's syndrome, whose intensity depends on the dose and duration of the treatment and on individual susceptibility. In patients with chronic inflammatory diseases treated with oral glucocorticoids iatrogenic Cushing's is expected and recognized and it only imposes that the dose of glucocorticoid be maintained as low as possible and that there is no better alternative therapy available.In some cases, however, iatrogenic Cushing's syndrome may be unexpected by the prescribing physician as the true exposure to corticoids may depend largely on the patient: this is the case for topical steroids used in inflammatory skin diseases such as psoriasis. Factitious Cushing's syndrome (FCS) is another cause of exogenous Cushing's syndrome in whom the exposure to glucocorticoid is unexpected, as it is hidden to the physician by a patient suffering from Münchausen syndrome. FCS might be very difficult to diagnose depending on the type of glucocorticoid used, the specificity of the dosage used for cortisol, and the timing of the measurement of cortisol and ACTH. The best evidence for FCS is the demonstration by LC-MS/MS of exogenous glucocorticoid in his urine or plasma but this requires that the patient has not stopped to take glucocorticoid at the time of exploration. FCS related to hydrocortisone can be difficult to prove and to distinguish from cyclical Cushing's syndrome. Analysis of the literature shows that FCS has led to prolonged or invasive explorations and even to adrenal surgery, while unrecognized FCS has led to fatal infectious complications., (Copyright © 2023 Elsevier Masson SAS. All rights reserved.)

Objective: Pneumocystis pneumonia (PcP) is an opportunistic infection occurring in immunocompromised patients. Cushing's syndrome (CS) impairs the immune system, and several authors have reported PcP in patients with CS. The present study aimed to characterize PcP occurring in a CS context and its management in French tertiary centers, in order to highlight the similarities in clinical presentation and treatment according to whether prophylaxis is implemented or not., Methods: This was a multicenter retrospective study conducted in several French University Hospitals and Cancer Centers. Patients with PcP and confirmed CS regardless of etiology were included. We excluded patients with other known causes of acquired immunodeficiency with increased risk of PcP., Results: Twenty-five patients were included. CS etiology was neoplastic in 84.0% of cases. CS clinical presentation associated predominant catabolic signs (76.0%), hypokalemia (91.7%) and lymphopenia (89.5%). CS was intense in most patients, with mean plasma cortisol levels at diagnosis of 2.424±1.102nmol/L and urinary free cortisol>10× the upper limit of normal in 85.0%. In all patients, PcP onset followed introduction of cortisol blockers, at a median 5.5 days. Patients were treated with 1 to 3 cortisol blockers, mainly metyrapone (88%), which significatively lowered plasma cortisol levels to 667±541nmol/L at the onset of PcP (P<0.001). PcP occurred in 7 patients despite prophylaxis. Finally, 60.0% patients were admitted to intensive care, and 20.0% died of PcP., Conclusion: High mortality in patients with PcP implies that clinicians should be better informed about this rare infectious complication. Prophylaxis remains controversial, requiring comparative studies., (Copyright © 2022. Published by Elsevier Masson SAS.)

Objective: The authors analyzed the current-intensity thresholds for electrostimulation of language fasciculi and the possible consequences of threshold variability on brain mapping., Methods: A prospective protocol of subcortical electrostimulation was used in 50 patients undergoing brain mapping, directly stimulating presumed language fasciculi identified by diffusion tensor imaging., Results: The stimulation-intensity thresholds for identification of language fasciculi varied among patients (mean minimum current intensity of 4.4 mA, range = 1.5-10 mA, standard deviation = 1.1 mA), and 23% of fascicular interferences were detected only above 5 mA. Repeated stimulation of the same site with the same intensity led to different types of interferences in 20% of patients, and a higher current intensity led to changes in the type of response in 27%. The mean minimum stimulation intensities did not differ significantly between different fasciculi, between the different types of interference obtained, or with age, sex, or type of tumor. Positive results on cortical mapping were significantly associated with positive results on subcortical mapping (P < 0.001). Subcortical intensity thresholds were slightly lower than cortical ones (mean = 4.43 vs. 5.25 mA, P = 0.034). In 23 of 50 subcortical mappings, fascicular stimulation produced no language interference., Conclusions: Individual variability of minimum stimulation-intensity thresholds for identification of language fasciculi is frequent. Nevertheless, even when a high current intensity was used, many stimulations on language fasciculi remained negative for various hypothetic reasons. Finding the optimal current intensity for identifying language fasciculi is of paramount importance to refine the clinical results and scientific data derived from brain mapping., (Copyright © 2022 Elsevier Inc. All rights reserved.)

Several cases of Pneumocystosis pneumonia (PCP) have been reported in patients with hypercortisolism, mainly in patients with severe ectopic ACTH syndrome (EAS). We report 2 cases of PCP that did not develop until after starting treatment with metyrapone, one of which occurred in an outpatient with Cushing's disease (CD) without pulmonary symptoms before medical treatment for CD. Patient 1 presented as an outpatient with CD and severe hypercortisolism but nonetheless in good general condition. Treatment with metyrapone was started before pituitary surgery. Patient 2 had EAS due to prostate cancer. Respiratory failure in the two patients occurred 4 days and 30 days, respectively, after the start of metyrapone treatment. In both cases, chest CT showed bilateral interstitial infiltrates, and Pneumocystis jirovecii was found on bronchoalveolar lavage (BAL). A literature review was performed to identify risk factors for PCP in patients with CD: we identified 20 other cases of PCP in patients treated for hypercortisolism, including 16 patients with EAS. Ninety percent of patients had free urinary cortisol greater than 6 times the upper limit of normal (ULN). In conclusion, onset of PCP after initiation of anticortisolic therapy is not limited to patients with EAS, and may occur in CD patients with elevated cortisol levels, even if the patient remains in good general condition and has no pulmonary symptoms before treatment. In such patients, routine prophylactic treatment with trimethoprim/sulfamethoxazole (TMP/SMX) should be considered., (Copyright © 2020 Elsevier Masson SAS. All rights reserved.)