Skip to main content
Top
Published in: World Journal of Surgery 12/2016

Open Access 01-12-2016 | Original Scientific Report

Unilateral Clearance for Primary Hyperparathyroidism in Selected Patients with Multiple Endocrine Neoplasia Type 1

Authors: Wouter P. Kluijfhout, Toni Beninato, Frederick Thurston Drake, Menno R. Vriens, Jessica Gosnell, Wen T. Shen, Insoo Suh, Chienying Liu, Quan-Yang Duh

Published in: World Journal of Surgery | Issue 12/2016

Login to get access

Abstract

Background

Primary hyperparathyroidism is the most common manifestation of multiple endocrine neoplasia type 1 (MEN1). Guidelines advocate subtotal parathyroidectomy (STP) or total parathyroidectomy with autotransplantation due to high prevalence of multiglandular disease; however, both are associated with a significant risk of permanent hypoparathyroidism. More accurate imaging and use of intraoperative PTH levels may allow a less extensive initial parathyroidectomy (unilateral clearance, removing both parathyroids with cervical thymectomy) in selected MEN1 patients with primary hyperparathyroidism.

Methods

We performed a retrospective cohort study at a high-volume tertiary medical center including patients with MEN1 and primary hyperparathyroidism, who underwent STP or unilateral clearance as their initial surgery from 1995 to 2015. Unilateral clearance was offered to patients who had concordant sestamibi and ultrasound showing a single enlarged parathyroid gland. For both the groups, we compared rates of persistent/recurrent disease and permanent hypoparathyroidism.

Results

Eight patients had unilateral clearance and 16 had STP. Subtotal parathyroidectomy patients were younger (37 vs 52 years). One patient in each group had persistent disease. One (13 %) unilateral clearance and five (31 %) STP patients had recurrent hyperparathyroidism after a mean follow-up of 47 and 68 months (p = 0.62). No unilateral clearance patients and two of 16 SPT patients had permanent hypoparathyroidism (p = 0.54).

