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Jiang T, Chai H, Shan X, Deng E, Yu S, Bo X, Li X, He H, Zhang Z, Weng N, Qin X, Fan J, Tang X, Heng W, Li Y, Li J, Zhu J, Zhang H, Sun L, Peng C, Yue W, Xu H. Safety of radiofrequency ablation in patients with hyperparathyroidism: a 10-year real-world multicenter experience. Eur Radiol 2025:10.1007/s00330-025-11688-w. [PMID: 40410329 DOI: 10.1007/s00330-025-11688-w] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/28/2024] [Revised: 03/24/2025] [Accepted: 04/19/2025] [Indexed: 05/25/2025]
Abstract
BACKGROUND US-guided percutaneous radiofrequency ablation (RFA) is a validated procedure for hyperparathyroidism (HPT). However, it is associated with potentially significant complications, and data regarding the incidence and risk factors associated with RFA-related complications are limited. OBJECTIVE To assess the complication rates associated with RFA for HPT and identify the potential influential factors. METHODS This retrospective study was to investigate the associated complications in HPT patients who underwent RFA between January 2013 and April 2024. We collected and analyzed complications for these patients, focusing on their categories and rates. Logistic regression analysis was used to assess the crucial factors. RESULTS Three hundred twenty patients were included, including 117 cases of primary hyperparathyroidism (pHPT) and 203 cases of secondary hyperparathyroidism (sHPT). The overall complication rate was 22.2% (71/320), with 9.4% (30/320) being major complications and 14.7% (47/320) minor complications. Six patients experienced both major and minor complications. Among sHPT patients, 54 cases (26.6%, 54/203) experienced complications, including 22 major cases and 36 minor cases; however, among pHPT patients, 17 cases experienced complications (14.5%, 17/117), with 8 being major and 11 being minor. sHPT was identified as a risk factor for overall complications (odds ratio, 2.132; p = 0.014) and minor complications (odds ratio, 2.077; p = 0.046). Maximum diameter was the only risk factor for major complications (odds ratio, 1.508, p = 0.047). CONCLUSIONS RFA is a well-tolerated technique with an acceptably low incidence of major complications for HPT. Enhanced management strategies involving patients with sHPT and large hyperplastic glands may help mitigate complications. KEY POINTS Question Is ultrasound-guided RFA a safe nonsurgical alternative to traditional parathyroidectomy for patients with pHPT and sHPT who require intervention? Findings Ultrasound-guided RFA is a safe procedure for treating HPT. However, patients with sHPT and large hyperplastic glands are more likely to develop complications. Clinical relevance RFA is a well-accepted technique with a relatively low complication rate and can be used as a non-surgical alternative in the management of HPT.
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Affiliation(s)
- Tingting Jiang
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China
- Department of Ultrasound, Zhongshan Hospital, Fudan University, Shanghai, China
| | - Huihui Chai
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China
| | - Xuexia Shan
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China
| | - Erya Deng
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China
| | - Songyuan Yu
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China
| | - Xiaowan Bo
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China
| | - Xiaolong Li
- Department of Ultrasound, Zhongshan Hospital, Fudan University, Shanghai, China
| | - Hongfeng He
- Department of Ultrasound, Zhejiang Provincial People's Hospital, Hangzhou Medical College, Hangzhou, China
| | - Zhengxian Zhang
- Department of Ultrasound, Hangzhou Hospital of Traditional Chinese Medicine, Hangzhou, China
| | - Ning Weng
- Department of Nephrology, Hangzhou Hospital of Traditional Chinese Medicine, Hangzhou, China
| | - Xiachuan Qin
- Department of Ultrasound, The First Affiliated Hospital of Anhui Medical University, Hefei, China
| | - Jing Fan
- Department of Ultrasound, Yiwu Fuyuan Hospital, Yiwu, China
| | - Xiaoqing Tang
- Department of Ultrasound, Yiwu Fuyuan Hospital, Yiwu, China
| | - Wei Heng
- Department of Ultrasound, The First People's Hospital of Aksu, Xinjiang, China
| | - Yong Li
- Department of Nephrology, The First People's Hospital of Aksu, Xinjiang, China
| | - Jiaxin Li
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China
| | - Jinge Zhu
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China
| | - Huili Zhang
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China
| | - Liping Sun
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China.
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China.
| | - Chengzhong Peng
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China.
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China.
| | - Wenwen Yue
- Center of Minimally Invasive Treatment for Tumor, Department of Medical Ultrasound, Shanghai Tenth People's Hospital, School of Medicine, Tongji University, Shanghai, China.
- Shanghai Engineering Research Center of Ultrasound Diagnosis and Treatment; National Clinical Research Center for Interventional Medicine, Shanghai, China.
| | - Huixiong Xu
- Department of Ultrasound, Zhongshan Hospital, Fudan University, Shanghai, China
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Kamimura K, Terai S. Importance of understanding a diagnostic-treatment algorithm for primary hyperparathyroidism-induced acute pancreatitis during pregnancy. World J Gastroenterol 2025; 31:105866. [DOI: 10.3748/wjg.v31.i18.105866] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/08/2025] [Revised: 03/12/2025] [Accepted: 03/26/2025] [Indexed: 05/13/2025] Open
Abstract
In this article, we have commented on the article by Augustin et al. The authors presented a systematic review of the diagnosis, treatment, and outcomes of primary hyperparathyroidism-induced acute pancreatitis in pregnant women. Since acute pancreatitis during pregnancy could cause maternal as well as fetal adverse outcomes, understanding this pathology is essential. Although there are various etiologies of acute pancreatitis during pregnancy, primary hyperparathyroidism is one of the causes that complicate hypercalcemia. Along with conventional treatment for acute pancreatitis, parathyroidectomy can effectively normalize calcium levels and improve acute pancreatitis. Augustin et al have provided vital information that can enable physicians to understand and treat hyperparathyroidism-induced acute pancreatitis in pregnant women, which could contribute to better maternal and fetal outcomes. In addition, since primary hyperparathyroidism is associated with multiple endocrine neoplasia, further consideration regarding screening for multiple endocrine neoplasia might lead to better prognoses.
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Affiliation(s)
- Kenya Kamimura
- Department of General Medicine, Niigata University, Niigata 9518510, Japan
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
| | - Shuji Terai
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
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Bisceglia N, Malagrinò M, Cantone C, Capra S, Piazza A, Vandi G, Zavatta G. Trabecular bone score as a possible new surgical recommendation in patients with asymptomatic primary hyperparathyroidism. J Endocrinol Invest 2025:10.1007/s40618-025-02590-9. [PMID: 40279053 DOI: 10.1007/s40618-025-02590-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/02/2024] [Accepted: 04/17/2025] [Indexed: 04/26/2025]
Abstract
PURPOSE TBS usefulness in primary hyperparathyroidism (PHPT) management is uncertain. The primary aim was to evaluate the significance of performing Trabecular Bone Score (TBS) in addition to classical PHPT surgical recommendations. The explorative objective was to characterize bone quality in a cohort of patients with asymptomatic PHPT. METHODS From 1/1/2021 to 5/31/2024, of 265 consecutively evaluated PHPT patients, 125 met inclusion and exclusion criteria. Patients underwent complete biochemical evaluation in the same laboratory, Dual Energy X-ray Absorptiometry (DXA) at three sites, TBS with the same densitometer, and renal ultrasound. We retrospectively applied to each patient all the criteria to recommend parathyroidectomy as per the Fifth International Workshop, adding also TBS ≤ 1.2. RESULTS Almost one-third of patients presented degraded TBS (≤ 1.2), 36.0% partially degraded TBS, and 31.2% preserved TBS (≥ 1.35). Mean TBS was partially degraded (1.286 ± 0.134). Osteoporotic patients had lower TBS than osteopenic patients (1.207 ± 0.101 vs. 1.27 9 ± 0.110; p <.001). Patients with degraded TBS had a greater number of fragility fractures than those with TBS > 1.2 (34.2% vs. 8.3%; p <.001). In the group of osteopenic patients (34.4%), 10/43 (23.3%) showed degraded TBS. 13 patients (10.4%) did not meet any surgical criteria, and of these, 3/13 (23.1%) had degraded TBS and were osteopenic. CONCLUSION More than one-fifth of patients who did not meet the current surgical recommendations presented degraded TBS and osteopenia. The combination of degraded TBS and osteopenia as a possible new surgical recommendation could increase the number of patients who might benefit from curative parathyroidectomy, with potential reduction of fracture risk.
