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Cited by 52Pituitary Gland Disorders and TreatmentsAdrenal and Paraganglionic TumorsAdrenal Hormones and Disorders

A probabilistic model for Cushing’s syndrome screening in at-risk populations: a prospective multicenter study

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León-Justel, Antonio, Madrazo-Atutxa, Ainara, Alvarez-Rios, Ana I., Infantes-Fontán, Rocio, Garcia-Arnés, Juan A., Lillo-Muñoz, Juan A., Aulinas, Anna, Urgell-Rull, Eulàlia +9 more·2016·The Journal of Clinical Endocrinology & Metabolism
DOI 10.1210/jc.2016-1673
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Abstract

Context: Cushing’s syndrome (CS) is challenging to diagnose. Increased prevalence of CS in specific patient populations has been reported, but routine screening for CS remains questionable. To decrease the diagnostic delay and improve disease outcomes, simple new screening methods for CS in at-risk populations are needed. Objective: To develop and validate a simple scoring system to predict CS based on clinical signs and an easy-to-use biochemical test. Design: Observational, prospective, multicenter. Setting: Referral hospital. Patients: A cohort of 353 patients attending endocrinology units for outpatient visits. Interventions: All patients were evaluated with late-night salivary cortisol (LNSC) and a low-dose dexamethasone suppression test for CS. Main Outcome Measures: Diagnosis or exclusion of CS. Results: Twenty-six cases of CS were diagnosed in the cohort. A risk scoring system was developed by logistic regression analysis, and cutoff values were derived from a receiver operating characteristic curve. This risk score included clinical signs and symptoms (muscular atrophy, osteoporosis, and dorsocervical fat pad) and LNSC levels. The estimated area under the receiver operating characteristic curve was 0.93, with a sensitivity of 96.2% and specificity of 82.9%. Conclusions: We developed a risk score to predict CS in an at-risk population. This score may help to identify at-risk patients in non-endocrinological settings such as primary care, but external validation is warranted.

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  • A recent multicenter screening study for neoplastic hypercortisolism in a high-risk population employed LNSC (by a platform cortisol immunoassay) and the overnight 1 mg dexamethasone suppression test for initial diagnostic screening due to the high sensitivity of these tests and their excellent negative predictive value.

    “A recent multicenter screening study for neoplastic hypercortisolism in a high-risk population employed LNSC (by a platform cortisol immunoassay) and the overnight 1 mg DST for initial diagnostic screening due to the high sensitivity of these tests and their excellent negative predictive value [89]”

    From Prospective Evaluation of Late-Night Salivary Cortisol and Cortisone by EIA and LC-MS/MS in Suspected Cushing Syndrome (2020) · cites this paper as [89]

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  • Of the 26 patients with neoplastic hypercortisolism in the recent multicenter study, 20 (77%) had ACTH-dependent Cushing syndrome (17 with Cushing disease and 3 with ectopic ACTH) and 6 (23%) had ACTH-independent (adrenal) Cushing syndrome.

    “Of these 26 patients, 20 (77%) had ACTH-dependent CS (17 with CD and 3 with ectopic ACTH), and 6 (23%) had ACTH-independent (adrenal) CS.”

    From Prospective Evaluation of Late-Night Salivary Cortisol and Cortisone by EIA and LC-MS/MS in Suspected Cushing Syndrome (2020) · cites this paper as [89]

  • In a recent multicenter screening study, 26 (7.4%) of 353 patients were found to have neoplastic hypercortisolism.

    “This study found that 26 (7.4%) of their screened population of 353 patients were found to have neoplastic hypercortisolism.”

    From Prospective Evaluation of Late-Night Salivary Cortisol and Cortisone by EIA and LC-MS/MS in Suspected Cushing Syndrome (2020) · cites this paper as [89]

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  1. [1]1. Lacroix A, Feelders RA, Stratakis CA, Nieman LK. Cushing's syn- drome. Lancet. 2015;386:913-927.

