The role of adrenal venous sampling (AVS) in primary bilateral macronodular adrenocortical hyperplasia (PBMAH): a study of 16 patients

OBJECTIVE: Primary bilateral macronodular adrenocortical hyperplasia (PBMAH) is a rare cause of ACTH-independent Cushing’s syndrome. Current guidelines recommend bilateral adrenalectomy for PBMAH, but several studies showed clinical effectiveness of unilateral adrenalectomy despite bilateral disease...

Descripción completa

Detalles Bibliográficos
Autores principales: Rubinstein, German, Osswald, Andrea, Braun, Leah Theresa, Vogel, Frederick, Kroiss, Matthias, Pilz, Stefan, Deniz, Sinan, Aigner, Laura, Knösel, Thomas, Bertherat, Jérôme, Bouys, Lucas, Ladurner, Roland, Riester, Anna, Bidlingmaier, Martin, Beuschlein, Felix, Reincke, Martin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer US 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9068666/
https://www.ncbi.nlm.nih.gov/pubmed/35274284
http://dx.doi.org/10.1007/s12020-022-03020-z
_version_ 1784700266840326144
author Rubinstein, German
Osswald, Andrea
Braun, Leah Theresa
Vogel, Frederick
Kroiss, Matthias
Pilz, Stefan
Deniz, Sinan
Aigner, Laura
Knösel, Thomas
Bertherat, Jérôme
Bouys, Lucas
Ladurner, Roland
Riester, Anna
Bidlingmaier, Martin
Beuschlein, Felix
Reincke, Martin
author_facet Rubinstein, German
Osswald, Andrea
Braun, Leah Theresa
Vogel, Frederick
Kroiss, Matthias
Pilz, Stefan
Deniz, Sinan
Aigner, Laura
Knösel, Thomas
Bertherat, Jérôme
Bouys, Lucas
Ladurner, Roland
Riester, Anna
Bidlingmaier, Martin
Beuschlein, Felix
Reincke, Martin
author_sort Rubinstein, German
collection PubMed
description OBJECTIVE: Primary bilateral macronodular adrenocortical hyperplasia (PBMAH) is a rare cause of ACTH-independent Cushing’s syndrome. Current guidelines recommend bilateral adrenalectomy for PBMAH, but several studies showed clinical effectiveness of unilateral adrenalectomy despite bilateral disease in selected patients. Our aim was to evaluate the gain of information which can be obtained through adrenal venous sampling (AVS) based cortisol lateralization ratios for guidance of unilateral adrenalectomy. DESIGN: We performed a retrospective analysis of 16 patients with PBMAH and clinical overt cortisol secretion in three centers METHODS: Selectivity of adrenal vein sampling during AVS was defined as a gradient of cortisol or a reference adrenal hormone ≥2.0 between adrenal and peripheral vein. Lateralization was assumed if the dominant to non-dominant ratio of cortisol to reference hormone was ≥4.0. RESULTS: AVS was technically successful in all patients based on absolute cortisol levels and in 13 of 16 patients (81%) based on reference hormone levels. Lateralization was documented in 8 of 16 patients. In patients with lateralization, in 5 of 8 cases this occurred toward morphologically larger adrenals, while in 3 patients lateralization was present in bilaterally identical adrenals. The combined volume of adrenals correlated positively with urinary free cortisol, suggesting that adrenal size is the dominant determinant of cortisol secretion. CONCLUSIONS: In this study the gain of information through AVS for unilateral adrenalectomy was limited in patients with PBMAH and marked adrenal asymmetry.
format Online
Article
Text
id pubmed-9068666
institution National Center for Biotechnology Information
language English
publishDate 2022
publisher Springer US
record_format MEDLINE/PubMed
spelling pubmed-90686662022-05-07 The role of adrenal venous sampling (AVS) in primary bilateral macronodular adrenocortical hyperplasia (PBMAH): a study of 16 patients Rubinstein, German Osswald, Andrea Braun, Leah Theresa Vogel, Frederick Kroiss, Matthias Pilz, Stefan Deniz, Sinan Aigner, Laura Knösel, Thomas Bertherat, Jérôme Bouys, Lucas Ladurner, Roland Riester, Anna Bidlingmaier, Martin Beuschlein, Felix Reincke, Martin Endocrine Original Article OBJECTIVE: Primary bilateral macronodular adrenocortical hyperplasia (PBMAH) is a rare cause of ACTH-independent Cushing’s syndrome. Current guidelines recommend bilateral adrenalectomy for PBMAH, but several studies showed clinical effectiveness of unilateral adrenalectomy despite bilateral disease in selected patients. Our aim was to evaluate the gain of information which can be obtained through adrenal venous sampling (AVS) based cortisol lateralization ratios for guidance of unilateral adrenalectomy. DESIGN: We performed a retrospective analysis of 16 patients with PBMAH and clinical overt cortisol secretion in three centers METHODS: Selectivity of adrenal vein sampling during AVS was defined as a gradient of cortisol or a reference adrenal hormone ≥2.0 between adrenal and peripheral vein. Lateralization was assumed if the dominant to non-dominant