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Affiliation(s)
- Nicolò Bisceglia
- Division of Endocrinology and Diabetes Prevention and Care, IRCCS Azienda Ospedaliero- Universitaria, Bologna, Italy
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, Via Massarenti, 9, Bologna, 40138, Italy
| | - Matteo Malagrinò
- Division of Endocrinology and Diabetes Prevention and Care, IRCCS Azienda Ospedaliero- Universitaria, Bologna, Italy
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, Via Massarenti, 9, Bologna, 40138, Italy
| | - Cristiana Cantone
- Division of Endocrinology and Diabetes Prevention and Care, IRCCS Azienda Ospedaliero- Universitaria, Bologna, Italy
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, Via Massarenti, 9, Bologna, 40138, Italy
| | - Sara Capra
- Division of Endocrinology and Diabetes Prevention and Care, IRCCS Azienda Ospedaliero- Universitaria, Bologna, Italy
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, Via Massarenti, 9, Bologna, 40138, Italy
| | - Anna Piazza
- Division of Endocrinology and Diabetes Prevention and Care, IRCCS Azienda Ospedaliero- Universitaria, Bologna, Italy
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, Via Massarenti, 9, Bologna, 40138, Italy
| | - Giulia Vandi
- Division of Endocrinology and Diabetes Prevention and Care, IRCCS Azienda Ospedaliero- Universitaria, Bologna, Italy
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, Via Massarenti, 9, Bologna, 40138, Italy
| | - Guido Zavatta
- Division of Endocrinology and Diabetes Prevention and Care, IRCCS Azienda Ospedaliero- Universitaria, Bologna, Italy.
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, Via Massarenti, 9, Bologna, 40138, Italy.
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Gheorghe AM, Stanciu M, Nistor C, Lebada IC, Carsote M. Primary Hyperparathyroidism: An Analysis Amid the Co-Occurrence of Type 2 Diabetes Mellitus. Life (Basel) 2025; 15:677. [PMID: 40283231 PMCID: PMC12028805 DOI: 10.3390/life15040677] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/18/2025] [Revised: 04/14/2025] [Accepted: 04/18/2025] [Indexed: 04/29/2025] Open
Abstract
BACKGROUND Apart from classical elements in primary hyperparathyroidism (PHPT), non-classical complications, including type 2 diabetes mellitus (T2DM), are reported in some patients, but currently, they do not represent a parathyroidectomy (PTx) indication. OBJECTIVE to explore the latest data regarding glucose profile, particularly, T2DM and metabolic syndrome (MetS) in PHPT, including post-PTx. METHODS PubMed-based review included English-published original studies between January 2020 and December 2024 (n = 20). RESULTS Studied population: 764,485 subjects (female-to-male ratio of 1.26:1; 23,931 were PHPT patients vs. 740,502 controls). T2DM prevalence (n = 13; N = 763,645 patients; 55.92% females): 4-60% (higher vs. controls); for the largest study (N = 699,157) of 31.3%. Age-based analysis: higher T2DM prevalence at >50 vs. <50 years (14.4% vs. 2.6%, p < 0.001), but not all studies agreed. Concurrent vitamin D deficiency as a contributor to a higher risk had limited evidence. The association MetS-PHPT (n = 2) had no clear conclusion. Post-PTx showed the following: lower glycaemia, fasting insulin, insulin resistance (HOMA-IR) improvement, and reduced rate (but not all studies agreed). PHPT patients with prediabetes might represent the population sub-group with the highest post-PTx benefit. CONCLUSIONS The panel of PHPT-T2DM interplay remains heterogeneous. Data regarding post-PTx improvement of glucose disorders are still conflicting, recent findings suggested that surgery has beneficial effects, especially in patients with confirmed pre-existing prediabetes. Patients with the normocalcemic variant seemed to be less affected by the glucose-related disturbances, but further studies are needed. A better understanding of the intricate relationship between PHPT and glucose metabolism anomalies will help in providing optimal management to reduce the overall disease burden.
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Affiliation(s)
- Ana-Maria Gheorghe
- PhD Doctoral School of “Carol Davila” University of Medicine and Pharmacy, 020021 Bucharest, Romania;
| | - Mihaela Stanciu
- Department of Endocrinology, Faculty of Medicine, “Lucian Blaga” University of Sibiu, 550024 Sibiu, Romania;
- Department of Endocrinology, Clinical County Emergency Hospital, 550245 Sibiu, Romania
| | - Claudiu Nistor
- Department 4-Cardio-Thoracic Pathology, Thoracic Surgery II Discipline, “Carol Davila” University of Medicine and Pharmacy, 050474 Bucharest, Romania
- Thoracic Surgery Department, “Dr. Carol Davila” Central Military University Emergency Hospital, 010242 Bucharest, Romania
| | - Ioana Codruta Lebada
- Department of Endocrinology, Faculty of Medicine, “Lucian Blaga” University of Sibiu, 550024 Sibiu, Romania;
| | - Mara Carsote
- Department of Endocrinology, “Carol Davila” University of Medicine and Pharmacy, 020021 Bucharest, Romania;
- Department of Clinical Endocrinology V, “C.I. Parhon” National Institute of Endocrinology, 011863 Bucharest, Romania
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Mutlu V, Yuksek MA, Pekkolay Z, Yegin Z, Yildirim IH, Uslukaya O. Radiologist- and Surgeon-Performed Ultrasound (RSUS) Facilitates Minimally İnvasive Parathyroidectomy (MIP): Optimal Biochemical Parameters and Patient Outcomes. J Clin Med 2025; 14:2279. [PMID: 40217729 PMCID: PMC11989316 DOI: 10.3390/jcm14072279] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/12/2025] [Revised: 03/08/2025] [Accepted: 03/24/2025] [Indexed: 04/14/2025] Open
Abstract
Background/Objectives: The high success rate of minimally invasive parathyroidectomy (MIP) is dependent upon the correct preoperative localization of the solitary parathyroid adenoma (SPA). Various studies have focused on comparisons of radiologist-performed ultrasound (RUS) and surgeon-performed ultrasound (SUS) to increase the sensitivity rate of US. However, the efficiency of radiologist- and surgeon-performed ultrasound (RSUS) before MIP has not frequently been reported. We aimed to evaluate the efficiency of RSUS in clinical practice. Methods: In total, 122 patients (107 females, 15 males, mean age: 47.62 ± 15.75 years) with SPA were enrolled in our study design. The patients underwent preoperative ultrasonography (US) and technetium-99-sestamibi scintigraphy. Patient data including demographic characteristics, levels of biochemical parameters (parathyroid hormone (PTH), total serum calcium and phosphorus levels), operation time, and length of hospital stay were recorded. Results: MIP was performed with success under local anesthesia following the accurate localization of the adenomas by RSUS. The mean operation time was 20.00 ± 3.87 min. The mean preoperative serum PTH, calcium, and phosphorus levels were 525.69 ± 1050.92 pg/mL, 11.38 ± 1.22 mg/dL, and 2.53 ± 0.60 mg/dL, respectively. The decline in the perioperative PTH and calcium levels reflecting a cure was observed on the first postoperative day. Postoperative sixth month evaluations of the PTH and calcium levels confirmed the significant decrease, reflecting the therapeutic cure. Since no complications occurred, the hospital discharge process was carried out on the same day. Conclusions: RSUS is a beneficial adjunctive tool to facilitate MIP, and it achieved satisfactory therapeutic success in all the patients.
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Affiliation(s)
- Vahit Mutlu
- Department of General Surgery, Faculty of Medicine, Uskudar University, 34662 Istanbul, Turkey; (V.M.); (O.U.)
| | - Mahmut Arif Yuksek
- Department of General Surgery, Faculty of Medicine, Hitit University, 19040 Corum, Turkey;
| | - Zafer Pekkolay
- Department of Endocrinology, Faculty of Medicine, Dicle University, 21280 Diyarbakır, Turkey;
| | - Zeynep Yegin
- Medical Laboratory Techniques Program, Vocational School of Health Services, Sinop University, 57000 Sinop, Turkey
| | - Ibrahim Halil Yildirim
- Department of Genetics, Faculty of Veterinary, Dicle University, 21280 Diyarbakir, Turkey;
| | - Omer Uslukaya
- Department of General Surgery, Faculty of Medicine, Uskudar University, 34662 Istanbul, Turkey; (V.M.); (O.U.)
