Impact of “Tailored” Parathyroidectomy for Treatment of Primary Hyperparathyroidism in Patients with Multiple Endocrine Neoplasia Type 1

Horiuchi, Kiyomi; Sakurai, Miki; Haniu, Kento; Nagai, Erin; Tokumitsu, Hiroki; Yoshida, Yusaku; Omi, Yoko; Sakamoto, Akiko; Okamoto, Takahiro

doi:10.1007/s00268-017-4366-z

Cited by 18 publications

(20 citation statements)

References 34 publications

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“…One major drawback of SPTX and TPTX, however, is their reported high rate of permanent postoperative hypoparathyroidism of up to 66% (Table ), which was also the case in the present study with 32% after TPTX and 17% after SPTX, respectively. All patients after TPTX developed a transient postoperative hypoparathyroidism requiring substitution therapy.…”

Section: Discussionsupporting

confidence: 76%

Single gland excision for MEN1‐associated primary hyperparathyroidism

Manoharan

Albers

Bollmann

et al. 2019

Clinical Endocrinology

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Importance: Guidelines advocate subtotal parathyroidectomy (SPTX) or total parathyroidectomy with autotransplantation (TPTX) with bilateral cervical thymectomy for primary hyperparathyroidism (pHPT) associated with multiple endocrine neoplasia type 1 (MEN1). However, both procedures are associated with a significant risk of permanent hypoparathyroidism. Objective:The aim of the current study was to compare long-term results of either single gland excision (SGE, 1-2 glands), SPTX and TPTX for the treatment of MEN1associated pHPT.Design and setting: Data of genetically confirmed MEN1 patients who underwent surgery for pHPT between 1987 and 2017 were retrieved from a prospective database and were retrospectively analysed.Results: Eighty-nine MEN1 patients underwent either TPTX (n = 38, 42.7%), SPTX (n = 23, 25.8%) or SGE (n = 28, 31.5%). The rate of disease persistence after initial surgery was 2.6%, 0% and 14.2% in the TPTX, SPTX and SGE groups, respectively.After median follow-up of 112 (range 7-411) months, the rate of recurrent pHPT was significantly higher in the SGE group (n = 19, 21.3%) compared with the TPTX (n = 4, 4.4%, P = .001) and the SPTX (n = 9, 10.1%, P = .03) groups. Analysis of the recurrence-free time among the surgical groups revealed a significant difference (P = .036). The median time to recurrence was significantly shorter after SGE (101, range 3-301 months) than after SPTX (139, range 28-278 months, P = .018) and TPTX (204, range 75-396 months, P = .049). Twelve (32%) patients who underwent TPTX developed permanent hypoparathyroidism compared with only 4 (17%, P = .06) in the SPTX and 0 in the SGE group (P = .001). Conclusion:Given the high rate of postoperative permanent hypoparathyroidism after TPTX and SPTX, SGE is a valid option for the treatment of MEN1-associated pHPT. K E Y W O R D Shypocalcemia, hypoparathyroidism, laryngeal nerve palsy, multiple endocrine neoplasia type 1, primary hyperparathyroidism, recurrence, surgery

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Section: Discussionsupporting

confidence: 76%

Single gland excision for MEN1‐associated primary hyperparathyroidism

Manoharan

Albers

Bollmann

et al. 2019

Clinical Endocrinology

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“…The main cause of the failure of a limited PTX is the inadequacy of the preoperative localizing studies, missing enlarged controlateral parathyroid glands in 86% of the patients. However, other authors have obtained positive results with limited interventions, such as the unilateral clearance (excision of the parathyroid glands and thymus from only one cervical side) ( 8 , 23 ) or selective parathyroidectomy ( 7 , 24 , 25 ). Most of the patients operated by limited PTX are older, and there is too little follow-up time for the results achieved to be reliable ( 22 ).…”

