A randomized, prospective trial of operative treatments for hyperparathyroidism in patients with multiple endocrine neoplasia type 1

Lairmore, Terry C.; Govednik, Cara M.; Gibson, Courtney; Sigmond, Benjamin R.; Lee, Cortney Y.; Jupiter, Daniel C.

doi:10.1016/j.surg.2014.08.006

Cited by 44 publications

(27 citation statements)

References 23 publications

(54 reference statements)

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“…Overall permanent hypoparathyroidism was 9 % (12 % in the subtotal parathyroidectomy vs. 7 % in the total parathyroidectomy + autotransplantation group). The study shows comparable outcomes for MEN 1 patients treated by either approach, but subtotal parathyroidectomy may have advantages because it involves only one surgical incision and avoids an obligate period of transient postoperative hypoparathyroidism [32].…”

Section: Surgical Treatmentmentioning

confidence: 80%

Hereditary hyperparathyroidism—a consensus report of the European Society of Endocrine Surgeons (ESES)

Iacobone

Carnaille

Palazzo

et al. 2015

Langenbecks Arch Surg

115

126

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Hereditary hyperparathyroidism typically presents at an earlier age than the sporadic variants. Gene penetrance and expressivity varies. Parathyroid multiple gland involvement is common, but in some variants, it may occur metachronously often with long disease-free intervals, simulating a single-gland involvement. Bilateral neck exploration with subtotal parathyroidectomy or total parathyroidectomy + autotransplantation should be performed, especially in MEN 1, in order to decrease the persistent and recurrent hyperparathyroidism rates; in some variants (MEN 2A, HPT-JT), limited parathyroidectomy can achieve long-term normocalcemia. In FHH, surgery is contraindicated; in NSHPT, urgent total parathyroidectomy is required. In FIHPT, MEN 4 and ADMH, a tailored case-specific approach is recommended.

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Section: Surgical Treatmentmentioning

confidence: 80%

Hereditary hyperparathyroidism—a consensus report of the European Society of Endocrine Surgeons (ESES)

Iacobone

Carnaille

Palazzo

et al. 2015

Langenbecks Arch Surg

115

126

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“…Some authors preferring TPTX report lower recurrence and disease persistence rates compared with SPTX. [22][23][24] However, the only randomized trial 25 comparing SPTX and TPTX in 32 MEN1 patients did not reveal a different recurrence rate (24% vs 13%, P = .66), as it was the case in the present study and some other retrospective studies. 19,[26][27][28] Recurrence rates after SGE were up to 100% and thus significantly higher compared with TPTX and SPTX.…”

Section: Discussionmentioning

confidence: 92%

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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“…This ideal study will include patients with new diagnosis of MEN1 who would be screened at baseline for thymic and bronchial carcinoid tumours and then randomized to different diagnostic strategies (modality and interval of evaluation) and assessed for differences in mortality (both overall and disease specific) and pathologic stage at diagnosis. Even though MEN1 is rare, a randomized trial assessing the effect of different surgical treatment options for primary hyperparathyroidism was carried out in two centres, suggesting feasibility of this study design …”

Section: Proposed Solutionsmentioning

confidence: 99%

When and how should patients with multiple endocrine neoplasia type 1 be screened for thymic and bronchial carcinoid tumours?

Ospina

Maraka

Montori

et al. 2015

Clinical Endocrinology

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Patients with multiple endocrine neoplasia type 1 (MEN1) are commonly evaluated for clinical manifestations of this syndrome with the rationale that early diagnosis and adequate treatment will result in improved survival and quality of life. Thymic and bronchial carcinoid tumours are uncommon but important manifestations of MEN1. Current practice guidelines recommend evaluation with computed tomography or magnetic resonance imaging scan of the chest every 1-2 years to detect these neoplasms. However, the certainty that patients will be better off (increased survival or quality of life) as a result of this case detection strategy is based on evidence at moderate-high risk of bias that yields only imprecise results of indirect relevance to these patients. In order to improve the care that patients with MEN1 receive, co-ordinated efforts from different stakeholders are required so that large, prospective, multicentre studies evaluating patient important outcomes are carried out.

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A randomized, prospective trial of operative treatments for hyperparathyroidism in patients with multiple endocrine neoplasia type 1

Cited by 44 publications

References 23 publications

Hereditary hyperparathyroidism—a consensus report of the European Society of Endocrine Surgeons (ESES)

Hereditary hyperparathyroidism—a consensus report of the European Society of Endocrine Surgeons (ESES)

Single gland excision for MEN1‐associated primary hyperparathyroidism

When and how should patients with multiple endocrine neoplasia type 1 be screened for thymic and bronchial carcinoid tumours?

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