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Khan S, Khan AA. Hypoparathyroidism: diagnosis, management and emerging therapies. Nat Rev Endocrinol 2025; 21:360-374. [PMID: 39905273 DOI: 10.1038/s41574-024-01075-8] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 11/29/2024] [Indexed: 02/06/2025]
Abstract
Hypoparathyroidism is characterized by inadequate parathyroid hormone (PTH) secretion or action and results in hypocalcaemia, and can lead to hyperphosphataemia and hypercalciuria. Most cases of hypoparathyroidism occur as a complication of surgery, with the remainder due to causes including autoimmune disease, genetic causes, infiltrative diseases, mineral deposition or due to abnormalities in serum levels of magnesium. Hypoparathyroidism can cause multisystem disease, with long-term complications resulting from ectopic calcification as well as renal complications with nephrocalcinosis, nephrolithiasis and renal impairment in addition to respiratory, cardiac or neurological manifestations. Conventional therapy consists of oral calcium salts and active vitamin D but it has limitations, including fluctuations in serum levels of calcium and a high pill burden, and can increase the risk of long-term complications. By contrast, PTH replacement therapy can effectively achieve normal serum levels of calcium, and lower serum levels of phosphate. The long-acting PTH analogue, palopegteriparatide, has been shown to normalize urine levels of calcium. In addition, PTH replacement therapy reduces the pill burden. Palopegteriparatide is also associated with improved quality of life in comparison to conventional therapy. This Review summarizes current recommendations regarding the pathophysiology, evaluation and management of hypoparathyroidism and also references the 2022 international hypoparathyroidism guidelines. Palopegteriparatide has now been approved as PTH replacement therapy for hypoparathyroidism. Emerging therapies will also be presented in this Review.
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Affiliation(s)
- Sarah Khan
- Trillium Health Partners, University of Toronto, Toronto, Ontario, Canada
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2
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Díez JJ. New therapeutic options in the management of chronic hypoparathyroidism. ENDOCRINOL DIAB NUTR 2025:101532. [PMID: 40368704 DOI: 10.1016/j.endien.2025.101532] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/18/2024] [Accepted: 11/22/2024] [Indexed: 05/16/2025]
Affiliation(s)
- Juan J Díez
- Servicio de Endocrinología y Nutrición, Hospital Universitario Puerta de Hierro Majadahonda, Madrid, Spain; Instituto de Investigación Sanitaria Puerta de Hierro Segovia de Arana, Majadahonda, Madrid, Spain; Departamento de Medicina, Universidad Autónoma de Madrid, Madrid, Spain.
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di Filippo L, Allora A, Formenti AM, Ferrari F, Villanova M, Russo A, Presciuttini B, Locatelli M, Brandi ML, Vezzoli G, Marelli C, Giustina A. Obesity and blunted FGF23 response associate with kidney impairment in patients with hypoparathyroidism. Endocrine 2025:10.1007/s12020-025-04221-y. [PMID: 40237977 DOI: 10.1007/s12020-025-04221-y] [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: 01/12/2025] [Accepted: 03/23/2025] [Indexed: 04/18/2025]
Abstract
PURPOSE Chronic kidney disease (CKD) is a common complication of patients affected by chronic hypoparathyroidism managed with conventional therapies. However, no data are currently available regarding the endocrine and metabolic determinants of renal function in these patients. METHODS This was a multicenter observational study performed in three health-care centres. Patients with hypoparathyroidism were consecutively enrolled during follow-up visits in 2022-2023. These exclusion criteria were adopted: patients managed with dialysis, proteinuria (>200 mg/24 h), use of antihypertensive drugs including thiazides, ACE-inhibitors, angiotensin-II-receptor antagonists, alpha-beta blocking-agents, aldosterone-antagonists, and insulin-treated diabetes. RESULTS A total of 46 patients were enrolled. Median age was 53 years, 34 (74%) were female and the median disease duration was 11 years. In all patients, the calcium-phosphate product was within the normal range. The 23.7% of patients was obese (BMI ≥ 30) and CKD (defined with an eGFR < 60 mL/min1.73m2) was found in the 21.7% of patients. Patients with CKD were older, affected by a longer-disease, more frequently obese and with higher BMI. In multivariate analyses, obesity resulted as the only significant independent risk factor associated with CKD. In addition, a significant negative correlation was found between BMI and eGFR, and ROC analyses showed a significant global-performances of BMI to predict CKD. Patients with CKD were characterized also by higher FGF23 levels. A significant negative correlation was found between FGF23 and eGFR, however, evaluating separately those with and without CKD, this correlation remained significant only in the second group. CONCLUSIONS For the first-time, obesity was demonstrated to be independently associated with CKD in patients with hypoparathyroidism, and a blunted eGFR-related response of FGF23 was shown in patients with CKD potentially worsening the renal function in the context of hypoparathyroidism.
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Affiliation(s)
- Luigi di Filippo
- Institute of Endocrine and Metabolic Sciences, Università Vita-Salute San Raffaele, IRCCS Ospedale San Raffaele, Milan, Italy
| | - Agnese Allora
- Institute of Endocrine and Metabolic Sciences, Università Vita-Salute San Raffaele, IRCCS Ospedale San Raffaele, Milan, Italy
| | - Anna Maria Formenti
- Institute of Endocrine and Metabolic Sciences, Università Vita-Salute San Raffaele, IRCCS Ospedale San Raffaele, Milan, Italy
| | - Francesca Ferrari
- Institute of Endocrine and Metabolic Sciences, Università Vita-Salute San Raffaele, IRCCS Ospedale San Raffaele, Milan, Italy
| | - Marta Villanova
- Institute of Endocrine and Metabolic Sciences, Università Vita-Salute San Raffaele, IRCCS Ospedale San Raffaele, Milan, Italy
| | - Antonino Russo
- Internal Medicine Division, Santa Chiara Hospital, Provincial Health Care Agency (APSS), Trento, Italy
| | | | - Massimo Locatelli
- Laboratory Medicine Service, IRCCS Ospedale San Raffaele, Milan, Italy
| | - Maria Luisa Brandi
- Institute of Endocrine and Metabolic Sciences, Università Vita-Salute San Raffaele, IRCCS Ospedale San Raffaele, Milan, Italy
| | - Giuseppe Vezzoli
- Unit of Nephrology and Dialysis, Università Vita-Salute San Raffaele, IRCCS Ospedale San Raffaele, Milan, Italy
| | | | - Andrea Giustina
- Institute of Endocrine and Metabolic Sciences, Università Vita-Salute San Raffaele, IRCCS Ospedale San Raffaele, Milan, Italy.