Conclusions

Some MEN1 patients with primary hyperparathyroidism who have concordant localizing studies may be selected for unilateral clearance as an alternative to STP. For appropriately selected MEN1 patients, unilateral clearance can achieve similar results as STP and has no risk of permanent hypoparathyroidism, and may facilitate possible future reoperations.
Literature
1.
go back to reference Machens A, Schaaf L, Karges W et al (2007) Age-related penetrance of endocrine tumours in multiple endocrine neoplasia type 1 (MEN1): a multicentre study of 258 gene carriers. Clin Endocrinol (Oxf) 67:613–622 Machens A, Schaaf L, Karges W et al (2007) Age-related penetrance of endocrine tumours in multiple endocrine neoplasia type 1 (MEN1): a multicentre study of 258 gene carriers. Clin Endocrinol (Oxf) 67:613–622
2.
go back to reference Kebebew E, Hwang J, Reiff E et al (2006) Predictors of single-gland vs multigland parathyroid disease in primary hyperparathyroidism: a simple and accurate scoring model. Arch Surg 141:777–782CrossRefPubMed Kebebew E, Hwang J, Reiff E et al (2006) Predictors of single-gland vs multigland parathyroid disease in primary hyperparathyroidism: a simple and accurate scoring model. Arch Surg 141:777–782CrossRefPubMed
3.
go back to reference Schreinemakers JMJ, Pieterman CRC, Scholten A et al (2011) The optimal surgical treatment for primary hyperparathyroidism in MEN1 patients: a systematic review. World J Surg 35:1993–2005CrossRefPubMed Schreinemakers JMJ, Pieterman CRC, Scholten A et al (2011) The optimal surgical treatment for primary hyperparathyroidism in MEN1 patients: a systematic review. World J Surg 35:1993–2005CrossRefPubMed
4.
go back to reference Nilubol N, Weinstein LS, Simonds WF et al (2015) Limited parathyroidectomy in multiple endocrine neoplasia type 1-associated primary hyperparathyroidism: a setup for failure. Ann Surg Oncol. doi:10.1245/s10434-015-4865-9 Nilubol N, Weinstein LS, Simonds WF et al (2015) Limited parathyroidectomy in multiple endocrine neoplasia type 1-associated primary hyperparathyroidism: a setup for failure. Ann Surg Oncol. doi:10.​1245/​s10434-015-4865-9
5.
go back to reference Thakker RV, Newey PJ, Walls GV et al (2012) Clinical practice guidelines for multiple endocrine neoplasia type 1 (MEN1). J Clin Endocrinol Metab 97:2990–3011CrossRefPubMed Thakker RV, Newey PJ, Walls GV et al (2012) Clinical practice guidelines for multiple endocrine neoplasia type 1 (MEN1). J Clin Endocrinol Metab 97:2990–3011CrossRefPubMed
6.
go back to reference Fraker DL, Norton JA (1989) The role of surgery in the management of islet cell tumors. Gastroenterol Clin N Am 8:805–830 Fraker DL, Norton JA (1989) The role of surgery in the management of islet cell tumors. Gastroenterol Clin N Am 8:805–830
7.
go back to reference Tonelli F, Marcucci T, Fratini G et al (2007) Is total parathyroidectomy the treatment of choice for hyperparathyroidism in multiple endocrine neoplasia type 1. Ann Surg 246:1075–1082CrossRefPubMed Tonelli F, Marcucci T, Fratini G et al (2007) Is total parathyroidectomy the treatment of choice for hyperparathyroidism in multiple endocrine neoplasia type 1. Ann Surg 246:1075–1082CrossRefPubMed
8.
go back to reference Lee CH, Tseng LM, Chen JY et al (2006) Primary hyperparathyroidism in multiple endocrine neoplasia type 1: individualized management with low recurrence rates. Ann Surg Oncol 13:103–109CrossRefPubMed Lee CH, Tseng LM, Chen JY et al (2006) Primary hyperparathyroidism in multiple endocrine neoplasia type 1: individualized management with low recurrence rates. Ann Surg Oncol 13:103–109CrossRefPubMed
9.
go back to reference Versnick M, Popadich A, Sidhu S et al (2013) Minimally invasive parathyroidectomy provides a conservative surgical option for multiple endocrine neoplasia type 1-primary hyperparathyroidism. Surgery 154:101–105CrossRefPubMed Versnick M, Popadich A, Sidhu S et al (2013) Minimally invasive parathyroidectomy provides a conservative surgical option for multiple endocrine neoplasia type 1-primary hyperparathyroidism. Surgery 154:101–105CrossRefPubMed
10.
go back to reference Hellman P, Skogseid B, Oberg K et al (1998) Primary and reoperative parathyroid operations in hyperparathyroidism of multiple endocrine neoplasia type 1. Surgery 124:993–999CrossRefPubMed Hellman P, Skogseid B, Oberg K et al (1998) Primary and reoperative parathyroid operations in hyperparathyroidism of multiple endocrine neoplasia type 1. Surgery 124:993–999CrossRefPubMed
11.
go back to reference Kraimps JL, Duh QY, Demeure M et al (1992) Hyperparathyroidism in multiple endocrine neoplasia syndrome. Surgery 112:1080–1088PubMed Kraimps JL, Duh QY, Demeure M et al (1992) Hyperparathyroidism in multiple endocrine neoplasia syndrome. Surgery 112:1080–1088PubMed
12.
