Differential diagnostic value of bilateral inferior Petrosal sinus sampling (BIPSS) in ACTH-dependent Cushing syndrome:

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RESEARCH ARTICLE

Open Access

Differential diagnostic value of bilateral inferior Petrosal sinus sampling (BIPSS) in ACTH-dependent Cushing syndrome: a systematic review and Meta-analysis Hao Wang* , Ying Ba*, Qian Xing and Run-Ce Cai

Abstract Background: Previous studies have shown inconsistent results about the usefulness of bilateral inferior petrosal sinus sampling (BIPSS) in differential diagnosis of adrenocorticotropic hormone (ACTH)-dependent Cushing syndrome. This meta-analysis evaluated the diagnostic value of BIPSS via the published literature. Methods: This study searched PubMed, Embase, Web of Science, Cochrane library, and Wanfang database for published data on the use of BIPSS in Cushing syndrome differential diagnosis as of October 2019. Sensitivity, specificity, positive likelihood ratio (PLR), negative likelihood ratio (NLR), diagnostic odds ratio (DOR), and receiver operating characteristic (ROC) curves were calculated based on the relevant data. Results: This meta-analysis included a total of 23 studies with 1642 patients. The calculated sensitivity, specificity, PLR, and NLR were 0.94 (95% confidence interval, CI: 0.91–0.96), 0.89 (95% CI: 0.79–0.95), 8.8 (95% CI: 4.3–17.9), and 0.07 (95% CI: 0.04–0.11), respectively. The pooled DOR and area under the ROC curve were 129 (95% CI: 48–345) and 0.97 (95% CI: 0.95–0.98), respectively. Conclusion: This meta-analysis indicated that BIPSS had high diagnostic value for detecting ACTH in patients with ACTH-dependent Cushing syndrome, and BIPSS should be used as an effective method to identify ACTH-secretion sources. Keywords: Bilateral inferior petrosal sinus sampling, ACTH-dependent Cushing syndrome, Differential diagnosis, Diagnostic meta-analysis

Background Adrenocorticotropic hormone (ACTH)-dependent Cushing syndrome (CS) is caused by excessive secretion of ACTH by the pituitary or pituitary tumors, causing bilateral adrenal hyperplasia and excessive cortisol secretion with clinical manifestations such as a moon-shaped face, buffalo hump, and hypertension. The majority of ACTH-dependent Cushing syndrome cases are caused * Correspondence: [email protected]; [email protected] Department of Endocrinology, The First Affiliated Hospital of Dalian Medical University, Dalian, Liaoning Province, China

by Cushing disease (CD), a condition in which ACTHsecreting tumors are responsible for elevated ACTH levels. Other cases, such as ectopic ACTH syndrome (EAS), have ectopic sources. These have different therapeutic principles and prognoses. Based only on clinical manifestations, detection of cortisol levels and ACTH, high- and low-dose dexamethasone suppression tests, and imaging, these conditions are not completely distinguishable. Studies have shown that non-functional pituitary tumors are common [1–3], suggesting that even if a pituitary tumor is revealed by magnetic resonance

© The Author(s). 2020 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and r