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Sharma A, Memon SS, Goroshi M, Goroshi S, Patil V, Badhe PV, Thakkar H, Sarathi V, Phadte A, Channaiah CY, Karlekar M, Barnabas R, Lila AR, Bandgar T. The polar vessel sign: insights from CT imaging analysis in Asian Indian primary hyperparathyroidism. Endocrine 2025; 87:800-809. [PMID: 39427108 DOI: 10.1007/s12020-024-04076-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/04/2024] [Accepted: 10/09/2024] [Indexed: 10/21/2024]
Abstract
PURPOSE Data on the polar vessel sign (enlarged feeding vessel terminating in parathyroid lesions) on four-dimensional computed tomography (4D-CT) is limited. We performed a retrospective analysis to determine the prevalence, predictors, and adjunctive utility of polar vessel sign in pre-operative 4D-CT of patients with primary hyperparathyroidism (PHPT). METHODS One radiologist blinded to the patients' details reported the 4D-CT of eighty-four operated patients with histopathology-proven single-gland PHPT. Two protocols were used to obtain arterial-phase images: timed via bolus tracking (n = 41) or fixed at 20 s after contrast injection (n = 43). RESULTS Seventy-one patients were symptomatic for PHPT, with median serum calcium 12.1 mg/dL. On the arterial phase of 4D-CT, 88.1% of lesions had the polar vessel sign, including 7/9 asymptomatic patients, 6/6 parathyroid carcinomas, and 3/4 ectopic(1:mediastinum, 2:thyro-thymic ligament). Predictors of polar vessel sign were maximum lesion dimension (2.2 vs. 1.4 cm; P = 0.03), solid-cystic CT morphology (47.3% vs. none; P = 0.004), and bolus tracking-timed arterial phase (55.4% vs. none; P = 0.001). Of these, bolus tracking improved the polar vessel's visualization (100% vs. 76.7%; P = 0.001) independent of lesion dimension and solid-cystic morphology. The latter two predicted polar vessel sign in images obtained at a fixed interval (20 s). A significantly lower proportion of bolus tracking-timed scans had lesion percentage arterial enhancement (PAE) < 128.9% (2/41 vs. 9/43; P = 0.04). Even with suboptimal PAE, the polar vessel helped identify 9/11 lesions. CONCLUSION The polar vessel sign demonstrated an additive role to PAE during CT reporting. Bolus tracking is valuable in optimizing vessel and tumor arterial enhancement and is easily incorporated into parathyroid 4D-CT protocol.
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Affiliation(s)
- Anima Sharma
- Department of Endocrinology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Saba Samad Memon
- Department of Endocrinology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Manjunath Goroshi
- Department of Endocrinology, Jawaharlal Nehru Medical College, Belagavi, Karnataka, 590010, India
| | - Shetteppa Goroshi
- Consultant Radiologist, Goroshi Scan and Diagnostics Center, Gokak, Karnataka, 591307, India
- Department of Radiodiagnosis, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Virendra Patil
- Department of Endocrinology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Padma Vikram Badhe
- Department of Radiodiagnosis, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Hemangini Thakkar
- Department of Radiodiagnosis, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Vijaya Sarathi
- Department of Endocrinology, Vydehi Institute of Medical Sciences and Research Center, Bengaluru, Karnataka, 560066, India
| | - Aditya Phadte
- Department of Endocrinology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Chethan Yami Channaiah
- Department of Endocrinology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Manjiri Karlekar
- Department of Endocrinology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Rohit Barnabas
- Department of Endocrinology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Anurag Ranjan Lila
- Department of Endocrinology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India
| | - Tushar Bandgar
- Department of Endocrinology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, Maharashtra, 400012, India.
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Tsoy U, Pogosian K, Ryzhkova D, Yudina O, Yakovenko K, Ryazanov P, Matsueva I, Sokolnikova P, Salov M, Karonova T, Grineva E. Somatostatin Receptor Imaging in the Diagnosis and Management of Parathyroid Neuroendocrine Neoplasia. Diagnostics (Basel) 2024; 14:2718. [PMID: 39682626 PMCID: PMC11640021 DOI: 10.3390/diagnostics14232718] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/16/2024] [Revised: 11/28/2024] [Accepted: 11/28/2024] [Indexed: 12/18/2024] Open
Abstract
BACKGROUND Parathyroid tumors are classified as parathyroid neuroendocrine neoplasia (NEN) by the IARC-WHO classification. These tumors can occur with NENs from other sites, which often require total-body [68Ga]-DOTA-peptides PET/CT. This study aimed to assess the utility of [68Ga]-DOTA-peptide PET/CT in imaging parathyroid NENs and to evaluate the underlying mechanisms. METHODS Fifty patients with primary hyperparathyroidism (PHPT) and parathyroid NENs histologically confirmed as parathyroid adenomas (PAs) were included. PET/CT with [68Ga]-DOTA-peptide was performed in 16 patients with localized PAs, including 10 with MEN1 syndrome. Somatostatin receptor types 2 and 5 (SST2 and SST5) staining was performed on PAs from 48 patients. Somatostatin analogs (SSA) were prescribed in four patients with MEN 1 syndrome and 1 with persistent acromegaly, all with PAs and PHPT. The therapy effects on calcium and parathyroid hormone (iPTH) were evaluated. RESULTS [68Ga]-DOTA-peptide PET/CT detected 20 PAs with high radiopharmaceutical uptake. SST2 expression was negative on PA cell membranes in all cases and positive on endothelium in 39 (81%) PAs. Membrane SST5 expression was positive in 25 (52%) PAs and endothelial was positive in 40 (83%). Serum calcium levels decreased in patients on SSA therapy, while iPTH did not. CONCLUSIONS PET/CT with [68Ga]-DOTA-peptides can detect parathyroid NENs. The incidental detection of high [68Ga]-DOTA-peptide uptake in the parathyroid region during whole-body PET/CT may prompt biochemical evaluation for PHPT. We suggest that endothelial SST expression mediates high radiopharmaceutical uptake by PAs and that SSA treatment can reduce hypercalcemia in PHPT patients.
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Affiliation(s)
| | - Karina Pogosian
- Almazov National Medical Research Centre, 2 Akkuratova Street, Saint Petersburg 197341, Russia; (U.T.)
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Gheorghe AM, Nistor C, Ranetti AE, Ciuche A, Ciobica ML, Stanciu M, Tanasescu D, Popa FL, Carsote M. Osteoporosis and Normocalcemic Primary Hyperparathyroidism (Conservatively or Surgically Managed). J Clin Med 2024; 13:6325. [PMID: 39518465 PMCID: PMC11545940 DOI: 10.3390/jcm13216325] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/27/2024] [Revised: 10/14/2024] [Accepted: 10/21/2024] [Indexed: 11/16/2024] Open
Abstract
Asymptomatic primary hyperparathyroidism (PHPT) involves 80-90% of the parathyroid tumor-associated cases of PHPT in the modern medical era, while normocalcemic PHPT (NPHPT) has a prevalence of 0.1-11%. We aimed to analyze the bone status and mineral metabolism in NPHPT amid conservative or surgical management. In this narrative review, we searched PubMed (between January 2020 and July 2024) via different keywords. Fourteen studies from the final analysis (388 patients with NPHPT; 1188 with PHPT; and 803 controls) showed that mean serum calcium levels varied between 2.57 and 2.26 mmol/L in NPHPT. Ten studies identified a similar 24 h urinary calcium in NPHPT versus hypercalcemic PHPT (HPHPT). Except for one study, a mandatory vitamin D analysis was performed, but the 25-hydroxyvitamin D cut-offs varied. Osteoporosis (n = 6 studies; N = 172 with NPHPT) was confirmed in 41.7-100% of NPHPT subjects. In surgery candidates, this rate might be overestimated. A DXA analysis was performed in eight studies (235 subjects with NPHPT, and 455 patients with HPHPT); two studies identified a lower BMD in HPHPT < NPHPT, but the results were not homogenous. A single study analyzed the TBS and found similar results in NPHPT. The prevalence of fractures (n = 9) varied between 7.4% and 42.8% in NPHPT. Bone turnover markers (N = 262 patients, n = 8 studies) showed lower bone formation markers in NPHPT versus PHPT (n = 3). Two studies analyzed the BMD and bone turnover markers following parathyroidectomy (161 patients, including 30 patients with NPHPT; mean ages over 60 years). To conclude, given the wide spectrum of complications associated with PHPT, an early diagnosis and proper management is essential. A more extensive screening in patients with osteoporosis and kidney stones might lead to the discovery of NPHPT, a more recently described form of PHPT. While it is still unclear whether NPHPT is an early stage of HPHPT or a separate entity, recent findings show similar osteoporosis and fracture occurrence, and an improvement in bone metabolism, following parathyroidectomy. More extensive prospective studies are crucial to understand the natural course of the disease, to reach a consensus regarding parathyroidectomy indications and surgery candidates' selection, and to ensure proper personalized management for these patients. With the evolving diagnosis methods, PHPT has become a condition with a changing clinical presentation, which now requires modern evaluation and treatment approaches.
