Potential benefits of intraoperative parathyroid autofluorescence imaging in a patient with multiple endocrine neoplasia type 1 and hyperparathyroidism - A case report

TY - JOUR

T1 - Potential benefits of intraoperative parathyroid autofluorescence imaging in a patient with multiple endocrine neoplasia type 1 and hyperparathyroidism - A case report

AU - Egebæk, Christine Holm

AU - Lilja-Fischer, Jacob

AU - Rejnmark, Lars

AU - Rolighed, Lars

N1 - Copyright © 2024. Published by Elsevier Ltd.

PY - 2025/1

Y1 - 2025/1

N2 - INTRODUCTION AND IMPORTANCE: Primary hyperparathyroidism (PHPT) is a frequent complication to multiple endocrine neoplasia type 1 (MEN1), presenting challenges due to increased risk of multi-gland disease and recurrence post parathyroidectomy (PTX). This case report examines the management of PHPT in a MEN1 patient, emphasizing possible benefits from intraoperative parathyroid autofluorescence imaging (AF).CASE PRESENTATION: A 21-year-old woman with MEN1 presented with mild hyperparathyroidism symptoms in 2014. Initial unilateral PTX in 2015 normalized plasma parathyroid hormone (PTH) and ionized calcium (Ca2+) but was followed by biochemical recurrence in 2018. In 2022 a subtotal PTX was performed using AF to reduce the risk of permanent hypoparathyroidism. This technique distinguished pathological from non-pathological parathyroid tissue. Most of two glands were removed, leaving a small rim of non-pathologic tissue, achieving long-term normalization of plasma PTH and Ca2+. Despite prior pregnancy complications suspectedly related to hypercalcemia, she gave birth to a healthy child after the second PTX, who tested negative for MEN1.CLINICAL DISCUSSION: This case highlights the complexity of managing MEN1-associated PHPT, demonstrating the importance of advanced surgical techniques like AF to minimize complications and enhance outcomes. Surgical and medical management of MEN1 is crucial given the high recurrence-rate. According to the literature, hypercalcemia in patients with PHPT may affect pregnancy outcomes.CONCLUSION: This case-report emphasize the need for a tailored approach in MEN1-related PHPT, combining innovative surgical methods to optimize outcomes. Further research on the impact of MEN1 on pregnancy outcomes and the long-term efficacy of AF are essential for improving management strategies.

AB - INTRODUCTION AND IMPORTANCE: Primary hyperparathyroidism (PHPT) is a frequent complication to multiple endocrine neoplasia type 1 (MEN1), presenting challenges due to increased risk of multi-gland disease and recurrence post parathyroidectomy (PTX). This case report examines the management of PHPT in a MEN1 patient, emphasizing possible benefits from intraoperative parathyroid autofluorescence imaging (AF).CASE PRESENTATION: A 21-year-old woman with MEN1 presented with mild hyperparathyroidism symptoms in 2014. Initial unilateral PTX in 2015 normalized plasma parathyroid hormone (PTH) and ionized calcium (Ca2+) but was followed by biochemical recurrence in 2018. In 2022 a subtotal PTX was performed using AF to reduce the risk of permanent hypoparathyroidism. This technique distinguished pathological from non-pathological parathyroid tissue. Most of two glands were removed, leaving a small rim of non-pathologic tissue, achieving long-term normalization of plasma PTH and Ca2+. Despite prior pregnancy complications suspectedly related to hypercalcemia, she gave birth to a healthy child after the second PTX, who tested negative for MEN1.CLINICAL DISCUSSION: This case highlights the complexity of managing MEN1-associated PHPT, demonstrating the importance of advanced surgical techniques like AF to minimize complications and enhance outcomes. Surgical and medical management of MEN1 is crucial given the high recurrence-rate. According to the literature, hypercalcemia in patients with PHPT may affect pregnancy outcomes.CONCLUSION: This case-report emphasize the need for a tailored approach in MEN1-related PHPT, combining innovative surgical methods to optimize outcomes. Further research on the impact of MEN1 on pregnancy outcomes and the long-term efficacy of AF are essential for improving management strategies.

KW - Autofluorescence

KW - Case report

KW - Hyperparathyroidism

KW - Multiple endocrine neoplasia

KW - Parathyroidectomy

UR - http://www.scopus.com/inward/record.url?scp=85213293118&partnerID=8YFLogxK

U2 - 10.1016/j.ijscr.2024.110764

DO - 10.1016/j.ijscr.2024.110764

M3 - Journal article

C2 - 39740418

SN - 2210-2612

VL - 126

SP - 110764

JO - International Journal of Surgery Case Reports

JF - International Journal of Surgery Case Reports

M1 - 110764

ER -