go back to reference Carling T (2005) Multiple endocrine neoplasia syndrome: genetic basis for clinical management. Curr Opin Oncol 17:7–12CrossRefPubMed Carling T (2005) Multiple endocrine neoplasia syndrome: genetic basis for clinical management. Curr Opin Oncol 17:7–12CrossRefPubMed
13.
go back to reference Goudet P, Cougard P, Vergès B et al (2001) Hyperparathyroidism in multiple endocrine neoplasia type 1: surgical trends and results of a 256-patient series from groupe d’etude des néoplasies endocriniennes multiples study group. World J Surg 225:886–890CrossRef Goudet P, Cougard P, Vergès B et al (2001) Hyperparathyroidism in multiple endocrine neoplasia type 1: surgical trends and results of a 256-patient series from groupe d’etude des néoplasies endocriniennes multiples study group. World J Surg 225:886–890CrossRef
14.
go back to reference Pieterman CRC, van Hulsteijn LT, den Heijer M et al (2012) Primary hyperparathyroidism in MEN1 patients. Ann Surg 255:1171–1178CrossRefPubMed Pieterman CRC, van Hulsteijn LT, den Heijer M et al (2012) Primary hyperparathyroidism in MEN1 patients. Ann Surg 255:1171–1178CrossRefPubMed
15.
go back to reference Lemmens I, Van de Ven WJ, Kas K et al (1997) Identification of the multiple endocrine neoplasia type 1 (MEN1) gene. The European Consortium on MEN1. Hum Mol Genet 6:1177–1183CrossRefPubMed Lemmens I, Van de Ven WJ, Kas K et al (1997) Identification of the multiple endocrine neoplasia type 1 (MEN1) gene. The European Consortium on MEN1. Hum Mol Genet 6:1177–1183CrossRefPubMed
16.
go back to reference Chandrasekharappa SC, Guru SC, Manickam P et al (1997) Positional cloning of the gene for multiple endocrine neoplasia-type 1. Science 276:404–407CrossRefPubMed Chandrasekharappa SC, Guru SC, Manickam P et al (1997) Positional cloning of the gene for multiple endocrine neoplasia-type 1. Science 276:404–407CrossRefPubMed
17.
go back to reference Bartsch DK, Langer P, Wild A et al (2000) Pancreaticoduodenal endocrine tumors in multiple endocrine neoplasia type 1: surgery or surveillance. Surgery 128:958–966CrossRefPubMed Bartsch DK, Langer P, Wild A et al (2000) Pancreaticoduodenal endocrine tumors in multiple endocrine neoplasia type 1: surgery or surveillance. Surgery 128:958–966CrossRefPubMed
18.
go back to reference Elaraj DM, Sippel RS, Lindsay S et al (2010) Are additional localization studies and referral indicated for patients with primary hyperparathyroidism who have negative sestamibi scan results. Arch Surg 145:578–581CrossRefPubMed Elaraj DM, Sippel RS, Lindsay S et al (2010) Are additional localization studies and referral indicated for patients with primary hyperparathyroidism who have negative sestamibi scan results. Arch Surg 145:578–581CrossRefPubMed
19.
go back to reference Berber E, Parikh RT, Ballem N et al (2008) Factors contributing to negative parathyroid localization: an analysis of 1000 patients. Surgery 144:74–79CrossRefPubMed Berber E, Parikh RT, Ballem N et al (2008) Factors contributing to negative parathyroid localization: an analysis of 1000 patients. Surgery 144:74–79CrossRefPubMed
20.
go back to reference Nilubol N, Weisbrod AB, Weinstein LS et al (2013) Utility of intraoperative parathyroid hormone monitoring in patients with multiple endocrine neoplasia type 1-associated primary hyperparathyroidism undergoing initial parathyroidectomy. World J Surg 37:1966–1972CrossRefPubMed Nilubol N, Weisbrod AB, Weinstein LS et al (2013) Utility of intraoperative parathyroid hormone monitoring in patients with multiple endocrine neoplasia type 1-associated primary hyperparathyroidism undergoing initial parathyroidectomy. World J Surg 37:1966–1972CrossRefPubMed
21.
go back to reference Malmaeus J, Benson L, Johansson H et al (1986) Parathyroid surgery in the multiple endocrine neoplasia type 1 syndrome: choice of surgical procedure. World J Surg 10:668–672CrossRefPubMed Malmaeus J, Benson L, Johansson H et al (1986) Parathyroid surgery in the multiple endocrine neoplasia type 1 syndrome: choice of surgical procedure. World J Surg 10:668–672CrossRefPubMed
22.
go back to reference Elaraj DM, Skarulis MC, Libutti SK et al (2003) Results of initial operation for hyperparathyroidism in patients with multiple endocrine neoplasia type 1. Surgery 134:855–858CrossRef Elaraj DM, Skarulis MC, Libutti SK et al (2003) Results of initial operation for hyperparathyroidism in patients with multiple endocrine neoplasia type 1. Surgery 134:855–858CrossRef
Metadata
Title
Unilateral Clearance for Primary Hyperparathyroidism in Selected Patients with Multiple Endocrine Neoplasia Type 1
Authors
Wouter P. Kluijfhout
Toni Beninato
Frederick Thurston Drake
Menno R. Vriens
Jessica Gosnell
Wen T. Shen
Insoo Suh
Chienying Liu
Quan-Yang Duh
Publication date
01-12-2016
Publisher
Springer International Publishing
Published in
World Journal of Surgery / Issue 12/2016
Print ISSN: 0364-2313
Electronic ISSN: 1432-2323
DOI
https://doi.org/10.1007/s00268-016-3624-9

Other articles of this Issue 12/2016

World Journal of Surgery 12/2016 Go to the issue