Skip to main content
Top
Published in: Endocrine 3/2019

Open Access 01-03-2019 | Original Article

The effects of sampling lateralization on bilateral inferior petrosal sinus sampling and desmopressin stimulation test for pediatric Cushing's disease

Authors: Shi Chen, Kang Chen, Lin Lu, Xiaobo Zhang, Anli Tong, Hui Pan, Huijuan Zhu, Zhaolin Lu

Published in: Endocrine | Issue 3/2019

Login to get access

Abstract

Purpose

Bilateral inferior petrosal sinus sampling (BIPSS) is useful for differential diagnosis of adult Cushing’s disease (CD) but may not be so reliable in pediatric cases. The purpose of this study was to evaluate the sensitivity of BIPSS before and after desmopressin stimulation in pediatric CD, and to explore related factors of false-negative results and meanings of sampling lateralization.

Methods

We retrospectively analyzed 16 pediatric CD patients who underwent 17 BIPSS procedures from 2006 to 2017. CD was diagnosed if inferior petrosal sinus (IPS) to peripheral adrenocorticotropic hormone (ACTH) ratio was >2 at baseline or >3 after desmopressin stimulation. Sampling lateralization was yielded if interpetrosal sinus gradient was >1.4. Magnetic resonance imaging (MRI) was conducted. All the patients underwent surgery and the diagnosis was confirmed.

Results

The sensitivity was 64.7% (11/17) at baseline and 83.3% (10/12) after desmopressin stimulation. After stimulation, BIPSS reached its best sensitivity at 3 min. Sampling lateralization rate was 62.5% and 63.6% before and after stimulation, and the accordant rate with actual tumor lateralization was 50.0% and 42.9%, respectively. The accuracy of MRI in predicting the tumor lateralization was 80.0%. Sampling lateralization rate (81.8% in true-positive, 20.0% in false-negative, p = 0.036) and ACTH at dominant IPS (p = 0.001) was lower among false-negative patients.