ratio of cortisol to reference hormone was ≥4.0. RESULTS: AVS was technically successful in all patients based on absolute cortisol levels and in 13 of 16 patients (81%) based on reference hormone levels. Lateralization was documented in 8 of 16 patients. In patients with lateralization, in 5 of 8 cases this occurred toward morphologically larger adrenals, while in 3 patients lateralization was present in bilaterally identical adrenals. The combined volume of adrenals correlated positively with urinary free cortisol, suggesting that adrenal size is the dominant determinant of cortisol secretion. CONCLUSIONS: In this study the gain of information through AVS for unilateral adrenalectomy was limited in patients with PBMAH and marked adrenal asymmetry. Springer US 2022-03-10 2022 /pmc/articles/PMC9068666/ /pubmed/35274284 http://dx.doi.org/10.1007/s12020-022-03020-z Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Original Article
Rubinstein, German
Osswald, Andrea
Braun, Leah Theresa
Vogel, Frederick
Kroiss, Matthias
Pilz, Stefan
Deniz, Sinan
Aigner, Laura
Knösel, Thomas
Bertherat, Jérôme
Bouys, Lucas
Ladurner, Roland
Riester, Anna
Bidlingmaier, Martin
Beuschlein, Felix
Reincke, Martin
The role of adrenal venous sampling (AVS) in primary bilateral macronodular adrenocortical hyperplasia (PBMAH): a study of 16 patients
title The role of adrenal venous sampling (AVS) in primary bilateral macronodular adrenocortical hyperplasia (PBMAH): a study of 16 patients
title_full The role of adrenal venous sampling (AVS) in primary bilateral macronodular adrenocortical hyperplasia (PBMAH): a study of 16 patients
title_fullStr The role of adrenal venous sampling (AVS) in primary bilateral macronodular adrenocortical hyperplasia (PBMAH): a study of 16 patients
title_full_unstemmed The role of adrenal venous sampling (AVS) in primary bilateral macronodular adrenocortical hyperplasia (PBMAH): a study of 16 patients
title_short The role of adrenal venous sampling (AVS) in primary bilateral macronodular adrenocortical hyperplasia (PBMAH): a study of 16 patients
title_sort role of adrenal venous sampling (avs) in primary bilateral macronodular adrenocortical hyperplasia (pbmah): a study of 16 patients
topic Original Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9068666/
https://www.ncbi.nlm.nih.gov/pubmed/35274284
http://dx.doi.org/10.1007/s12020-022-03020-z
work_keys_str_mv AT rubinsteingerman theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT osswaldandrea theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT braunleahtheresa theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT vogelfrederick theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT kroissmatthias theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT pilzstefan theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT denizsinan theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT aignerlaura theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT knoselthomas theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT bertheratjerome theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT bouyslucas theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT ladurnerroland theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT riesteranna theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT bidlingmaiermartin theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT beuschleinfelix theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT reinckemartin theroleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT rubinsteingerman roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT osswaldandrea roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT braunleahtheresa roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT vogelfrederick roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT kroissmatthias roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT pilzstefan roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT denizsinan roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT aignerlaura roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT knoselthomas roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT bertheratjerome roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT bouyslucas roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT ladurnerroland roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT riesteranna roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT bidlingmaiermartin roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT beuschleinfelix roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients
AT reinckemartin roleofadrenalvenoussamplingavsinprimarybilateralmacronodularadrenocorticalhyperplasiapbmahastudyof16patients