Accuracy of the 10 μg desmopressin test for differential diagnosis of Cushing syndrome: a systematic review and meta-analysis

We evaluated the accuracy of the 10 μg desmopressin test in differentiating Cushing disease (CD) from non-neoplastic hypercortisolism (NNH) and ectopic ACTH syndrome (EAS). A systematic review of studies on diagnostic test accuracy in patients with CD, NNH, or EAS subjected to the desmopressin test...

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Main Authors: Rodrigo Rosa Giampietro, Marcos Vinicius Gama Cabral, Elizandra Gomes Pereira, Marcio Carlos Machado, Lucio Vilar, Vania dos Santos Nunes-Nogueira
Format: Article
Language:English
Published: Frontiers Media S.A. 2024-01-01
Series:Frontiers in Endocrinology
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fendo.2024.1332120/full
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author Rodrigo Rosa Giampietro
Marcos Vinicius Gama Cabral
Elizandra Gomes Pereira
Marcio Carlos Machado
Lucio Vilar
Vania dos Santos Nunes-Nogueira
author_facet Rodrigo Rosa Giampietro
Marcos Vinicius Gama Cabral
Elizandra Gomes Pereira
Marcio Carlos Machado
Lucio Vilar
Vania dos Santos Nunes-Nogueira
author_sort Rodrigo Rosa Giampietro
collection DOAJ
description We evaluated the accuracy of the 10 μg desmopressin test in differentiating Cushing disease (CD) from non-neoplastic hypercortisolism (NNH) and ectopic ACTH syndrome (EAS). A systematic review of studies on diagnostic test accuracy in patients with CD, NNH, or EAS subjected to the desmopressin test obtained from LILACS, PubMed, EMBASE, and CENTRAL databases was performed. Two reviewers independently selected the studies, assessed the risk of bias, and extracted the data. Hierarchical and bivariate models on Stata software were used for meta-analytical summaries. The certainty of evidence was measured using the GRADE (Grading of Recommendations Assessment, Development, and Evaluation Working Group) approach. In total, 14 studies were included: 3 studies on differentiated CD versus NNH and 11 studies on differentiated CD versus EAS. Considering ΔACTH in 8 studies involving 429 patients, the pooled sensitivity for distinguishing CD from EAS was 0.85 (95% confidence interval [CI]: 0.80–0.89, I2 = 17.6%) and specificity was 0.64 (95% CI: 0.49–0.76, I2 = 9.46%). Regarding Δcortisol in 6 studies involving 233 participants, the sensitivity for distinguishing CD from EAS was 0.81 (95% CI: 0.74–0.87, I2 = 7.98%) and specificity was 0.80 (95% CI: 0.61–0.91, I2 = 12.89%). The sensitivity and specificity of the combination of ΔACTH > 35% and Δcortisol > 20% in 5 studies involving 511 participants were 0.88 (95% CI: 0.79–0.93, I2 = 35%) and 0.74 (95% CI: 0.55–0.87, I2 = 27%), respectively. The pooled sensitivity for distinguishing CD from NNH in 3 studies involving 170 participants was 0.88 (95% CI: 0.79–0.93) and the specificity was 0.94 (95% CI: 0.86–0.97). Based on the desmopressin test for differentiating CD from EAS, considering ΔACTH, Δcortisol, or both percent increments, 15%, 19%, or 20% of patients with CD, respectively, would be incorrectly classified as having EAS. For CD versus NNH, 11% of patients with CD would be falsely diagnosed as having NNH, whereas 7% of patients with NNH would be falsely diagnosed as having CD. However, in all hierarchical plots, the prediction intervals were considerably wider than the confidence intervals. This indicates low confidence in the estimated accuracy, and the true accuracy is likely to be different.Systematic review registrationhttps://www.crd.york.ac.uk/prospero/display_record.php?RecordID=85634, identifier CRD42018085634; https://www.crd.york.ac.uk/prospero/display_record.php?RecordID=68317, identifier CRD42017068317.
