ABSTRACT
An 18-year-old man presenting with speech and behaviour disorders was found to suffer from Cushing's syndrome. Ectopic ACTH secretion was initially suspected on the basis of very high cortisoluria and ACTH concentrations, severe hypokalemia, a lung hypermetabolic lesion and a normal pituitary on conventional magnetic resonance imaging (MRI). After lung surgery, this lesion proved to be chronic necrotising aspergillosis (CNA). Diagnostic reevaluation by bilateral inferior petrosal sinus sampling disclosed a right-sided petrosal sinus gradient consistent with Cushing's disease (CD). A new high resolution thin-section MRI demonstrated a 5 mm pituitary adenoma, which could be removed successfully. Thus, a lung mass associated with several clues in favour of ectopic ACTH secretion may hide a diagnosis of CD. Such a mass can be CNA, an opportunistic infection favoured by immunosuppression, which to our knowledge, is the first case reported in CD.
Subject(s)
Pituitary ACTH Hypersecretion/complications , Pituitary ACTH Hypersecretion/diagnosis , Pulmonary Aspergillosis/complications , Pulmonary Aspergillosis/diagnosis , ACTH Syndrome, Ectopic/diagnosis , Adolescent , Chronic Disease , Diagnosis, Differential , Humans , Magnetic Resonance Imaging , Male , Necrosis , Petrosal Sinus Sampling , Pituitary ACTH Hypersecretion/surgery , Pulmonary Aspergillosis/surgeryABSTRACT
BACKGROUND: Although the reliability of cyclosporine (CyA) concentration at 2 (C2) hours postdosing has been established for kidney, liver, and heart transplant recipients, its use in lung cases remains to be validated. We investigated the relationship between CyA dual time point monitoring and long-term functional outcomes after lung transplantation. METHODS: We included data from 38 lung transplant recipients receiving CyA, azathioprins, and steroids in the study. CyA dosages were based on the trough concentrations. CyA concentrations at 0 (C0) and 2 (C2) hours postdosing were obtained at 1, 2, 3, 6, 9, 12, 15, 18, and 24 months postoperative. We retrospectively compared average CyA level (C0 and C2) during the first 3 posttransplantation months with forced expiratory volume in 1 second (FEV1), forced vital capacity (FVC), forced expiratory flow 25%-75 % (FEF 25-75), creatinine, systolic blood pressure (SBP), and diastolic blood pressure (DBP) using regression analysis via generalized estimating equations (GEE). RESULTS: Only improvement in FVC (P = .033) and deterioration of SBP (P < .001) were related to C0 monitoring. No correlation was observed between C0 and FEV1 (P = .13), FEF 25-75 (P = .48), creatinine (P = .07), and DBP (P = .97). Nor was any relationship observed between C2 concentrations and FEV1 (P = .64), FVC (P = .38), FEF 25-75 (P = .09), creatinine (P = .95), SBP (P = .73), or DBP (P = .51). CONCLUSION: There was a lack of a relationship between CyA concentrations (C0 and C2) and functional outcomes among de novo lung transplantations except for a positive correlation of 0 value with long-term improved FVC and increased SBP. This study suggested that C2 determinations may not improving lung recipient management.