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Authors
León-Justel, A., Madrazo-Atutxa, A., Alvarez-Rios, A. I., Infantes-Fontán, R., Garcia-Arnés, J. A., Lillo-Muñoz, J. A., Aulinas, A., Urgell-Rull, E., Boronat, M., Sánchez-de-Abajo, A., Fajardo-Montañana, C., Ortuño-Alonso, M., Salinas-Vert, I., Granada, M. L., Cano, D. A., Leal-Cerro, A., & for the Spanish CRISALIDA Study Group
Affiliations
  • Medicine Department (A.L.-J.), Huelva University Hospital, Instituto de Biomedicina de Sevilla (IBiS), Hospital Universitario Virgen del Rocío/Consejo Superior de Investigaciones Científicas/Universidad de Sevilla, 41013 Sevilla, Spain;
  • Unidad de Gestión Clínica de Endocrinología y Nutrición (A.M.-A., D.A.C., A.L.-C.), IBiS, Hospital Universitario Virgen del Rocío/Consejo Superior de Investigaciones Científicas/Universidad de Sevilla, 41013 Sevilla, Spain;
  • Department of Clinical Biochemistry (A.I.A.-R.), Virgen del Rocío University Hospital (IBiS/CSIC/SAS/University of Seville), 41013 Sevilla, Spain;
  • Servicio de Bioquímica (R.I.-F.), Sección Hormonas especiales, Hospital Universitario Virgen del Rocio, 41013 Sevilla, Spain;
  • Department of Clinical Endocrinology and Nutrition (J.A.G.-A.), Carlos Haya Hospital, 29010 Málaga, Spain;
  • Hospital Regional Universitario de Málaga (J.A.L.-M.), 29010 Málaga, Spain;
  • Universitat Autònoma de Barcelona (ES)
  • Clinical Biochemistry Department (E.U.-R.), Hospital de Sant Pau, 08025 Barcelona, Spain;
  • Universidad de Las Palmas de Gran Canaria (ES)
  • Servicio de Bioquímica Clínica (A.S.-d.-A.), Hospital Universitario Insular de Gran Canaria, 35016 Las Palmas de Gran Canaria, Spain;
  • Hospital Universitario de La Ribera (C.F.-M., M.O.-A.), 46600 Alzira, Valencia, Spain;
  • Hospital de La Ribera (ES)
  • Servicio Endocrinología y Nutrición (I.S.-V.), Hospital Universitari Germans Trias i Pujol, 08916 Badalona, Barcelona, Spain
  • Servicio de Bioquímica (M.L.G.), Hospital Universitari Germans Trias i Pujol, 08916 Badalona, Barcelona, Spain
  • Consejo Superior de Investigaciones Científicas (ES)
ORCID
Venue
The Journal of Clinical Endocrinology & Metabolism
Publisher
The Endocrine Society
Published
2016-08-04
Volume
101
Issue
10
Pages
3747-3754
ISSN
  • 0021-972X (ISSN-L)
  • 1945-7197
DOI
10.1210/jc.2016-1673
OpenAlex
W2505969566
Cited by
52
References
29
Citations by year
2026: 2 · 2025: 5 · 2024: 4 · 2023: 9 · 2022: 13 · 2021: 6 · 2020: 4 · 2019: 2 · 2018: 3 · 2017: 3 · 2016: 1
FWCI
3.01
Type
journal article
Language
en
Open access
Locations
  • The Journal of Clinical Endocrinology & Metabolism (publishedVersion)
  • PubMed (publishedVersion)
  • RISalud Institutional Health Repository of Andalucía (submittedVersion, other-oa)
  • Acceda (Universidad de Las Palmas de Gran Canaria) (submittedVersion)
Funders
  • Universidad de Sevilla
  • Consejo Superior de Investigaciones Científicas
Topics
  • Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Pituitary Gland Disorders and Treatments
  • Health Sciences › Medicine › Surgery › Adrenal and Paraganglionic Tumors
  • Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Adrenal Hormones and Disorders
Keywords
Cushing's syndrome, screening, risk scoring, late-night salivary cortisol, low-dose dexamethasone suppression test, delayed diagnosis, vulnerable populations, clinical signs and symptoms, primary care
MeSH terms
Adult, Aged, Cushing Syndrome, Dexamethasone, Female, Glucocorticoids, Humans, Hydrocortisone, Male, Middle Aged, Prospective Studies, Saliva, Sensitivity and Specificity, Risk Assessment

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