A noninvasive scoring model for the differential diagnosis of ACTH-dependent Cushing’s syndrome: a retrospective analysis of 311 patients based on easy-to-use parameters

Objectives The differential diagnosis of ACTH-dependent Cushing’s disease (CS) is challenging. The gold standard approach bilateral inferior petrosal sinus sampling (BIPSS) is expensive and invasive, while other noninvasive tests, like the high-dose dexamethasone suppression test (HDDST), provide un...

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Veröffentlicht in:Endocrine 2022-10, Vol.78 (1), p.114-122
Hauptverfasser: Lyu, Xiaohong, Zhang, Dingyue, Pan, Hui, Zhu, Huijuan, Chen, Shi, Lu, Lin
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container_end_page 122
container_issue 1
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container_title Endocrine
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creator Lyu, Xiaohong
Zhang, Dingyue
Pan, Hui
Zhu, Huijuan
Chen, Shi
Lu, Lin
description Objectives The differential diagnosis of ACTH-dependent Cushing’s disease (CS) is challenging. The gold standard approach bilateral inferior petrosal sinus sampling (BIPSS) is expensive and invasive, while other noninvasive tests, like the high-dose dexamethasone suppression test (HDDST), provide unsatisfactory diagnostic accuracy. This study aimed to find a new noninvasive practical approach with higher diagnostic accuracy to differently diagnose ACTH-dependent CS, which can be used in centers where BIPSS cannot be applied. Methods 264 Cushing’s disease (CD) patients and 47 ectopic ACTH secretion syndrome (EAS) patients were analyzed in this single-center retrospective study (2011–2021). The multivariate logistic model was used to construct the scoring model. Results Female (adjusted OR 3.030, 95%CI 1.229–7.471), hypokalemia (0.209, 0.076–0.576), ACTH (0.988, 0.982–0.994), MRI pituitary lesion positive (8.671, 3.521–21.352), and HDDST positive (2.768, 1.139–6.726) have a strong association with the differential diagnosis of ACTH-dependent CS and were included in the final multivariable logistic regression model. A -14-to-14-point noninvasive scoring model was built on the model. The AUC of the noninvasive scoring model was 0.915 (95% CI 0.869–0.960), significantly higher than the AUC of HDDST (0.756, 95% CI 0.685–0.825, P  = 0.004). The optimal cutoff of the model was ≥0 to diagnose CD. The sensitivity of the noninvasive scoring model was 91.3% (95% CI 87.3%–94.1%), and the specificity was 80.9% (95% CI 67.5%–89.6%). When the model’s sensitivity was 100.0%, the cutoff was ≥ −10 with a specificity of 19.2%; when the model’s specificity was 100.0%, the cutoff was ≥ 13 with a sensitivity of 22.7%. Conclusions We developed a noninvasive scoring model to distinguish CD and EAS in ACTH-dependent CS patients with higher diagnostic utility than HDDST in the same cohort. The noninvasive scoring model might be applied in areas where BIPSS is unavailable, the CRH is hard to obtain, or the desmopressin stimulation is not widely applied. It also provided a triage tool for selecting patients that might benefit the most from a further BIPSS test.
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The gold standard approach bilateral inferior petrosal sinus sampling (BIPSS) is expensive and invasive, while other noninvasive tests, like the high-dose dexamethasone suppression test (HDDST), provide unsatisfactory diagnostic accuracy. This study aimed to find a new noninvasive practical approach with higher diagnostic accuracy to differently diagnose ACTH-dependent CS, which can be used in centers where BIPSS cannot be applied. Methods 264 Cushing’s disease (CD) patients and 47 ectopic ACTH secretion syndrome (EAS) patients were analyzed in this single-center retrospective study (2011–2021). The multivariate logistic model was used to construct the scoring model. Results Female (adjusted OR 3.030, 95%CI 1.229–7.471), hypokalemia (0.209, 0.076–0.576), ACTH (0.988, 0.982–0.994), MRI pituitary lesion positive (8.671, 3.521–21.352), and HDDST positive (2.768, 1.139–6.726) have a strong association with the differential diagnosis of ACTH-dependent CS and were included in the final multivariable logistic regression model. A -14-to-14-point noninvasive scoring model was built on the model. The AUC of the noninvasive scoring model was 0.915 (95% CI 0.869–0.960), significantly higher than the AUC of HDDST (0.756, 95% CI 0.685–0.825, P  = 0.004). The optimal cutoff of the model was ≥0 to diagnose CD. The sensitivity of the noninvasive scoring model was 91.3% (95% CI 87.3%–94.1%), and the specificity was 80.9% (95% CI 67.5%–89.6%). When the model’s sensitivity was 100.0%, the cutoff was ≥ −10 with a specificity of 19.2%; when the model’s specificity was 100.0%, the cutoff was ≥ 13 with a sensitivity of 22.7%. Conclusions We developed a noninvasive scoring model to distinguish CD and EAS in ACTH-dependent CS patients with higher diagnostic utility than HDDST in the same cohort. The noninvasive scoring model might be applied in areas where BIPSS is unavailable, the CRH is hard to obtain, or the desmopressin stimulation is not widely applied. 