Conclusions

The sensitivity of BIPSS in pediatric CD was low at baseline, but increased after desmopressin stimulation. Sampling lateralization cannot accurately indicate the tumor lateralization, but the absence of sampling lateralization with low ACTH at IPS is a hint of false-negative cases in BIPSS.
Literature
3.
go back to reference F. Pecori Giraldi, L.M. Cavallo, F. Tortora, R. Pivonello, A. Colao, P. Cappabianca, F. Mantero, Altogether to Beat Cushing's Syndrome Group The role of inferior petrosal sinus sampling in ACTH-dependent Cushing's syndrome: review and joint opinion statement by members of the Italian Society for Endocrinology, Italian Society for Neurosurgery, and Italian Society for Neuroradiology. Neurosurg. Focus 38(2), E5 (2015). https://doi.org/10.3171/2014.11.FOCUS14766 CrossRefPubMed F. Pecori Giraldi, L.M. Cavallo, F. Tortora, R. Pivonello, A. Colao, P. Cappabianca, F. Mantero, Altogether to Beat Cushing's Syndrome Group The role of inferior petrosal sinus sampling in ACTH-dependent Cushing's syndrome: review and joint opinion statement by members of the Italian Society for Endocrinology, Italian Society for Neurosurgery, and Italian Society for Neuroradiology. Neurosurg. Focus 38(2), E5 (2015). https://​doi.​org/​10.​3171/​2014.​11.​FOCUS14766 CrossRefPubMed
8.
go back to reference H.L. Storr, K.I. Alexandraki, L. Martin, A.M. Isidori, G.A. Kaltsas, J.P. Monson, G.M. Besser, M. Matson, J. Evanson, F. Afshar, I. Sabin, M.O. Savage, A.B. Grossman, Comparisons in the epidemiology, diagnostic features and cure rate by transsphenoidal surgery between paediatric and adult-onset Cushing's disease. Eur. J. Endocrinol. 164(5), 667–674 (2011). https://doi.org/10.1530/EJE-10-1120 CrossRefPubMed H.L. Storr, K.I. Alexandraki, L. Martin, A.M. Isidori, G.A. Kaltsas, J.P. Monson, G.M. Besser, M. Matson, J. Evanson, F. Afshar, I. Sabin, M.O. Savage, A.B. Grossman, Comparisons in the epidemiology, diagnostic features and cure rate by transsphenoidal surgery between paediatric and adult-onset Cushing's disease. Eur. J. Endocrinol. 164(5), 667–674 (2011). https://​doi.​org/​10.​1530/​EJE-10-1120 CrossRefPubMed
9.
go back to reference F. Castinetti, I. Morange, H. Dufour, P. Jaquet, B. Conte-Devolx, N. Girard, T. Brue, Desmopressin test during petrosal sinus sampling: a valuable tool to discriminate pituitary or ectopic ACTH-dependent Cushing's syndrome. Eur. J. Endocrinol. 157(3), 271–277 (2007). https://doi.org/10.1530/EJE-07-0215 CrossRefPubMed F. Castinetti, I. Morange, H. Dufour, P. Jaquet, B. Conte-Devolx, N. Girard, T. Brue, Desmopressin test during petrosal sinus sampling: a valuable tool to discriminate pituitary or ectopic ACTH-dependent Cushing's syndrome. Eur. J. Endocrinol. 157(3), 271–277 (2007). https://​doi.​org/​10.​1530/​EJE-07-0215 CrossRefPubMed
10.
11.
go back to reference R.A. Deipolyi, B. Alexander, J. Rho, A.J. Hirsch, R. Oklu, Bilateral inferior petrosal sinus sampling using desmopressin or corticotropic-releasing hormone: a single-center experience. J. Neurointerv. Surg. 7(9), 690 (2015)CrossRefPubMed R.A. Deipolyi, B. Alexander, J. Rho, A.J. Hirsch, R. Oklu, Bilateral inferior petrosal sinus sampling using desmopressin or corticotropic-releasing hormone: a single-center experience. J. Neurointerv. Surg. 7(9), 690 (2015)CrossRefPubMed
13.
go back to reference M. Feng, Z. Liu, X. Liu, X. Zhang, X. Bao, Y. Yao, K. Deng, B. Xing, W. Lian, H. Zhu, L. Lu, R. Wang, Tumour lateralization in Cushing's disease by inferior petrosal sinus sampling with desmopressin. Clin. Endocrinol. 88(2), 251–257 (2018). https://doi.org/10.1111/cen.13505 CrossRef M. Feng, Z. Liu, X. Liu, X. Zhang, X. Bao, Y. Yao, K. Deng, B. Xing, W. Lian, H. Zhu, L. Lu, R. Wang, Tumour lateralization in Cushing's disease by inferior petrosal sinus sampling with desmopressin. Clin. Endocrinol. 88(2), 251–257 (2018). https://​doi.​org/​10.​1111/​cen.​13505 CrossRef
19.
go back to reference A. Lienhardt, A.B. Grossman, J.E. Dacie, J. Evanson, A. Huebner, F. Afshar, P.N. Plowman, G.M. Besser, M.O. Savage, Relative contributions of inferior petrosal sinus sampling and pituitary imaging in the investigation of children and adolescents with ACTH-dependent Cushing's syndrome. J. Clin. Endocrinol. Metab. 86(12), 5711–5714 (2001). https://doi.org/10.1210/jcem.86.12.8086 CrossRefPubMed A. Lienhardt, A.B. Grossman, J.E. Dacie, J. Evanson, A. Huebner, F. Afshar, P.N. Plowman, G.M. Besser, M.O. Savage, Relative contributions of inferior petrosal sinus sampling and pituitary imaging in the investigation of children and adolescents with ACTH-dependent Cushing's syndrome. J. Clin. Endocrinol. Metab. 86(12), 5711–5714 (2001). https://​doi.​org/​10.​1210/​jcem.​86.​12.​8086 CrossRefPubMed
20.
go back to reference A.R. Deipolyi, J.A. Hirsch, R. Oklu, Caution during use of desmopressin in IPSS. J. Neurointerv. Surg. 5(5), e28 (2013)CrossRefPubMed A.R. Deipolyi, J.A. Hirsch, R. Oklu, Caution during use of desmopressin in IPSS. J. Neurointerv. Surg. 5(5), e28 (2013)CrossRefPubMed
Metadata
Title
The effects of sampling lateralization on bilateral inferior petrosal sinus sampling and desmopressin stimulation test for pediatric Cushing's disease
Authors
Shi Chen
Kang Chen
Lin Lu
Xiaobo Zhang
Anli Tong
Hui Pan
Huijuan Zhu
Zhaolin Lu
Publication date
01-03-2019
Publisher
Springer US
Published in
Endocrine / Issue 3/2019
Print ISSN: 1355-008X
Electronic ISSN: 1559-0100
DOI
https://doi.org/10.1007/s12020-018-1779-x

Other articles of this Issue 3/2019

Endocrine 3/2019 Go to the issue
Live Webinar | 27-06-2024 | 18:00 (CEST)

Keynote webinar | Spotlight on medication adherence

Live: Thursday 27th June 2024, 18:00-19:30 (CEST)

WHO estimates that half of all patients worldwide are non-adherent to their prescribed medication. The consequences of poor adherence can be catastrophic, on both the individual and population level.

Join our expert panel to discover why you need to understand the drivers of non-adherence in your patients, and how you can optimize medication adherence in your clinics to drastically improve patient outcomes.

Prof. Kevin Dolgin
Prof. Florian Limbourg
Prof. Anoop Chauhan
Developed by: Springer Medicine
Obesity Clinical Trial Summary

At a glance: The STEP trials

A round-up of the STEP phase 3 clinical trials evaluating semaglutide for weight loss in people with overweight or obesity.

Developed by: Springer Medicine