Objective Cushing’s disease (CD) may recur despite corticotropic insufficiency (COI) following pituitary surgery. The predictive value of the desmopressin test (DT) for recurrence in this setting remains controversial. We have evaluated whether the disappearance of the response to DT predicts a low probability recurrence in a large cohort of patients with post-operative COI. Design Multicentre retrospective study. Methods Ninety-five patients with CD (women 82%, age 41 ± 14 years), responding preoperatively to DT and with early post-operative COI (08 00 am cortisol: <138 nmol/L), underwent a DT within 3 months post-surgery. Association between DT findings and the prediction of recurrence was tested using regression and ROC analyses. Results Recurrence occurred in 17/95 patients within 29 to 91 months. The cortisol peak (327, 95% CI (237–417) vs 121 (79–164) nmol/L, P = 0.0001) and absolute increment during DT (208 (136–280) vs 56 (22–90) nmol/L, P = 0.005) were greater in the recurrence vs remission group. Cortisol peak (AUC: 0.786 (0.670–0.902)) and increment (0.793 (0.672–0.914)) yielded a higher prognostic performance for recurrence than did the early post-operative 08 00 am cortisol (0.655 (0.505–0.804)). In the context of COI, cortisol peak >100 nmol/L and increment >30 nmol/L had a high negative predictive value (94, 95% CI (88–100) and 94, (88–100), respectively). Patients with a cortisol peak ≤100 nmol/L (vs >100) or an increment ≤30 nmol/L (vs >30) were less likely to have CD recurrence (odds ratios: 0.12, 95% CI (0.03–0.41) and 0.11 (0.02–0.36), respectively). Conclusion The disappearance of the response to the post-operative DT was independently associated with a lower odds of CD recurrence and offers an incremental prognostic value, which may help to stratify patients with COI and refine their follow-up according to the risk of recurrence.
OBJECTIVE The objectives of our study were to analyze the influence of age on the survival of patients with RAIR-DTC and to determine their prognostic factors according to age. METHODS This single centre, retrospective study enrolled 155 patients diagnosed with RAIR-DTC. The primary end point was overall survival (OS) according to different cut-off (45,55,65,75 years). Secondary endpoints were progression free survival (PFS) and prognostic factors in patients under and over 65 years. RESULTS Median OS after RAIR diagnosis was 8.2 years (95% IC: 5.3-9.6). There was no difference according to age with a 65 (p=0.47) and 55 years old cut-off (p=0.28). Median OS was significantly improved before 45 years old (p=0,0043). After 75 years old, median OS significantly decrease (p= 0,0008). Median PFS was 2.1 years (95% CI: 0.8-3) in patients < 65 years, and 1 year in patients ≥ 65 years (95% CI: 0.8-1.55) with no statistical difference (p=0.22). There was no impact of age on PFS with any cut-off. In both groups, progressive disease despite 131I treatment reduced OS. In patients < 65, an interval of less than 3 years between the initial diagnosis and the diagnosis of RAIR metastatic disease was predictive of poor survival. In patients > 65, the presence of a mediastinum metastasis was a significant factor for mortality (HR: 4.55, 95% CI: 2.27-9.09). CONCLUSION In RAIR-DTC patients, a cut-off age of 65 years old was not a significant predictive factor of survival. 45 and 75 years old cut off were predictive for OS, but not PFS.
Context When an SDHx mutation is identified in a patient with a pheochromocytoma (PCC) or a paraganglioma (PGL), predictive genetic testing can detect mutation carriers who would benefit from screening protocols. Objective To define tumor detection rate in a large cohort of asymptomatic SDHX mutation carriers. Design and Setting Retrospective multicentric study in 6 referral centers. Patients Between 2005 and 2019, 249 asymptomatic SDHx (171 SDHB, 31 SDHC, 47 SDHD) mutation carriers, with at least one imaging work-up were enrolled. Results Initial work-up including anatomical (98% of subjects [97-100 % according to center]) and/or functional imaging (67% [14-90%]) detected 48 tumors in 40 patients. After a negative initial work-up, 124 patients benefited from one to nine subsequent follow-up assessments (mean: 1.9/patient) with a median follow-up time of 5 [1-13] years. Anatomical (86% [49-100 %]) and/or functional imaging ((36% [7-60 %]) identified 10 new tumors (mean size: 16 mm [4-50]) in 10 patients. Altogether, 58 tumors (55 PGL, including 45 head and neck PGL, 2 PCC, 1 GIST), were detected in 50 patients (22 (13%) SDHB, 1 (3.2%) SDHC and 27 (57%) SDHD) with a median age of 41 years old [11-86], 76% without catecholamine secretion and 80% during initial imaging work-up. Conclusions Imaging screening enabled detection of tumors in 20% of asymptomatic SDHx mutation carriers with a higher detection rate in SDHD (57%) than in SDHB (13%) and SDHC (3%) mutations carriers, arguing for a gene-by-gene approach. Prospective studies using well-defined protocols are needed to obtain strong and useful data.
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