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Affiliation(s)
- Ana-Maria Gheorghe
- PhD Doctoral School, “Carol Davila” University of Medicine and Pharmacy, 0505474 Bucharest, Romania;
- Department of Clinical Endocrinology V, “C.I. Parhon” National Institute of Endocrinology, 011863 Bucharest, Romania;
| | - Claudiu Nistor
- Department 4—Cardio-Thoracic Pathology, Thoracic Surgery II Discipline, “Carol Davila” University of Medicine and Pharmacy, 0505474 Bucharest, Romania
- Thoracic Surgery Department, “Dr. Carol Davila” Central Emergency University Military Hospital, 010825 Bucharest, Romania
| | - Aurelian-Emil Ranetti
- Department of Endocrinology, “Carol Davila” University of Medicine and Pharmacy, 020021 Bucharest, Romania
- Endocrinology Department, “Dr. Carol Davila” Central Emergency University Military Hospital, 010825 Bucharest, Romania
| | - Adrian Ciuche
- Department 4—Cardio-Thoracic Pathology, Thoracic Surgery II Discipline, “Carol Davila” University of Medicine and Pharmacy, 0505474 Bucharest, Romania
- Thoracic Surgery Department, “Dr. Carol Davila” Central Emergency University Military Hospital, 010825 Bucharest, Romania
| | - Mihai-Lucian Ciobica
- Department of Internal Medicine and Gastroenterology, “Carol Davila” University of Medicine and Pharmacy, 020021 Bucharest, Romania;
- Department of Internal Medicine I and Rheumatology, “Dr. Carol Davila” Central Military University Emergency Hospital, 010825 Bucharest, Romania
| | - Mihaela Stanciu
- Department of Endocrinology, Faculty of Medicine, “Lucian Blaga” University of Sibiu, 550024 Sibiu, Romania;
| | - Denisa Tanasescu
- Medical Clinical Department, Faculty of Medicine, “Lucian Blaga” University of Sibiu, 550169 Sibiu, Romania;
| | - Florina Ligia Popa
- Department of Physical Medicine and Rehabilitation, Faculty of Medicine, “Lucian Blaga” University of Sibiu, 550024 Sibiu, Romania;
| | - Mara Carsote
- Department of Clinical Endocrinology V, “C.I. Parhon” National Institute of Endocrinology, 011863 Bucharest, Romania;
- Department of Endocrinology, “Carol Davila” University of Medicine and Pharmacy, 020021 Bucharest, Romania
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Castellano E, Attanasio R, Borretta G. Letter Regarding: Modern Trends for Primary Hyperparathyroidism: Intervening on Less Biochemically Severe Disease. J Surg Res 2024; 302:975-976. [PMID: 38926061 DOI: 10.1016/j.jss.2024.04.039] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/05/2024] [Accepted: 04/10/2024] [Indexed: 06/28/2024]
Affiliation(s)
- Elena Castellano
- Department of Endocrinology, Diabetes and Metabolism, Santa Croce and Carle Hospital, Cuneo, Italy
| | - Roberto Attanasio
- Associazione Medici Endocrinologi, Scientific Committee, Milan, Italy
| | - Giorgio Borretta
- Department of Endocrinology, Diabetes and Metabolism, Santa Croce and Carle Hospital, Cuneo, Italy
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10
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Hargitai L, Boryshchuk D, Arikan M, Binter T, Scheuba C, Riss P. Is intraoperative parathyroid monitoring during minimally invasive parathyroidectomy still justified? Front Endocrinol (Lausanne) 2024; 15:1442972. [PMID: 39104811 PMCID: PMC11298376 DOI: 10.3389/fendo.2024.1442972] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/03/2024] [Accepted: 07/04/2024] [Indexed: 08/07/2024] Open
Abstract
Introduction Primary hyperparathyroidism (PHPT) is the third most common endocrine disease. With parathyroidectomy, a cure rate of over 95% at initial surgery is reported. Localization of the abnormal parathyroid gland is critical for the operation to be successful. The aim of this study is to analyze data of patients with single gland disease (SGD) and positive concordant localization imaging undergoing minimally invasive parathyroidectomy (MIP) and intraoperative parathyroid hormone monitoring (IOPTH) to evaluate if IOPTH is still justified in patients with localized SGD. Methods A retrospective database analysis of all minimally invasive operations with IOPTH for PHPT and positive concordant localization in ultrasound (US) and 99mTc-sestamibi scintigraphy (MIBI) between 2016-2021. When both US and MIBI were negative, patients underwent either choline or methionine PET-CT. The patients were also analyzed a second time without applying IOPTH. Results In total, 198 patients were included in the study. The sensitivity of US, MIBI and PET-CT was 96%, 94% and 100%, respectively. Positive predictive value was 88%, 89% and 94% with US, MIBI and PET-CT, respectively. IOPTH was true positive in 185 (93.4%) patients. In 13 (6.6%) patients, no adequate IOPTH decline was observed after localizing and extirpating the assumed enlarged parathyroid gland. Without IOPTH, the cure rate decreased from 195 (98.5%) to 182 (92%) patients and the rate of persisting disease increased from 2 (1.0%) to 15 (7.5%) patients. Conclusion Discontinuing IOPTH significantly increases the persistence rate by a factor of 7.5 in patients with concordantly localized adenoma. Therefore, IOPTH appears to remain necessary even for this group of patients.
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Affiliation(s)
- Lindsay Hargitai
- Department of General Surgery, Division of Visceral Surgery, Medical University Vienna, Vienna, Austria
| | - Daniela Boryshchuk
- Center for Medical Data Science, Institute of Medical Statistics, Medical University Vienna, Vienna, Austria
| | - Melisa Arikan
- Department of General Surgery, Division of Visceral Surgery, Medical University Vienna, Vienna, Austria
| | - Teresa Binter
- Department of General Surgery, Division of Visceral Surgery, Medical University Vienna, Vienna, Austria
| | - Christian Scheuba
- Department of General Surgery, Division of Visceral Surgery, Medical University Vienna, Vienna, Austria
| | - Philipp Riss
- Department of General Surgery, Division of Visceral Surgery, Medical University Vienna, Vienna, Austria
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Castellano E, Attanasio R, Boriano A, Gianotti L, Borretta G. Focus on Non-osteoporotic Post-menopausal Women with Asymptomatic Primary Hyperparathyroidism: A Monocentric Series. Endocr Metab Immune Disord Drug Targets 2024; 25:EMIDDT-EPUB-140228. [PMID: 38715334 PMCID: PMC11851140 DOI: 10.2174/0118715303288678240424074110] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/28/2023] [Revised: 03/04/2024] [Accepted: 03/07/2024] [Indexed: 02/27/2025]
Abstract
OBJECTIVE Primary Hyperparathyroidism (PHPT) is a common disease, frequently diagnosed in post-menopausal women, among whom Osteoporosis (OP) is a common finding. To date, no study has specifically evaluated the asymptomatic PHPT (aPHPT) patients without OP, in particular post-menopausal women who are exposed to an increased risk of developing OP. DESIGN This study involved a retrospective cross-sectional evaluation. PATIENTS From our database of 500 consecutive patients diagnosed with PHPT, 178 postmenopausal aPHPT were retrieved. RESULTS The clinical, biochemical, and imaging data of the 85 patients without OP were not different from those of the 93 with OP, except for bone alkaline phosphatase (significantly higher in the latter group). Among these 85 patients without OP, the 45 patients meeting surgical criteria for parathyroidectomy had significantly higher values of serum PTH (240 vs. 99 ng/L, p =0.03) and calcium (total, 11.2 ± 0.7 vs. 10.6 ± 0.4 mg/dL, p <0.001; ionized, 1.45 ± 0.12 vs. 1.36 ± 0.8 mmol/L, p =0.044) and lower values of serum phosphate (2.57 ± 0.7 vs. 2.94 ± 0.5 mg/dL, p =0.009) and eGFR (68.5 ± 23.8 vs 80.8 ± 14.4 mL/min/1.73 m2, p =0.006) than the 40 aPHPT patients not meeting surgical criteria, without any difference in densitometric data and calculated fracture risk. CONCLUSION In our series, post-menopausal aPHPT patients without OP accounted for almost a sixth of the whole PHPT series. About half of these patients did not meet surgical criteria, but their T scores and 10-year fracture risk calculated by FRAX were not significantly different from post-menopausal aPHPT without OP meeting surgical criteria.
