Long-term outcome of primary bilateral macronodular adrenocortical hyperplasia after unilateral adrenalectomy
Open accessAbstract
CONTEXT: Unilateral adrenalectomy has been proposed in selected patients with primary bilateral macronodular adrenocortical hyperplasia (PBMAH), but its long-term outcome is unclear. OBJECTIVE: The aim of this study was to analyze long-term clinical and biochemical outcomes of unilateral adrenalectomy vs bilateral adrenalectomy in patients with PBMAH in comparison with the outcome of cortisol-producing adenoma (CPA) treated with unilateral adrenalectomy. DESIGN: Retrospective observational study in three German and one Italian academic tertiary care center. PATIENTS AND METHODS: Twenty-five patients with PBMAH after unilateral adrenalectomy (unilat-ADX-PBMAH), nine patients with PBMAH and bilateral adrenalectomy (bilat-ADX-PBMAH), and 39 patients with CPA and unilateral adrenalectomy (unilat-ADX-CPA) were included. RESULTS: Baseline clinical and biochemical parameters were comparable in patients with unilat-ADX-PBMAH, bilat-ADX-PBMAH, and unilat-ADX-CPA. Directly after surgery, 84% of the patients with unilat-ADX-PBMAH experienced initial remission of Cushing syndrome (CS). In contrast, at last follow-up (median, 50 months), 32% of the patients with unilat-ADX-PBMAH were biochemically controlled compared with nearly all patients in the other two groups (P = 0.000). Adrenalectomy of the contralateral side had to be performed in 12% of the initial patients with unilat-ADX-PBMAH. Three of 20 patients with unilat-ADX-PBMAH (15%) died during follow-up, presumably of CS-related causes; no deaths occurred in the other two groups (P = 0.008). Deaths occurred exclusively in patients who were not biochemically controlled after unilateral ADX. CONCLUSIONS: Our data suggest that unilateral adrenalectomy of patients with PBMAH leads to clinical remission and a lower incidence of adrenal crisis but in less sufficient biochemical control of hypercortisolism, potentially leading to higher mortality.
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In a retrospective observational study, unilateral adrenalectomy for PBMAH led to initial clinical remission in 84% of patients.
“The data, albeit retrospective and observational, showed that unilateral adrenalectomy of patients with PMAH lead to initial clinical remission in 84% of patients”
From Adrenal incidentaloma (2020) · cites this paper as [328]
3 deaths occurred in the group not biochemically controlled after unilateral adrenalectomy.
“Additionally, 3 deaths occurred in the group of patients who were not biochemically controlled after unilateral adrenalectomy.”
From Adrenal incidentaloma (2020) · cites this paper as [328]
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- Authors
- Osswald, A., Quinkler, M., Di Dalmazi, G., Deutschbein, T., Rubinstein, G., Ritzel, K., Zopp, S., Bertherat, J., Beuschlein, F., & Reincke, M.
- Affiliations
- LMU Klinikum (DE)
- Endocrinology in Charlottenburg, Berlin, Germany
- Endocrinology Unit and Center for Applied Biomedical Research, Department of Medical and Surgical Sciences, Alma Mater University of Bologna, S. Orsola-Malpighi Hospital, Bologna, Italy
- Universitätsklinikum Würzburg (DE)
- Hôpital Cochin (FR)
- Medizinische Klinik und Poliklinik IV, Klinikum der Universität, Ludwig-Maximilians-Universität München, München, Germany
- ORCID
- Venue
- The Journal of Clinical Endocrinology & Metabolism
- Publisher
- The Endocrine Society
- Published
- 2019-03-07
- Volume
- 104
- Issue
- 7
- Pages
- 2985-2993
- ISSN
- 0021-972X (ISSN-L)
- 1945-7197
- OpenAlex
- W2922396602
- Cited by
- 76
- References
- 26
- Citations by year
- 2026: 7 · 2025: 8 · 2024: 4 · 2023: 16 · 2022: 14 · 2021: 10 · 2020: 12 · 2019: 5
- FWCI
- 10.03
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- The Journal of Clinical Endocrinology & Metabolism (publishedVersion)
- PubMed (publishedVersion)
- Zurich Open Repository and Archive (University of Zurich) (acceptedVersion, other-oa)
- Archivio istituzionale della ricerca (Alma Mater Studiorum Università di Bologna) (submittedVersion)
- Zurich Open Repository and Archive (University of Zurich) (submittedVersion, other-oa)
- Zurich Open Repository and Archive (University of Zurich)
- Funders
- Else Kröner-Fresenius-Stiftung (2012_A103 and 2015_A228)
- Deutsche Forschungsgemeinschaft (CRC/TRR 205 “The Adrenal Gland”)
- Interdisziplinäres Zentrum für Klinische Forschung, Universitätsklinikum Würzburg (Z-2/57)
- Topics
- Health Sciences › Medicine › Surgery › Adrenal and Paraganglionic Tumors
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Pituitary Gland Disorders and Treatments
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Hormonal Regulation and Hypertension
- Keywords
- unilateral adrenalectomy, bilateral adrenalectomy, cortisol-producing adenoma, Cushing's syndrome, hypercortisolism, adrenal crisis, long-term outcomes, mortality, biochemical remission
- MeSH terms
- Adrenal Cortex, Adrenal Cortex Diseases, Adrenal Cortex Neoplasms, Adrenalectomy, Adult, Aged, Cushing Syndrome, Female, Humans, Hydrocortisone, Hyperplasia, Male, Middle Aged, Quality of Life, Retrospective Studies, Treatment Outcome, Adrenocortical Adenoma
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