Section: Discussionmentioning

confidence: 99%

Total and Subtotal Parathyroidectomy in Young Patients With Multiple Endocrine Neoplasia Type 1-Related Primary Hyperparathyroidism: Potential Post-surgical Benefits and Complications

et al. 2018

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Background: The choice of surgical treatment for patients with Multiple Endocrine Neoplasia type 1 (MEN1)-related primary hyperparathyroidism (PHPT) remains controversial and it has not been specifically addressed in young patients.Methods: This is a retrospective case series study. The study includes the surgical data and the follow-up of 38 patients younger than 30 years of age, all diagnosed with MEN1, collected and followed-up between 1991 and 2017 at the Regional Referral Center for Inherited Endocrine Tumors of the Tuscany Region, and operated by parathyroidectomy. Genetic and/or clinical MEN1 diagnosis was made before surgery in all patients. Subtotal (9/38 patients) or total parathyroidectomy with auto-transplantation (28/38 patients) were performed in all patients but one, in whom a single mediastinal adenoma was excised from the aorto-pulmonary window. All patients but one, who was operated in 2017, had a post-operatory follow-up of at least 12 months.Results: Total parathyroidectomy (TPTX), with auto-transplantation, was the most frequently adopted operation both as primary (20/38 patients) and secondary (8/38 patients) surgery, followed by subtotal parathyroidectomy (SPTX; 9/38 patients) and limited parathyroidectomy (1/38 patient). At follow-up, lasting a mean of 11.8 ± 6.6 years (range 0–23 years), no persistent PHPT was observed. PHPT recurred in 4/28 TPTX (14%) and in 2/9 SPTX (22%). Permanent hypoparathyroidism showed no statistically significant difference between the procedures (2/9 in SPTX and 5/28 in TPTX).Conclusions: Data from this retrospective study showed the efficacy of TPTX for the treatment of MEN1-PHPT, also in adolescent and young patients, showing, in our series, no risk of PHPT permanence and a longer disease-free period and, subsequently, the possibility to postpone re-intervention with respect to both limited PTX and SPTX. The risk of permanent hypoparathyroidism in TPTX was comparable to STPX, and could be mitigated over the years.

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“…A few centers have recently reported their experience with performing a less-than-subtotal (SPTX) or total (TPTX) parathyroidectomy for MEN1 patients. Given the high rate of post-operative hypocalcemia from those two procedures, especially TPTX, which can be debilitating, as well as the high rate of recurrent disease requiring reoperation after SPTX, a more limited initial approach seems reasonable in select patients [65][66][67] . Patients with positive pre-operative localization would be offered a more focused approach, whether single-gland or unilateral (i.e.…”

Section: Long-term Outcomesmentioning

confidence: 99%

Recent advances in the understanding and management of primary hyperparathyroidism

Goldfarb

Singer

2020

F1000Res

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Primary hyperparathyroidism is a hormonal disorder whose prevalence is approximately 1–2% in the United States of America. The disease has become more recognizable to clinicians in an earlier phase and, at present, patients can be diagnosed with “classic”, “normocalcemic”, “normohormonal”, or “mild, asymptomatic” primary hyperparathyroidism. Surgery, with a focused parathyroidectomy when possible, or a four-gland exploration, is the only way to cure the disease. Cure is determined by use of intra-operative parathyroid hormone monitoring with long-term cure rates ranging from 90–95%. Newer adjuncts to surgery include CT or PET imaging and near-infrared immunofluorescence. This article highlights updates in parathyroid disease and advances in parathyroid surgery; it does not provide a comprehensive summary of the disease process or a review of surgical indications, which can be found in the AAES guidelines or NIH Symposium on primary hyperparathyroidism.

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Impact of “Tailored” Parathyroidectomy for Treatment of Primary Hyperparathyroidism in Patients with Multiple Endocrine Neoplasia Type 1

Abstract: Although LPTX was not satisfactory as a standard procedure, both SPTX and TPTX are effective treatment methods for PHPT in patients with MEN1. The parathyroidectomy strategy should be based on intraoperative evaluation of the parathyroid glands.

Cited by 18 publications

References 34 publications

Single gland excision for MEN1‐associated primary hyperparathyroidism

Single gland excision for MEN1‐associated primary hyperparathyroidism

Total and Subtotal Parathyroidectomy in Young Patients With Multiple Endocrine Neoplasia Type 1-Related Primary Hyperparathyroidism: Potential Post-surgical Benefits and Complications

Recent advances in the understanding and management of primary hyperparathyroidism

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