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Affiliation(s)
- Elena Castellano
- Department of Endocrinology, Diabetes and Metabolism, Santa Croce and Carle Hospital, Cuneo, Italy
| | - Roberto Attanasio
- Scientific Committee, Associazione Medici Endocrinologi, Milano, Italy
| | - Alberto Boriano
- Department of Medical Physics, Santa Croce and Carle Hospital, Cuneo, Italy
| | - Laura Gianotti
- Department of Endocrinology, Diabetes and Metabolism, Santa Croce and Carle Hospital, Cuneo, Italy
| | - Giorgio Borretta
- Department of Endocrinology, Diabetes and Metabolism, Santa Croce and Carle Hospital, Cuneo, Italy
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12
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Szalat A, Shpitzen S, Pollack R, Mazeh H, Durst R, Meiner V. GCM2 p.Tyr394Ser variant in Ashkenazi Israeli patients with suspected familial isolated hyperparathyroidism. Front Endocrinol (Lausanne) 2023; 14:1254156. [PMID: 38130397 PMCID: PMC10733520 DOI: 10.3389/fendo.2023.1254156] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/06/2023] [Accepted: 11/20/2023] [Indexed: 12/23/2023] Open
Abstract
Context A germline mutation can be identified in up to 10% of patients with primary hyperparathyroidism (PHPT). In 2017, a high frequency of the GCM2 [(NM_ 004752.4) c.1181A> C; p.Tyr394Ser; rs142287570] variant was reported in PHPT Ashkenazi Jews (AJ). Objective To evaluate the presence of the GCM2 p.Tyr394Ser variant in Israeli patients addressed for genetic evaluation to characterize their phenotype and clinical management. Method Patients with PHPT who underwent addressed for genetic screening for suspected familial hypocalciuric hypercalcemia (FHH), a family history of isolated hyperparathyroidism (FIHP), or failed parathyroidectomy with persistent PHPT were recruited. Those with normal initial selected gene sequencing or hyperparathyroid genetic panel completed the GCM2 p.Tyr394Ser variant sequencing. The prevalence of this variant was evaluated using our local genomic database. Results A total of 42 single individuals from unrelated kindreds were evaluated. A disease-causing mutation was found in 11 (26.1%) patients: 10 were diagnosed with FHH (eight CASR and two AP2S1 mutations), and one patient had a CKN2B mutation. In 28 of the remaining patients, the GCM2 p.Tyr394Ser variant was positive in three (10.7%), and all were AJ. Within AJ (15/28, 53.5%), the rate of the p.Tyr394Ser variant was 3/15 (20%), and of those, two had a history of familial isolated hyperparathyroidism. Multi-glandular parathyroid adenoma/hyperplasia was also observed in two of these patients. No clinical or laboratory findings could discriminate patients with the GCM2 p.Tyr394Ser variant from those with FHH. Cinacalcet normalized the calcium levels in one patient. The prevalence of the GCM2 p.Tyr394Ser variant in 15,407 tests in our local genomic database was 0.98%. Conclusion In contrast to previous observations, the GCM2 p.Tyr394Ser variant-associated phenotype may be mild in AJ with FIHP, sometimes mimicking FHH. Because surgery may be curative, surgeons should be aware of the possibility of multiple gland diseases in these patients. The clinical spectrum and clinical utility of screening for this variant warrant further investigation.
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Affiliation(s)
- Auryan Szalat
- Endocrinology and Metabolism Service, Department of Internal Medicine, Osteoporosis Center, Hadassah-Hebrew University Medical Center, Jerusalem, Israel
| | - Shoshana Shpitzen
- Center for Research, Prevention and Treatment of Atherosclerosis, Hadassah-Hebrew University Medical Center, Jerusalem, Israel
| | - Rena Pollack
- Endocrinology and Metabolism Service, Department of Internal Medicine, Osteoporosis Center, Hadassah-Hebrew University Medical Center, Jerusalem, Israel
| | - Haggi Mazeh
- Department of Surgery, Hadassah-Hebrew University Medical Center, Jerusalem, Israel
| | - Ronen Durst
- Center for Research, Prevention and Treatment of Atherosclerosis, Hadassah-Hebrew University Medical Center, Jerusalem, Israel
| | - Vardiella Meiner
- Department of Genetics, Hadassah-Hebrew University Medical Center, Jerusalem, Israel
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13
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Hargitai L, Clerici T, Musholt TJ, Riss P. Surgery for primary hyperparathyroidism in Germany, Switzerland, and Austria: an analysis of data from the EUROCRINE registry. Langenbecks Arch Surg 2023; 408:150. [PMID: 37055669 PMCID: PMC10102096 DOI: 10.1007/s00423-023-02819-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/03/2022] [Accepted: 01/18/2023] [Indexed: 04/15/2023]
Abstract
PURPOSE EUROCRINE is an endocrine surgical register documenting diagnostic processes, indication for surgical treatment, surgical procedures, and outcomes. The purpose was to analyse data for PHPT in German speaking countries regarding differences in clinical presentation, diagnostic workup, and treatment. METHODS All operations for PHPT performed from 07/2015 to 12/2019 were analysed. RESULTS Three thousand two hundred ninety-one patients in Germany (9 centres; 1762 patients), Switzerland (16 centres; 971 patients) and Austria (5 centres; 558 patients) were analysed. Hereditary disease was seen in 36 patients in Germany, 16 patients in Switzerland and 8 patients in Austria. In sporadic disease before primary operation, PET-CT showed the highest sensitivity in all countries. In re-operations, CT and PET-CT achieved the highest sensitivities. The highest sensitivity of IOPTH was seen in Austria (98.1%), followed by Germany (96.4%) and Switzerland (91.3%). Operation methods and mean operative time reached statistical significance (p<0.05). Complication rates are low. Overall, 656 (19.9%) patients were asymptomatic; the remainder showed bone manifestations, kidney stones, fatigue and/or neuropsychiatric symptoms. CONCLUSION Early postoperative normocalcaemia ranged between 96.8 and 97.1%. Complication rates are low. PET-CT had the highest sensitivity in all three countries in patients undergoing primary operation as well as in Switzerland and Austria in patients undergoing re-operation. PET-CT could be considered a first-line preoperative imaging modality in patients with inconclusive ultrasound examination. The EUROCRINE registry is a beneficial and comprehensive data source for outcome analysis of endocrine procedures on a supranational level.
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Affiliation(s)
- L Hargitai
- Division of Visceral Surgery, Department of General Surgery, Medical University Vienna, Vienna, Austria
| | - T Clerici
- Department of Surgery, Cantonal Hospital St. Gallen, St. Gallen, Switzerland
| | - T J Musholt
- Department of General and Abdominal Surgery, University Medical Centre of the Johannes Gutenberg University Mainz, Mainz, Germany
| | - P Riss
- Division of Visceral Surgery, Department of General Surgery, Medical University Vienna, Vienna, Austria.
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14
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Bilezikian JP, Silverberg SJ, Bandeira F, Cetani F, Chandran M, Cusano NE, Ebeling PR, Formenti AM, Frost M, Gosnell J, Lewiecki EM, Singer FR, Gittoes N, Khan AA, Marcocci C, Rejnmark L, Ye Z, Guyatt G, Potts JT. Management of Primary Hyperparathyroidism. J Bone Miner Res 2022; 37:2391-2403. [PMID: 36054638 DOI: 10.1002/jbmr.4682] [Citation(s) in RCA: 32] [Impact Index Per Article: 10.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/11/2022] [Revised: 08/11/2022] [Accepted: 08/14/2022] [Indexed: 11/11/2022]
Abstract
Since the last international guidelines were published in 2014 on the evaluation and management of primary hyperparathyroidism (PHPT), new information has become available with regard to evaluation, diagnosis, epidemiology, genetics, classical and nonclassical manifestations, surgical and nonsurgical approaches, and natural history. To provide the most current summary of these developments, an international group, consisting of over 50 experts in these various aspects of PHPT, was convened. This paper provides the results of the task force that was assigned to review the information on the management of PHPT. For this task force on the management of PHPT, two questions were the subject of systematic reviews using the Grading of Recommendations, Assessment, Development and Evaluations (GRADE) methodology. The full report addressing surgical and nonsurgical management of PHPT, utilizing the GRADE methodology, is published separately in this series. In this report, we summarize the results of that methodological review and expand them to encompass a much larger body of new knowledge that did not specifically fit the criteria of the GRADE methodology. Together, both the systematic and narrative reviews of the literature, summarized in this paper, give the most complete information available to date. A panel of experts then considered the last set of international guidelines in light of the newer data and assessed the need for their revision. This report provides the evidentiary background to the guidelines report. In that report, evidence from all task forces is synthesized into a summary statement and revised guidelines for the evaluation and management of PHPT. © 2022 The Authors. Journal of Bone and Mineral Research published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research (ASBMR).