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spelling doaj.art-25ced5bd9c4d44919590000c912c0b9c2024-01-30T04:24:22ZengFrontiers Media S.A.Frontiers in Endocrinology1664-23922024-01-011510.3389/fendo.2024.13321201332120Accuracy of the 10 μg desmopressin test for differential diagnosis of Cushing syndrome: a systematic review and meta-analysisRodrigo Rosa Giampietro0Marcos Vinicius Gama Cabral1Elizandra Gomes Pereira2Marcio Carlos Machado3Lucio Vilar4Vania dos Santos Nunes-Nogueira5Department of Internal Medicine, Sao Paulo State University/UNESP, Medical School, Sao Paulo, BrazilDepartment of Internal Medicine, Sao Paulo State University/UNESP, Medical School, Sao Paulo, BrazilDepartment of Internal Medicine, Sao Paulo State University/UNESP, Medical School, Sao Paulo, BrazilNeuroendocrine Unit, Division of Endocrinology and Metabolism, Hospital das Clínicas, University of Sao Paulo Medical School, Sao Paulo, BrazilDivision of Endocrinology, Hospital das Clínicas, Federal University of Pernambuco, Recife, BrazilDepartment of Internal Medicine, Sao Paulo State University/UNESP, Medical School, Sao Paulo, BrazilWe evaluated the accuracy of the 10 μg desmopressin test in differentiating Cushing disease (CD) from non-neoplastic hypercortisolism (NNH) and ectopic ACTH syndrome (EAS). A systematic review of studies on diagnostic test accuracy in patients with CD, NNH, or EAS subjected to the desmopressin test obtained from LILACS, PubMed, EMBASE, and CENTRAL databases was performed. Two reviewers independently selected the studies, assessed the risk of bias, and extracted the data. Hierarchical and bivariate models on Stata software were used for meta-analytical summaries. The certainty of evidence was measured using the GRADE (Grading of Recommendations Assessment, Development, and Evaluation Working Group) approach. In total, 14 studies were included: 3 studies on differentiated CD versus NNH and 11 studies on differentiated CD versus EAS. Considering ΔACTH in 8 studies involving 429 patients, the pooled sensitivity for distinguishing CD from EAS was 0.85 (95% confidence interval [CI]: 0.80–0.89, I2 = 17.6%) and specificity was 0.64 (95% CI: 0.49–0.76, I2 = 9.46%). Regarding Δcortisol in 6 studies involving 233 participants, the sensitivity for distinguishing CD from EAS was 0.81 (95% CI: 0.74–0.87, I2 = 7.98%) and specificity was 0.80 (95% CI: 0.61–0.91, I2 = 12.89%). The sensitivity and specificity of the combination of ΔACTH > 35% and Δcortisol > 20% in 5 studies involving 511 participants were 0.88 (95% CI: 0.79–0.93, I2 = 35%) and 0.74 (95% CI: 0.55–0.87, I2 = 27%), respectively. The pooled sensitivity for distinguishing CD from NNH in 3 studies involving 170 participants was 0.88 (95% CI: 0.79–0.93) and the specificity was 0.94 (95% CI: 0.86–0.97). Based on the desmopressin test for differentiating CD from EAS, considering ΔACTH, Δcortisol, or both percent increments, 15%, 19%, or 20% of patients with CD, respectively, would be incorrectly classified as having EAS. For CD versus NNH, 11% of patients with CD would be falsely diagnosed as having NNH, whereas 7% of patients with NNH would be falsely diagnosed as having CD. However, in all hierarchical plots, the prediction intervals were considerably wider than the confidence intervals. This indicates low confidence in the estimated accuracy, and the true accuracy is likely to be different.Systematic review registrationhttps://www.crd.york.ac.uk/prospero/display_record.php?RecordID=85634, identifier CRD42018085634; https://www.crd.york.ac.uk/prospero/display_record.php?RecordID=68317, identifier CRD42017068317.https://www.frontiersin.org/articles/10.3389/fendo.2024.1332120/fullCushing syndromeCushing diseasepseudo-Cushing syndromenon-neoplastic hypercortisolismdesmopressin testsystematic review
spellingShingle Rodrigo Rosa Giampietro
Marcos Vinicius Gama Cabral
Elizandra Gomes Pereira
Marcio Carlos Machado
Lucio Vilar
Vania dos Santos Nunes-Nogueira
Accuracy of the 10 μg desmopressin test for differential diagnosis of Cushing syndrome: a systematic review and meta-analysis
Frontiers in Endocrinology
Cushing syndrome
Cushing disease
pseudo-Cushing syndrome
non-neoplastic hypercortisolism
desmopressin test
systematic review
title Accuracy of the 10 μg desmopressin test for differential diagnosis of Cushing syndrome: a systematic review and meta-analysis
title_full Accuracy of the 10 μg desmopressin test for differential diagnosis of Cushing syndrome: a systematic review and meta-analysis
title_fullStr Accuracy of the 10 μg desmopressin test for differential diagnosis of Cushing syndrome: a systematic review and meta-analysis
title_full_unstemmed Accuracy of the 10 μg desmopressin test for differential diagnosis of Cushing syndrome: a systematic review and meta-analysis
title_short Accuracy of the 10 μg desmopressin test for differential diagnosis of Cushing syndrome: a systematic review and meta-analysis
title_sort accuracy of the 10 μg desmopressin test for differential diagnosis of cushing syndrome a systematic review and meta analysis
topic Cushing syndrome
Cushing disease
pseudo-Cushing syndrome
non-neoplastic hypercortisolism
desmopressin test
systematic review
url https://www.frontiersin.org/articles/10.3389/fendo.2024.1332120/full
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