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Posterior retroperitoneoscopic adrenal surgery for clinical and subclinical Cushing’s syndrome in patients with bilateral adrenal disease

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Abstract

Background

The treatment of hypercortisolism for patients with bilateral adrenal disease (BAD) is controversial. Bilateral total adrenalectomy results in permanent hypocortisolaemia requiring lifelong steroid replacement. A more conservative surgical approach, with less than bilateral total adrenalectomy (leaving functional adrenal tissue either unilaterally or bilaterally), represents an alternative option; however, long-term outcome or recurrence data are limited. We report our experience with the surgical management of hypercortisolism caused by BAD.

Methods

Between 2004 and 2016, 42 patients (12 male, 30 female; mean age 58 ± 10 years) with clinical or subclinical Cushing’s syndrome (CS/sCS) caused by BAD underwent adrenal surgery via the posterior retroperitoneoscopic approach. Adrenal surgery was defined as “adrenalectomy” when total gland excision was performed or “resection” when a partial or subtotal adrenal resection was performed. Clinical, radiological and biochemical parameters were evaluated preoperatively and postoperatively.

Results

Seventy adrenal operations performed in total included unilateral resection (n = 3), unilateral adrenalectomy (n = 15), bilateral resection (n = 9), adrenalectomy and contralateral resection (n = 14) and bilateral total adrenalectomy (n = 3). Median operating time was 47.5 min (30–150) with no difference between unilateral and bilateral (synchronous included) procedures (p = 0.15). Mortality was zero. Clavien–Dindo grade of postoperative complications was I (n = 5) and IV (n = 3). All but one patient with CS and 17/31 patients with sCS received postoperative steroid supplementation for a median duration of 20 (1.5–129) months. After median follow-up of 40 months (3–129), the remission rate was 92%; 11 patients required ongoing steroid supplementation. There were three biochemical recurrences (two underwent contralateral resection); two patients with new/progressive radiological nodularity are biochemically eucortisolaemic. A significant reduction in BMI (p = 0.01) and antihypertensive requirements (p = 0.04) was observed postoperatively.

Conclusion

A surgical approach which facilitates the conservation of functional adrenal tissue represents a suitable strategy for hypercortisolism caused by BAD. This approach avoids the necessity for lifelong steroid replacement in the majority of cases with low rates of adrenal insufficiency and recurrence.

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References

  1. Lacroix A et al (2015) Cushing’s syndrome. Lancet 386(9996):913–927

    Article  CAS  PubMed  Google Scholar 

  2. Lacroix A (2009) ACTH-independent macronodular adrenal hyperplasia. Best Pract Res Clin Endocrinol Metab 23(2):245–259

    Article  CAS  PubMed  Google Scholar 

  3. Stratakis CA (2008) Cushing syndrome caused by adrenocortical tumors and hyperplasias (corticotropin-independent Cushing syndrome). Endocr Dev 13:117–132

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  4. Nieman LK (2015) Cushing’s syndrome: update on signs, symptoms and biochemical screening. Eur J Endocrinol 173(4):M33–M38

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  5. Terzolo M, Pia A, Reimondo G (2012) Subclinical Cushing’s syndrome: definition and management. Clin Endocrinol 76(1):12–18

    Article  CAS  Google Scholar 

  6. Nieman LK et al (2015) Treatment of Cushing’s syndrome: an Endocrine Society Clinical Practice Guideline. J Clin Endocrinol Metab 100(8):2807–2831

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  7. Di Dalmazi G et al (2014) Cardiovascular events and mortality in patients with adrenal incidentalomas that are either non-secreting or associated with intermediate phenotype or subclinical Cushing’s syndrome: a 15-year retrospective study. Lancet Diabetes Endocrinol 2(5):396–405

    Article  PubMed  Google Scholar 

  8. Lacroix A et al (2010) Aberrant G-protein coupled receptor expression in relation to adrenocortical overfunction. Clin Endocrinol 73(1):1–15