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Affiliation(s)
- John P Bilezikian
- Division of Endocrinology, Department of Medicine, Vagelos College of Physicians and Surgeons, Columbia University, New York, NY, USA
| | - Shonni J Silverberg
- Division of Endocrinology, Department of Medicine, Vagelos College of Physicians and Surgeons, Columbia University, New York, NY, USA
| | - Francisco Bandeira
- Division of Endocrinology, University of Pernambuco Medical School Recife, Recife, Brazil
| | | | - Manju Chandran
- Osteoporosis and Bone Metabolism Unit, Department of Endocrinology, Singapore General Hospital, Singapore, Singapore
| | - Natalie E Cusano
- Division of Endocrinology, Department of Medicine, Lenox Hill Hospital, New York, NY, USA
| | - Peter R Ebeling
- Department of Medicine, School of Clinical Sciences, Monash University, Clayton, Victoria, Australia
| | - Anna Maria Formenti
- Institute of Endocrine and Metabolic Sciences, San Raffaele Vita-Salute University and IRCCS Hospital, Milan, Italy
| | - Morten Frost
- Bone and Calcium Unit & Molecular Endocrinology Unit, Department of Endocrinology, Odense University Hospital, Odense, Denmark
| | - Jessica Gosnell
- Department of Surgery, University of California, San Francisco, CA, USA
| | - E Michael Lewiecki
- New Mexico Clinical Research & Osteoporosis Center, Albuquerque, NM, USA
| | - Frederick R Singer
- Endocrine/Bone Disease Program, Saint John's Cancer Institute, Santa Monica, CA, USA
| | - Neil Gittoes
- Centre for Endocrinology, Diabetes and Metabolism, Queen Elizabeth Hospital and University of Birmingham, Birmingham, UK
| | - Aliya A Khan
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada
| | - Claudio Marcocci
- Department of Clinical and Experimental Medicine, University of Pisa, Endocrine Unit 2, University Hospital of Pisa, Pisa, Italy
| | - Lars Rejnmark
- Department of Endocrinology and Internal Medicine, Aarhus University Hospital, Aarhus, Denmark
| | - Zhikang Ye
- Department of Health Research Methods, Evidence and Impact, McMaster University, Hamilton, ON, Canada
| | - Gordon Guyatt
- Department of Health Research Methods, Evidence and Impact, McMaster University, Hamilton, ON, Canada
| | - John T Potts
- Endocrine Unit, Department of Medicine, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA
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15
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Bilezikian JP, Khan AA, Silverberg SJ, Fuleihan GEH, Marcocci C, Minisola S, Perrier N, Sitges-Serra A, Thakker RV, Guyatt G, Mannstadt M, Potts JT, Clarke BL, Brandi ML, Balaya Z, Hofbauer L, Insogna K, Lacroix A, Liberman UA, Palermo A, Rizzoli R, Wermers R, Hannan FM, Pepe J, Cipriani C, Eastell R, Liu J, Mithal A, Moreira CA, Peacock M, Silva B, Walker M, Chakhtoura M, Schini M, Zein OE, Almquist M, Farias LCB, Duh Q, Lang BH, LiVolsi V, Swayk M, Vriens MR, Vu T, Yeh MW, Yeh R, Shariq O, Poch LL, Bandeira F, Cetani F, Chandran M, Cusano NE, Ebeling PR, Gosnell J, Lewiecki EM, Singer FR, Frost M, Formenti AM, Karonova T, Gittoes N, Rejnmark L. Evaluation and Management of Primary Hyperparathyroidism: Summary Statement and Guidelines from the Fifth International Workshop. J Bone Miner Res 2022; 37:2293-2314. [PMID: 36245251 DOI: 10.1002/jbmr.4677] [Citation(s) in RCA: 201] [Impact Index Per Article: 67.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/10/2022] [Revised: 08/11/2022] [Accepted: 08/14/2022] [Indexed: 11/10/2022]
Abstract
The last international guidelines on the evaluation and management of primary hyperparathyroidism (PHPT) were published in 2014. Research since that time has led to new insights into epidemiology, pathophysiology, diagnosis, measurements, genetics, outcomes, presentations, new imaging modalities, target and other organ systems, pregnancy, evaluation, and management. Advances in all these areas are demonstrated by the reference list in which the majority of listings were published after the last set of guidelines. It was thus, timely to convene an international group of over 50 experts to review these advances in our knowledge. Four Task Forces considered: 1. Epidemiology, Pathophysiology, and Genetics; 2. Classical and Nonclassical Features; 3. Surgical Aspects; and 4. Management. For Task Force 4 on the Management of PHPT, Grading of Recommendations, Assessment, Development, and Evaluations (GRADE) methodology addressed surgical management of asymptomatic PHPT and non-surgical medical management of PHPT. The findings of this systematic review that applied GRADE methods to randomized trials are published as part of this series. Task Force 4 also reviewed a much larger body of new knowledge from observations studies that did not specifically fit the criteria of GRADE methodology. The full reports of these 4 Task Forces immediately follow this summary statement. Distilling the essence of all deliberations of all Task Force reports and Methodological reviews, we offer, in this summary statement, evidence-based recommendations and guidelines for the evaluation and management of PHPT. Different from the conclusions of the last workshop, these deliberations have led to revisions of renal guidelines and more evidence for the other recommendations. The accompanying papers present an in-depth discussion of topics summarized in this report. © 2022 The Authors. Journal of Bone and Mineral Research published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research (ASBMR).
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Affiliation(s)
- John P Bilezikian
- Division of Endocrinology, Department of Medicine, Vagelos College of Physicians and Surgeons, Columbia University, New York, NY, USA
| | - Aliya A Khan
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada
| | - Shonni J Silverberg
- Division of Endocrinology, Department of Medicine, Vagelos College of Physicians and Surgeons, Columbia University, New York, NY, USA
| | - Ghada El-Hajj Fuleihan
- Calcium Metabolism and Osteoporosis Program, WHO CC for Metabolic Bone Disorders, Division of Endocrinology, American University of Beirut, Beirut, Lebanon
| | - Claudio Marcocci
- Department of Clinical and Experimental Medicine, University of Pisa, Endocrine Unit 2, University Hospital of Pisa, Pisa, Italy
| | - Salvatore Minisola
- Department of Clinical, Internal, Anaesthesiologic and Cardiovascular Sciences, 'Sapienza', Rome University, Rome, Italy
| | - Nancy Perrier
- Department of Surgical Oncology, University of Texas MD Anderson Cancer Center, Houston, Texas, USA
| | | | - Rajesh V Thakker
- Academic Endocrine Unit, Radcliffe Department of Medicine, University of Oxford, Oxford Centre for Diabetes, Endocrinology and Metabolism (OCDEM), Churchill Hospital, Oxford, UK
| | - Gordon Guyatt
- Department of Health Research Methods, Evidence and Impact, McMaster University, Hamilton, ON, Canada
| | - Michael Mannstadt
- Endocrine Unit, Department of Medicine, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA
| | - John T Potts
- Endocrine Unit, Department of Medicine, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA
| | - Bart L Clarke
- Mayo Clinic Division of Endocrinology, Diabetes, Metabolism, and Nutrition, Mayo Clinic, Rochester, MN, USA
| | - Maria Luisa Brandi
- Fondazione Italiana sulla Ricerca sulle Malattie dell'Osso (F.I.R.M.O. Foundation), Florence, Italy
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Hargitai L, Schefner M, Traub-Weidinger T, Haug A, Arikan M, Scheuba C, Riss P. Accessing the influence of 99mTc-Sesta-MIBI-positive thyroid nodules on preoperative localisation studies in patients with primary hyperparathyroidism. Langenbecks Arch Surg 2022; 407:1183-1191. [PMID: 35061094 PMCID: PMC9151570 DOI: 10.1007/s00423-022-02442-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/28/2021] [Accepted: 01/13/2022] [Indexed: 11/26/2022]
Abstract
Purpose Curative treatment for primary hyperparathyroidism (PHPT) is parathyroidectomy (PTX) with removal of the hyperfunctioning gland(s). In an endemic goitre region, 35–78% of PHPT patients show concomitant thyroid disease. This study aimed to evaluate if 99mTc-sestamibi (MIBI)-positive thyroid nodules decrease sensitivity in regard to localising the hyperfunctioning parathyroid gland(s) in PHPT patients. Methods Within 5 years, 497 consecutive patients with biochemically proven PHPT were included in this study. The data was analysed retrospectively. Results In total, 198 patients underwent PTX with thyroid surgery and 299 patients underwent sole PTX. Sensitivity of MIBI scan for PTX with and without thyroid surgery was 72.1% and 73.6%, respectively. A statistically significant difference in sensitivity of ultrasound for PTX with and without thyroid surgery (57.0% and 70.9%, respectively) was observed (p = 0.029). Thyroid nodule histology did not have a significant effect on the MIBI scan. Unilateral neck exploration (UNE) was performed in 110 patients and bilateral neck exploration (BNE) in 177 patients. The probability of surgical conversion from UNE to BNE due to incorrect localisation was 1.733 times higher in patients with thyroid nodules. Conclusions Concomitant benign thyroid nodules did not influence MIBI sensitivity. No correlation between thyroid carcinoma and MIBI uptake was determined. However, MIBI detection of thyroid malignancy is important in patients initially being considered for minimal invasive parathyroidectomy. Sensitivity and positive predictive value of ultrasound were significantly lower in patients with thyroid nodules. The probability of conversion from UNE to BNE due to incorrect localisation was 1.733 times higher in patients with thyroid nodules.