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The gold standard approach bilateral inferior petrosal sinus sampling (BIPSS) is expensive and invasive, while other noninvasive tests, like the high-dose dexamethasone suppression test (HDDST), provide unsatisfactory diagnostic accuracy. This study aimed to find a new noninvasive practical approach with higher diagnostic accuracy to differently diagnose ACTH-dependent CS, which can be used in centers where BIPSS cannot be applied. Methods 264 Cushing’s disease (CD) patients and 47 ectopic ACTH secretion syndrome (EAS) patients were analyzed in this single-center retrospective study (2011–2021). The multivariate logistic model was used to construct the scoring model. Results Female (adjusted OR 3.030, 95%CI 1.229–7.471), hypokalemia (0.209, 0.076–0.576), ACTH (0.988, 0.982–0.994), MRI pituitary lesion positive (8.671, 3.521–21.352), and HDDST positive (2.768, 1.139–6.726) have a strong association with the differential diagnosis of ACTH-dependent CS and were included in the final multivariable logistic regression model. A -14-to-14-point noninvasive scoring model was built on the model. The AUC of the noninvasive scoring model was 0.915 (95% CI 0.869–0.960), significantly higher than the AUC of HDDST (0.756, 95% CI 0.685–0.825, P  = 0.004). The optimal cutoff of the model was ≥0 to diagnose CD. The sensitivity of the noninvasive scoring model was 91.3% (95% CI 87.3%–94.1%), and the specificity was 80.9% (95% CI 67.5%–89.6%). When the model’s sensitivity was 100.0%, the cutoff was ≥ −10 with a specificity of 19.2%; when the model’s specificity was 100.0%, the cutoff was ≥ 13 with a sensitivity of 22.7%. Conclusions We developed a noninvasive scoring model to distinguish CD and EAS in ACTH-dependent CS patients with higher diagnostic utility than HDDST in the same cohort. The noninvasive scoring model might be applied in areas where BIPSS is unavailable, the CRH is hard to obtain, or the desmopressin stimulation is not widely applied. It also provided a triage tool for selecting patients that might benefit the most from a further BIPSS test.</description><subject>Adrenocorticotropic hormone</subject><subject>Desmopressin</subject><subject>Dexamethasone</subject><subject>Diabetes</subject><subject>Differential diagnosis</subject><subject>Endocrinology</subject><subject>Humanities and Social Sciences</subject><subject>Hypokalemia</subject><subject>Internal Medicine</subject><subject>Medicine</subject><subject>Medicine &amp; Public Health</subject><subject>multidisciplinary</subject><subject>Nervous system diseases</subject><subject>Original Article</subject><subject>Pituitary</subject><subject>Science</subject><issn>1559-0100</issn><issn>1355-008X</issn><issn>1559-0100</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><recordid>eNp9kU1uFDEQhS1EJMIkF2BliQ0bg-3-cTe70QgIUiQ2ybpV0y4njrrtxuWONDuuwRm4FSfBw0QCsWDjH73vvZL9GHul5FslpXlHSksthdRayEp2Sshn7Fw1TS9k0Z__dX7BXhI9yELq1pyzH1seYvDhEcg_IqcxJh_u-BwtTtzFxPM9cuudw4Qhe5jKBe5CJE88Or7d3VwJiwsGW2S-W-m-2H9--06cDsGmOON7DjxhTpEWHPNxCASYDk8BlVJ8geyLm_geCC2PgSPQQeQoVsKiJpgxY6ILduZgIrx82jfs9uOHm92VuP7y6fNuey3GqtFZQC17LcEgKGNaW0MzqtYZu-_QjnXf9VY7ZW0HbW0UYjNK6_aN6Y3Z97p3XbVhb065S4pfV6Q8zJ5GnCYIGFcadNt3baVrWRX09T_oQ1xTeV-hjKprU3W6LZQ-UWP5BUrohiX5GdJhUHI49jec-htKK8Pv_sq6YdXJRMuxEkx_ov_j-gVwJ6Er</recordid><startdate>20221001</startdate><enddate>20221001</enddate><creator>Lyu, Xiaohong</creator><creator>Zhang, Dingyue</creator><creator>Pan, Hui</creator><creator>Zhu, Huijuan</creator><creator>Chen, Shi</creator><creator>Lu, Lin</creator><general>Springer US</general><general>Springer Nature B.V</general><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope></search><sort><creationdate>20221001</creationdate><title>A noninvasive scoring model for the differential diagnosis of ACTH-dependent Cushing’s syndrome: a retrospective analysis of 311 patients based on easy-to-use parameters</title><author>Lyu, Xiaohong ; Zhang, Dingyue ; Pan, Hui ; Zhu, Huijuan ; Chen, Shi ; Lu, Lin</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c352t-a40920a7ea1776d4a5c16f7db8edc4989d2f1dd8a6471ee5c0dfb57977b929f83</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2022</creationdate><topic>Adrenocorticotropic hormone</topic><topic>Desmopressin</topic><topic>Dexamethasone</topic><topic>Diabetes</topic><topic>Differential diagnosis</topic><topic>Endocrinology</topic><topic>Humanities and Social Sciences</topic><topic>Hypokalemia</topic><topic>Internal Medicine</topic><topic>Medicine</topic><topic>Medicine &amp; Public Health</topic><topic>multidisciplinary</topic><topic>Nervous system diseases</topic><topic>Original Article</topic><topic>Pituitary</topic><topic>Science</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Lyu, Xiaohong</creatorcontrib><creatorcontrib>Zhang, Dingyue</creatorcontrib><creatorcontrib>Pan, Hui</creatorcontrib><creatorcontrib>Zhu, Huijuan</creatorcontrib><creatorcontrib>Chen, Shi</creatorcontrib><creatorcontrib>Lu, Lin</creatorcontrib><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Endocrine</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Lyu, Xiaohong</au><au>Zhang, Dingyue</au><au>Pan, Hui</au><au>Zhu, Huijuan</au><au>Chen, Shi</au><au>Lu, Lin</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>A noninvasive scoring model for the differential diagnosis of ACTH-dependent Cushing’s syndrome: a retrospective analysis of 311 patients based on easy-to-use parameters</atitle><jtitle>Endocrine</jtitle><stitle>Endocrine</stitle><date>2022-10-01</date><risdate>2022</risdate><volume>78</volume><issue>1</issue><spage>114</spage><epage>122</epage><pages>114-122</pages><issn>1559-0100</issn><issn>1355-008X</issn><eissn>1559-0100</eissn><abstract>Objectives The differential diagnosis of ACTH-dependent Cushing’s disease (CS) is challenging. The gold standard approach bilateral inferior petrosal sinus sampling (BIPSS) is expensive and invasive, while other noninvasive tests, like the high-dose dexamethasone suppression test (HDDST), provide unsatisfactory diagnostic accuracy. This study aimed to find a new noninvasive practical approach with higher diagnostic accuracy to differently diagnose ACTH-dependent CS, which can be used in centers where BIPSS cannot be applied. Methods 264 Cushing’s disease (CD) patients and 47 ectopic ACTH secretion syndrome (EAS) patients were analyzed in this single-center retrospective study (2011–2021). The multivariate logistic model was used to construct the scoring model. Results Female (adjusted OR 3.030, 95%CI 1.229–7.471), hypokalemia (0.209, 0.076–0.576), ACTH (0.988, 0.982–0.994), MRI pituitary lesion positive (8.671, 3.521–21.352), and HDDST positive (2.768, 1.139–6.726) have a strong association with the differential diagnosis of ACTH-dependent CS and were included in the final multivariable logistic regression model. A -14-to-14-point noninvasive scoring model was built on the model. The AUC of the noninvasive scoring model was 0.915 (95% CI 0.869–0.960), significantly higher than the AUC of HDDST (0.756, 95% CI 0.685–0.825, P  = 0.004). The optimal cutoff of the model was ≥0 to diagnose CD. The sensitivity of the noninvasive scoring model was 91.3% (95% CI 87.3%–94.1%), and the specificity was 80.9% (95% CI 67.5%–89.6%). When the model’s sensitivity was 100.0%, the cutoff was ≥ −10 with a specificity of 19.2%; when the model’s specificity was 100.0%, the cutoff was ≥ 13 with a sensitivity of 22.7%. Conclusions We developed a noninvasive scoring model to distinguish CD and EAS in ACTH-dependent CS patients with higher diagnostic utility than HDDST in the same cohort. The noninvasive scoring model might be applied in areas where BIPSS is unavailable, the CRH is hard to obtain, or the desmopressin stimulation is not widely applied. It also provided a triage tool for selecting patients that might benefit the most from a further BIPSS test.</abstract><cop>New York</cop><pub>Springer US</pub><doi>10.1007/s12020-022-03081-0</doi><tpages>9</tpages></addata></record>
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subjects Adrenocorticotropic hormone
Desmopressin
Dexamethasone
Diabetes
Differential diagnosis
Endocrinology
Humanities and Social Sciences
Hypokalemia
Internal Medicine
Medicine
Medicine & Public Health
multidisciplinary
Nervous system diseases
Original Article
Pituitary
Science
title A noninvasive scoring model for the differential diagnosis of ACTH-dependent Cushing’s syndrome: a retrospective analysis of 311 patients based on easy-to-use parameters
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