    CAS  Google Scholar 

  9. Albiger NM et al (2015) An analysis of different therapeutic options in patients with Cushing’s syndrome due to bilateral macronodular adrenal hyperplasia: a single-centre experience. Clin Endocrinol 82(6):808–815

    Article  CAS  Google Scholar 

  10. Hahner S et al (2015) High incidence of adrenal crisis in educated patients with chronic adrenal insufficiency: a prospective study. J Clin Endocrinol Metab 100(2):407–416

    Article  CAS  PubMed  Google Scholar 

  11. Prajapati OP et al (2015) Bilateral adrenalectomy for Cushing’s syndrome: pros and cons. Indian J Endocrinol Metab 19(6):834–840

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  12. Guerin C et al (2016) Bilateral adrenalectomy in the 21st century: when to use it for hypercortisolism? Endocr Relat Cancer 23(2):R131–R142

    Article  CAS  PubMed  Google Scholar 

  13. Grossman A et al (2013) Therapy of endocrine disease: perspectives on the management of adrenal insufficiency: clinical insights from across Europe. Eur J Endocrinol 169(6):R165–R175

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  14. Debillon E et al (2015) Unilateral adrenalectomy as a first-line treatment of Cushing’s syndrome in patients with primary bilateral macronodular adrenal hyperplasia. J Clin Endocrinol Metab 100(12):4417–4424

    Article  CAS  PubMed  Google Scholar 

  15. Perogamvros I et al (2015) Biochemical and clinical benefits of unilateral adrenalectomy in patients with subclinical hypercortisolism and bilateral adrenal incidentalomas. Eur J Endocrinol 173(6):719–725

    Article  CAS  PubMed  Google Scholar 

  16. Li J, Yang CH (2015) Diagnosis and treatment of adrenocorticotrophic hormone-independent macronodular adrenocortical hyperplasia: a report of 23 cases in a single center. Exp Ther Med 9(2):507–512

    PubMed  Google Scholar 

  17. Xu Y et al (2013) The role of unilateral adrenalectomy in corticotropin-independent bilateral adrenocortical hyperplasias. World J Surg 37(7):1626–1632

    Article  PubMed  Google Scholar 

  18. Young WF et al (2008) The clinical conundrum of corticotropin-independent autonomous cortisol secretion in patients with bilateral adrenal masses. World J Surg 32(5):856–862

    Article  PubMed  Google Scholar 

  19. Alesina PF et al (2010) Posterior retroperitoneoscopic adrenalectomy for clinical and subclinical Cushing’s syndrome. World J Surg 34(6):1391–1397

    Article  PubMed  Google Scholar 

  20. Walz MK et al (2006) Posterior retroperitoneoscopic adrenalectomy—results of 560 procedures in 520 patients. Surgery 140(6):943–948 discussion 948-50

    Article  PubMed  Google Scholar 

  21. Di Dalmazi G et al (2015) Subclinical hypercortisolism: a state, a syndrome, or a disease? Eur J Endocrinol 173(4):M61–M71

    Article  CAS  PubMed  Google Scholar 

  22. Iacobone M et al (2015) Systematic review of surgical treatment of subclinical Cushing’s syndrome. Br J Surg 102(4):318–330

    Article  CAS  PubMed  Google Scholar 

  23. Papierska L et al (2015) Adrenal (131)I-6β-iodomethylnorcholesterol scintigraphy in choosing the side for adrenalectomy in bilateral adrenal tumors with subclinical hypercortisolemia. Abdom Imaging 40(7):2453–2460

    Article  PubMed  PubMed Central  Google Scholar 

  24. Castinetti F et al (2014) Outcomes of adrenal-sparing surgery or total adrenalectomy in phaeochromocytoma associated with multiple endocrine neoplasia type 2: an international retrospective population-based study. Lancet Oncol 15(6):648–655

    Article  PubMed  Google Scholar 

  25. Lowery AJ et al (2013) Molecular and therapeutic advances in the diagnosis and management of malignant pheochromocytomas and paragangliomas. Oncologist 18(4):391–407