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Affiliation(s)
- Lindsay Hargitai
- Endocrine Surgery, Division of Visceral Surgery, Department of General Surgery, Medical University of Vienna, Waehringer Guertel 18-20, Vienna, Austria.
| | - Maria Schefner
- Endocrine Surgery, Division of Visceral Surgery, Department of General Surgery, Medical University of Vienna, Waehringer Guertel 18-20, Vienna, Austria
| | - Tatjana Traub-Weidinger
- Division of Nuclear Medicine, Department of Biomedical Imaging and Image-Guided Therapy, Medical University of Vienna, Waehringer Guertel 18-20, Vienna, Austria
| | - Alexander Haug
- Division of Nuclear Medicine, Department of Biomedical Imaging and Image-Guided Therapy, Medical University of Vienna, Waehringer Guertel 18-20, Vienna, Austria
| | - Melisa Arikan
- Endocrine Surgery, Division of Visceral Surgery, Department of General Surgery, Medical University of Vienna, Waehringer Guertel 18-20, Vienna, Austria
| | - Christian Scheuba
- Endocrine Surgery, Division of Visceral Surgery, Department of General Surgery, Medical University of Vienna, Waehringer Guertel 18-20, Vienna, Austria
| | - Philipp Riss
- Endocrine Surgery, Division of Visceral Surgery, Department of General Surgery, Medical University of Vienna, Waehringer Guertel 18-20, Vienna, Austria
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17
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Silva BC, Bilezikian JP. Skeletal abnormalities in Hypoparathyroidism and in Primary Hyperparathyroidism. Rev Endocr Metab Disord 2021; 22:789-802. [PMID: 33200346 DOI: 10.1007/s11154-020-09614-0] [Citation(s) in RCA: 31] [Impact Index Per Article: 7.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 11/11/2020] [Indexed: 12/22/2022]
Abstract
Both hypoparathyroidism (HypoPT), as well as its pathological counterpart, primary hyperparathyroidism (PHPT), can lead to skeletal abnormalities. Chronic deficiency of PTH in patients with HypoPT is associated with a profound reduction in bone remodeling, with consequent increases in bone density, and abnormalities in microarchitecture and bone strength. It is still not clear whether there is an increase in fracture risk in HypoPT. While standard therapy with calcium supplements and active vitamin D does not restore bone homeostasis, treatment of HypoPT with PTH appears to correct some of those abnormalities. In PHPT, the continuous exposure to high levels of PTH causes an increase in bone remodeling, in which bone resorption prevails. In the symptomatic form of PHPT, patients can present with fragility fractures, and/or the classical radiological features of osteitis fibrosa cystica. However, even in mild PHPT, catabolic skeletal actions of PTH are evident through reduced BMD, deterioration of bone microarchitecture and increased risk of fragility fractures. Successful parathyroidectomy improves skeletal abnormalities. Medical treatment, such as bisphosphonates and denosumab, can also increase bone density in patients with PHPT who do not undergo surgery. This article reviews skeletal involvement in HypoPT and in PHPT, as assessed by bone remodeling, DXA, trabecular bone score, and quantitative computed tomography, as well as data on bone strength and fracture risk. The effects of PTH replacement on the skeleton in subjects with HypoPT, and the outcome of parathyroidectomy in patients with PHPT, are also reviewed here.
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Affiliation(s)
- Barbara C Silva
- Department of Medicine, Centro Universitario de Belo Horizonte - UNI BH, Belo Horizonte, Brazil
- Endocrinology Unit, Felicio Rocho Hospital, Belo Horizonte, Brazil
- Endocrinology Unit, Santa Casa Hospital, Belo Horizonte, Brazil
| | - John P Bilezikian
- Metabolic Bone Diseases Unit, Division of Endocrinology, Department of Medicine, Vagelos College of Physicians and Surgeons, Columbia University, 630 W. 168th Street, PH 8E: 105G, New York, NY, 10032, USA.
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18
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Muñoz de Nova JL, Sampedro-Nuñez M, Huguet-Moreno I, Marazuela Azpiroz M. A practical approach to normocalcemic primary hyperparathyroidism. Endocrine 2021; 74:235-244. [PMID: 34386939 DOI: 10.1007/s12020-021-02845-4] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/13/2021] [Accepted: 08/01/2021] [Indexed: 10/20/2022]
Abstract
Normocalcemic primary hyperparathyroidism is an entity on which several issues about its clinical management remains unclear. This is reflected in the main current guidelines, where there are no evidence-based specific recommendations. Through an exhaustive review of current literature, a clinical management algorithm for these patients is proposed. We consider the diagnosis criteria, the differential diagnosis, the clinical manifestations, and the treatment indications. When indicated, we also review the preoperative locations techniques and the surgical approach. Finally, when surgical treatment is not indicated, the patient is not a candidate to surgery or refuse surgical management, we review the medical treatment options and the follow-up schemes.
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Affiliation(s)
- José Luis Muñoz de Nova
- Department of General and Digestive Surgery, Hospital Universitario de la Princesa; Instituto de Investigación Sanitaria Princesa (ISS-IP); Universidad Autónoma de Madrid, Madrid, Spain
| | - Miguel Sampedro-Nuñez
- Department of Endocrinology and Nutrition, Hospital Universitario de la Princesa; Instituto de Investigación Sanitaria Princesa (ISS-IP); Universidad Autónoma de Madrid, Madrid, Spain
| | - Isabel Huguet-Moreno
- Department of Endocrinology and Nutrition, Hospital Universitario Infanta Leonor, Madrid, Spain
| | - Mónica Marazuela Azpiroz
- Department of Endocrinology and Nutrition, Hospital Universitario de la Princesa; Instituto de Investigación Sanitaria Princesa (ISS-IP); Universidad Autónoma de Madrid, Madrid, Spain.
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19
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Hendricks A, Lenschow C, Kroiss M, Buck A, Kickuth R, Germer CT, Schlegel N. Evaluation of diagnostic efficacy for localization of parathyroid adenoma in patients with primary hyperparathyroidism undergoing repeat surgery. Langenbecks Arch Surg 2021; 406:1615-1624. [PMID: 33993327 PMCID: PMC8370933 DOI: 10.1007/s00423-021-02191-z] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/30/2020] [Accepted: 05/04/2021] [Indexed: 11/05/2022]
Abstract
PURPOSE Repeat surgery in patients with primary hyperparathyroidism (pHPT) is associated with an increased risk of complications and failure. This stresses the need for optimized strategies to accurately localize a parathyroid adenoma before repeat surgery is performed. However, evidence on the extent of required diagnostics for a structured approach is sparse. METHODS A retrospective single-center evaluation of 28 patients with an indication for surgery due to pHPT and previous thyroid or parathyroid surgery was performed. Diagnostic workup, surgical approach, and outcome in terms of complications and successful removement of parathyroid adenoma with biochemical cure were evaluated. RESULTS Neck ultrasound, sestamibi scintigraphy, C11-methionine PET-CT, and selective parathyroid hormone venous sampling, but not MRI imaging, effectively detected the presence of a parathyroid adenoma with high positive predictive values. Biochemical cure was revealed by normalization of calcium and parathormone levels 24-48h after surgery and was achieved in 26/28 patients (92.9%) with an overall low rate of complications. Concordant localization by at least two diagnostic modalities enabled focused surgery with success rates of 100%, whereas inconclusive localization significantly increased the rate of bilateral explorations and significantly reduced the rate of biochemical cure to 80%. CONCLUSION These findings suggest that two concordant diagnostic modalities are sufficient to accurately localize parathyroid adenoma before repeat surgery for pHPT. In cases of poor localization, extended diagnostic procedures are warranted to enhance surgical success rates. We suggest an algorithm for better orientation when repeat surgery is intended in patients with pHPT.
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Affiliation(s)
- Anne Hendricks
- Department of General, Visceral, Vascular and Pediatric Surgery, University Hospital Würzburg, Oberduerrbacherstrasse 6, 97080, Wuerzburg, Germany
| | - Christina Lenschow
- Department of General, Visceral, Vascular and Pediatric Surgery, University Hospital Würzburg, Oberduerrbacherstrasse 6, 97080, Wuerzburg, Germany
| | - Matthias Kroiss
- Department for Diabetology and Endocrinology, Clinic for Internal Medicine I, University Hospital Wuerzburg, Oberduerrbacherstrasse 6, 97080, Wuerzburg, Germany
| | - Andreas Buck
- Clinic for Nuclear Medicine, University Hospital Wuerzburg, Oberduerrbacherstrasse 6, 97080, Wuerzburg, Germany
| | - Ralph Kickuth
- Institute for Diagnostic and Interventional Radiology, University Hospital Wuerzburg, Oberduerrbacherstrasse 6, 97080, Wuerzburg, Germany
| | - Christoph-Thomas Germer
- Department of General, Visceral, Vascular and Pediatric Surgery, University Hospital Würzburg, Oberduerrbacherstrasse 6, 97080, Wuerzburg, Germany
| | - Nicolas Schlegel
- Department of General, Visceral, Vascular and Pediatric Surgery, University Hospital Würzburg, Oberduerrbacherstrasse 6, 97080, Wuerzburg, Germany.
