Recurrence appears to be more frequent and earlier after TSS in patients with USP8 mutant corticotroph tumours.
Context Signs and symptoms of Cushing’s syndrome (CS) overlap with common diseases, such as the metabolic syndrome, obesity, osteoporosis, and depression. Therefore, it can take years to finally diagnose CS, although early diagnosis is important for prevention of complications. Objective The aim of this study was to assess the time span between first symptoms and diagnosis of CS in different populations to identify factors associated with an early diagnosis. Data Sources A systematic literature search via PubMed was performed to identify studies reporting on time to diagnosis in CS. In addition, unpublished data from patients of our tertiary care center and 4 other centers were included. Study Selection Clinical studies reporting on the time to diagnosis of CS were eligible. Corresponding authors were contacted to obtain additional information relevant to the research question. Data Extraction Data were extracted from the text of the retrieved articles and from additional information provided by authors contacted successfully. From initially 3326 screened studies 44 were included. Data Synthesis Mean time to diagnosis for patients with CS was 34 months (ectopic CS: 14 months; adrenal CS: 30 months; and pituitary CS: 38 months; P < .001). No difference was found for gender, age (<18 and ≥18 years), and year of diagnosis (before and after 2000). Patients with pituitary CS had a longer time to diagnosis in Germany than elsewhere. Conclusions Time to diagnosis differs for subtypes of CS but not for gender and age. Time to diagnosis remains to be long and requires to be improved.
Background/Aims: The current diagnostic workup of Cushing’s syndrome (CS) requires various tests which only capture short-term cortisol exposure, whereas patients with endogenous CS generally have elevated cortisol levels over longer periods of time. Scalp hair assessment has emerged as a convenient test in capturing glucocorticoid concentrations over long periods of time. The aim of this multicenter, multinational, prospective, case-control study was to evaluate the diagnostic efficacy of scalp hair glucocorticoids in screening of endogenous CS. Methods: We assessed the diagnostic performances of hair cortisol (HairF), hair cortisone (HairE), and the sum of both (sumHairF+E), as measured by a state-of-the-art LC-MS/MS technique, in untreated patients with confirmed endogenous CS (n = 89) as well as in community controls (n = 295) from the population-based Lifelines cohort study. Results: Both glucocorticoids were significantly elevated in CS patients when compared to controls. A high diagnostic efficacy was found for HairF (area under the curve 0.87 [95% CI: 0.83–0.92]), HairE (0.93 [0.89–0.96]), and sumHairF+E (0.92 [0.88–0.96]) (all p < 0.001). The participants were accurately classified at the optimal cutoff threshold in 86% of the cases (81% sensitivity, 88% specificity, and 94% negative predictive value [NPV]) by HairF, in 90% of the cases (87% sensitivity, 90% specificity, and 96% NPV) by HairE, and in 87% of the cases (86% sensitivity, 88% specificity, and 95% NPV) by the sumHairF+E. HairE was shown to be the most accurate in differentiating CS patients from controls. Conclusion: Scalp hair glucocorticoids, especially hair cortisone, can be seen as a promising biomarker in screening for CS. Its convenience in collection and workup additionally makes it feasible for first-line screening.
PMT may confound the interpretation of immediate postoperative outcome. Follow-up is recommended to definitely evaluate surgical results.
Context Glucocorticoid-induced myopathy is a characteristic symptom of endogenous Cushing’s syndrome. Its long-term outcome is largely unknown Objective To evaluate long-term muscle function following remission of endogenous Cushing’s syndrome Study design Observational longitudinal cohort study Setting Tertiary care hospitals and specialized outpatient clinic Patients As part of the prospective multicenter German Cushing’s Registry we assessed muscle strength in patients with overt endogenous Cushing’s syndrome. We studied the patients at the time of diagnosis (n=88), after 6 months (n=69) and thereafter annually following surgical remission over a period of up to four years (1 year: n=55; 2 years: n=34; 3 years: n=29; 4 years: n=22). Muscle function was evaluated by hand grip strength and by chair rising test Results Grip strength was decreased to 83 % of normal controls (100 %) at time of diagnosis. It further decreased to 71 % after 6 months in remission (p≤0.001) and showed no improvement during further follow-up compared to baseline. Chair rising test performance improved initially (8 seconds at baseline vs 7 seconds after 6 months, p=0.004) but remained at this reduced level thereafter (7 seconds after 3 years vs 5 seconds in controls, p=0.038). In multivariate analysis we identified as predictors for long-term muscle dysfunction age, waist-to-hip-ratio and HbA1c at baseline. Furthermore, muscle strength during follow-up was strongly correlated with quality of life Conclusion This study shows that Cushing’s syndrome associated myopathy does not spontaneously resolve during remission. This calls for action to identify effective interventions to improve muscle dysfunction in this setting
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