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  26. Walz MK et al (2008) Retroperitoneoscopic adrenalectomy in Conn’s syndrome caused by adrenal adenomas or nodular hyperplasia. World J Surg 32(5):847–853

    Article  PubMed  Google Scholar 

  27. Alesina PF et al (2012) Minimally invasive cortical-sparing surgery for bilateral pheochromocytomas. Langenbeck's Arch Surg 397(2):233–238

    Article  Google Scholar 

  28. Yasuda A et al (2014) A rare case of Cushing’s syndrome due to bilateral adrenocortical adenomas. Tokai J Exp Clin Med 39(4):158–165

    PubMed  Google Scholar 

  29. Inoue T et al (2006) Laparoscopic bilateral partial adrenalectomy for adrenocortical adenomas causing Cushing’s syndrome: report of a case. Surg Today 36(1):94–97

    Article  PubMed  Google Scholar 

  30. Domino JP et al (2007) Laparoscopic partial adrenalectomy for bilateral cortisol-secreting adenomas. Asian J Surg 30(2):154–157

    Article  PubMed  Google Scholar 

  31. Kawasaki Y et al (2011) Laparoscopic simultaneous bilateral adrenalectomy: assessment of feasibility and potential indications. Int J Urol 18(11):762–767

    Article  PubMed  Google Scholar 

  32. Lombardi CP et al (2011) ACTH-dependent Cushing syndrome: the potential benefits of simultaneous bilateral posterior retroperitoneoscopic adrenalectomy. Surgery 149(2):299–300

    Article  PubMed  Google Scholar 

  33. Raffaelli M et al (2014) Synchronous bilateral adrenalectomy for Cushing’s syndrome: laparoscopic versus posterior retroperitoneoscopic versus robotic approach. World J Surg 38(3):709–715

    Article  PubMed  Google Scholar 

  34. Alexandraki KI et al (2013) Long-term remission and recurrence rates in Cushing’s disease: predictive factors in a single-centre study. Eur J Endocrinol 168(4):639–648

    Article  CAS  PubMed  Google Scholar 

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Authors’ Contributions

A. Lowery contributed to study design, acquisition, analysis and interpretation of data, drafting of manuscript and critical revision of manuscript. B. Seeliger contributed to acquisition, analysis and interpretation of data, drafting and critical revision of the manuscript. These authors (A. Lowery and B. Seeliger) contributed equally to this work. P. Alesina contributed to acquisition, analysis and interpretation of data, drafting of manuscript and critical appraisal of manuscript. M. Walz conceived of the study, contributed to acquisition and interpretation of data and critical appraisal of the manuscript.

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Authors and Affiliations

  1. Graduate Entry Medical School, University of Limerick and University Hospital Limerick, Limerick, Ireland

    Aoife J. Lowery

  2. Klinik für Chirurgie und Zentrum für Minimal Invasive Chirurgie, Kliniken Essen-Mitte, Evang. Huyssens-Stiftung/Knappschaft GmbH, Akademisches Lehrkrankenhaus der Universität Duisburg-Essen, Essen, Germany

    Barbara Seeliger, Pier F. Alesina & Martin K. Walz

Authors
  1. Aoife J. Lowery
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  2. Barbara Seeliger
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  4. Martin K. Walz
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Corresponding author

Correspondence to Aoife J. Lowery.

Ethics declarations

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.

All patients were treated by the institutional standard of care. Patient data were retrospectively analysed from the prospectively maintained institutional database. For this type of study, formal consent is not required (Professional Code of the German Medical Association, article B.III. § 15.1).

Funding

No external funding has been received to support this study.

Conflict of interest

The authors declare that they have no conflict of interest.

Additional information

Lowery AJ and Seeliger B contributed equally to this work.

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Lowery, A.J., Seeliger, B., Alesina, P.F. et al. Posterior retroperitoneoscopic adrenal surgery for clinical and subclinical Cushing’s syndrome in patients with bilateral adrenal disease. Langenbecks Arch Surg 402, 775–785 (2017). https://doi.org/10.1007/s00423-017-1569-6

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  • DOI: https://doi.org/10.1007/s00423-017-1569-6

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