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20
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Nouikes Zitouni S. Monocentric experience of primary hyperparathyroidism surgery in Algeria. Surg Open Sci 2021; 4:32-36. [PMID: 33748732 PMCID: PMC7966862 DOI: 10.1016/j.sopen.2021.02.002] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/20/2020] [Revised: 01/25/2021] [Accepted: 02/05/2021] [Indexed: 11/24/2022] Open
Abstract
Introduction Primary hyperparathyroidism (PHPT) remains a relatively underdiagnosed disease in developing countries. The aim of this study was to assess the demographic, pathological, biochemical, and surgical characteristics of patients with primary hyperparathyroidism in a university hospital department of otolaryngology in eastern Algeria. Materials and method We performed a retrospective analysis of the records of 62 patients operated in our department for primary hyperparathyroidism between January 2002 and December 2013. Results The mean age was 47.7 ± 15 years with a female preponderance (88.7%). The mode of discovery was during a biological assessment for bone syndrome in 42% of cases. The mean calcemia was 2.92 ± 0.6 mmol/L, and the intact serum parathyroid hormone was 867.78 ± 954.50 pmol/L. A total of 54.8% of patients had bilateral neck exploration, and 45.2% had minimally invasive open parathyroidectomy. Postoperative complications were dominated by severe hypocalcemia and hungry bone syndrome. Conclusion The diagnosis of primary hyperparathyroidism in our country is late, and management is often performed after the appearance of bone and renal complications.
Primary hyperparathyroidism is an underdiagnosed disease in developing countries. The diagnosis is made after the appearance of bone and renal complications. Bone manifestations are the most common presenting symptoms. Biochemical profile is dominated by hypercalcemia and elevated serum parathyroid hormone levels. Average adenoma weight is very high, correlating with severe hyperparathyroidism. Hypocalcemia and hungry bone syndrome are the most common postoperative complications.
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Affiliation(s)
- Souad Nouikes Zitouni
- Faculty of Medicine, Badji Mokhtar Annaba University, Boulevard Mohamed Seddik Benyahia, Annaba, Algeria.,Department of Otolaryngology, Annaba University Hospital, Ibn Rochd, Seraidi Rd, Annaba, Algeria
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21
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Juhlin CC, Falhammar H, Zedenius J, Nilsson IL, Höög A. Lipoadenoma of the Parathyroid Gland: Characterization of an Institutional Series Spanning 28 Years. Endocr Pathol 2020; 31:156-165. [PMID: 32193823 PMCID: PMC7250804 DOI: 10.1007/s12022-020-09616-3] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/16/2022]
Abstract
Parathyroid lipoadenomas (PLAs) are rare tumors, and case descriptions are limited, < 80 to date. As a consequence, scarce information regarding the etiology of these enigmatic lesions is available. We searched our departmental pathology database for PLAs diagnosed between 1992 and 2020, reexamined the histology of each case, and recorded clinical parameters from the patients' medical charts. As the diagnostic criteria of this lesion vary over this time period, we defined PLA as an enlarged parathyroid gland with > 50% fat on histologic examination with preoperative evidence of primary hyperparathyroidism (PHPT). A total of 8 bona fide PLA cases and 2 equivocal PLAs (close to 50% fat) were included. As approximately 4000 conventional parathyroid adenomas were diagnosed at our department during the same time interval, the prevalence of PLA was 0.20%. PLA patients were predominately female (63%) and presented with classical PHPT-related symptoms. Majority of cases were successfully located preoperatively and had an average tumor weight of 445 mg. Histologically, all PLAs consisted of > 50% mature adipose tissue, except a single case with brown fat. Of note, PLA patients exhibited a body mass index in line with PHPT patients in general, but a relatively high, near-significant prevalence of arterial hypertension was observed when compared to tumors with less fat (P = 0.0584). Future studies on this finding might be warranted. To summarize, we present one of the largest institutional PLA case series to date, and conclude that PLAs are rare, sporadic tumors mirroring many clinical aspects of conventional adenomas-with a potential coupling to hypertension worthy of follow-up studies.
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Affiliation(s)
- C Christofer Juhlin
- Department of Pathology and Cytology, Karolinska University Hospital, Stockholm, Sweden.
- Department of Oncology-Pathology, Karolinska Institutet, BioClinicum J6:20, 171 64, Solna, Stockholm, Sweden.
| | - Henrik Falhammar
- Department of Endocrinology, Metabolism and Diabetes, Karolinska University Hospital, Stockholm, Sweden
- Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
| | - Jan Zedenius
- Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
- Department of Breast, Endocrine Tumors and Sarcoma, Karolinska University Hospital, Stockholm, Sweden
| | - Inga-Lena Nilsson
- Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
- Department of Breast, Endocrine Tumors and Sarcoma, Karolinska University Hospital, Stockholm, Sweden
| | - Anders Höög
- Department of Pathology and Cytology, Karolinska University Hospital, Stockholm, Sweden
- Department of Oncology-Pathology, Karolinska Institutet, BioClinicum J6:20, 171 64, Solna, Stockholm, Sweden
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22
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Nachtigall LB, Guarda FJ, Lines KE, Ghajar A, Dichtel L, Mumbach G, Zhao W, Zhang X, Tritos NA, Swearingen B, Miller KK, Thakker RV. Clinical MEN-1 Among a Large Cohort of Patients With Acromegaly. J Clin Endocrinol Metab 2020; 105:dgaa142. [PMID: 32311048 PMCID: PMC7180000 DOI: 10.1210/clinem/dgaa142] [Citation(s) in RCA: 18] [Impact Index Per Article: 3.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/27/2020] [Accepted: 04/01/2020] [Indexed: 02/06/2023]
Abstract
CONTEXT Clinical multiple endocrine neoplasia type 1 (MEN-1) is diagnosed by the presence of at least 2 MEN-1-associated tumors. Many patients with acromegaly and clinical MEN-1 yield negative testing for MEN1 mutations. While cases of acromegaly and primary hyperparathyroidism (PHP) with negative genetic testing have been reported, its prevalence among patients with acromegaly is undetermined, and the clinical presentation has not been well characterized. OBJECTIVES The main goals of this study are: (1) To determine the prevalence of clinical MEN-1 with PHP in patients with acromegaly and characterize their clinical features; and (2) to evaluate the genetic basis for the coexistence of acromegaly and PHP. DESIGN Retrospective record review and genetic analysis. SETTING Clinical Research Centers. PARTICIPANTS 414 patients with acromegaly. INTERVENTIONS Clinical evaluation and DNA sequencing for MEN1, CDKN1A, CDKN1B, CDKN2B, CDKN2C, and AIP genes. MAIN OUTCOME MEASUREMENTS Clinical and genetic analysis. RESULTS Among patients with acromegaly, clinical MEN-1, as defined by the presence of at least one other MEN-1-associated tumor, was present in 6.6%. PHP occurred in 6.1%; more than half had parathyroid hyperplasia. DNA sequencing was unrevealing for genetic mutations, except for 1 case of a CDC73 mutation. Acromegaly was diagnosed at an older age with a higher prevalence of malignancies (specifically breast and thyroid) in patients with coexisting PHP than those with isolated acromegaly. CONCLUSIONS A distinct phenotype is described in patients with clinical MEN-1 and negative genetic testing for mutations previously associated with this syndrome. Further studies are needed to identify other genes that may explain the association between PHP and acromegaly.
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Affiliation(s)
- Lisa B Nachtigall
- Neuroendocrine Unit, Massachusetts General Hospital and Department of Medicine, Harvard Medical School, Boston, Massachusetts
| | - Francisco J Guarda
- Neuroendocrine Unit, Massachusetts General Hospital and Department of Medicine, Harvard Medical School, Boston, Massachusetts
- Endocrinology Department and Center for Translational Endocrinology (CETREN), School of Medicine, Pontificia Universidad Católica de Chile, Santiago, Chile
| | - Kate E Lines
- Academic Endocrine Unit, OCDEM, Radcliffe Department of Medicine, University of Oxford, Churchill Hospital, Oxford, UK
| | - Alireza Ghajar
- Neuroendocrine Unit, Massachusetts General Hospital and Department of Medicine, Harvard Medical School, Boston, Massachusetts
| | - Laura Dichtel
- Neuroendocrine Unit, Massachusetts General Hospital and Department of Medicine, Harvard Medical School, Boston, Massachusetts
| | - Giselle Mumbach
- Neuroendocrine Unit, Massachusetts General Hospital and Department of Medicine, Harvard Medical School, Boston, Massachusetts
| | - Wenxiu Zhao
- Neuroendocrine Unit, Massachusetts General Hospital and Department of Medicine, Harvard Medical School, Boston, Massachusetts
| | - Xun Zhang
- Neuroendocrine Unit, Massachusetts General Hospital and Department of Medicine, Harvard Medical School, Boston, Massachusetts
| | - Nicholas A Tritos
- Neuroendocrine Unit, Massachusetts General Hospital and Department of Medicine, Harvard Medical School, Boston, Massachusetts
| | - Brooke Swearingen
- Neuroendocrine Unit, Massachusetts General Hospital and Department of Medicine, Harvard Medical School, Boston, Massachusetts
| | - Karen K Miller
- Neuroendocrine Unit, Massachusetts General Hospital and Department of Medicine, Harvard Medical School, Boston, Massachusetts
| | - Rajesh V Thakker
- Academic Endocrine Unit, OCDEM, Radcliffe Department of Medicine, University of Oxford, Churchill Hospital, Oxford, UK
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