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Trummer C, Blaschke M, Quint D, Schulz M, Herrmann-Lingen C, Büttner M, Siggelkow H. Normative values for the hypoparathyroidism patient questionnaire (HPQ28) in the German general population. J Patient Rep Outcomes 2025; 9:38. [PMID: 40178746 PMCID: PMC11968587 DOI: 10.1186/s41687-025-00868-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/20/2024] [Accepted: 03/19/2025] [Indexed: 04/05/2025] Open
Abstract
BACKGROUND Patients with hypoparathyroidism (HypoPT) suffer from several complaints and reduced quality of life (QoL), even if disease-specific biochemical parameters are within the target range. To be able to quantify symptoms in HypoPT patients, we recently developed a disease-specific questionnaire, the Hypoparathyroidism Patient Questionnaire with 28 items (HPQ28). The aim of this study was to find normative values for the HPQ28 in the German general population. METHODS We tasked an independent market and social research institute to obtain sociodemographic data and HPQ28 results from a representative sample of the German general population. The HPQ28 comprises five scales and three single items. The five scales indicate different areas of complaints: Pain and cramps (PaC) including five items, neurovegetative symptoms (NVS) including five items, loss of vitality (LoV) including six items, depression and anxiety (DaA) including five items, gastro-intestinal symptoms (GiS) including two items and two control items for depression. Three items were not attributable to any of the five scales: numbness and tingling in certain parts of the body (NT), troubled memory (TM), and racing heart (RH). RESULTS Mean age (± standard deviation) in the representative general population sample (n = 2506) was 49.5 ± 17.8 years, 51% were female. All scales and single items were affected by gender with women presenting significantly more complaints on every scale and single item in comparison to men (p < 0.01, Mann-Whitney U test). In addition, all scales and single items, except for GiS, were affected by age in males and females (p < 0.001, Spearman's correlation). Regression analyses proved a linear trend in the different scores regarding age and gender (p < 0.05 except for age on the GiS scale). CONCLUSIONS We present data from the first application of the HPQ28 in a representative sample of the German general population. Almost all scales and single item of the HPQ28 were dependent on age and gender, with older individuals and females presenting a higher burden of complaints. TRIAL REGISTRATION DRKS, DRKS00027581. Registered 17th of January 2022, https//drks.de/search/de/trial/DRKS00027581.
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Affiliation(s)
- Christian Trummer
- Clinic of Gastroenterology, Gastrointestinal Oncology and Endocrinology, University Medical Center Göttingen, Robert-Koch-Straße 40, 37075, Göttingen, Germany
- Division of Endocrinology and Diabetology, Department of Internal Medicine, Medical University of Graz, Auenbruggerplatz 15, Graz, 8036, Austria
| | - Martina Blaschke
- Clinic of Gastroenterology, Gastrointestinal Oncology and Endocrinology, University Medical Center Göttingen, Robert-Koch-Straße 40, 37075, Göttingen, Germany
- MVZ Endokrinologikum Göttingen, Von-Siebold-Straße 3, 37075, Göttingen, Germany
| | - Deborah Quint
- Clinic of Gastroenterology, Gastrointestinal Oncology and Endocrinology, University Medical Center Göttingen, Robert-Koch-Straße 40, 37075, Göttingen, Germany
| | - Maxi Schulz
- Institute for Medical Statistics, University Medical Center Göttingen, Humboldtallee 32, 37073, Göttingen, Germany
| | - Christoph Herrmann-Lingen
- Clinic for Psychosomatic Medicine and Psychotherapy, University Medical Center Göttingen, Von-Siebold-Straße 5, 37075, Göttingen, Germany
| | - Matthias Büttner
- Institute of Medical Biostatistics, Epidemiology and Informatics (IMBEI), University Medical Center Mainz, Obere Zahlbacherstraße 69, 55131, Mainz, Germany
- University Cancer Centre, Langenbeckstraße 1, 55131, Mainz, Germany
| | - Heide Siggelkow
- MVZ Endokrinologikum Göttingen, Von-Siebold-Straße 3, 37075, Göttingen, Germany.
- Department of Trauma, Orthopedics and Reconstructive Surgery, University Medical Center Göttingen, Robert-Koch-Straße 40, 37075, Götingen, Germany.
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Khan AA, AbuAlrob H, Ali DS, Al Kassem Z, Almoulia A, Afifi H, Braga M, Cheng A, Malhem J, Millar A, Morgante E, Muhammad P, Paul TL, Prebtani A, Punthakee Z, Khan T, Khan S, Shrayyef M, Van Uum S, Young JEM, Brandi ML, Ovize M, Weiss B. Skeletal health status among patients with chronic hypoparathyroidism: results from the Canadian National Hypoparathyroidism Registry (CNHR). Osteoporos Int 2025; 36:673-684. [PMID: 39955688 PMCID: PMC12064612 DOI: 10.1007/s00198-025-07410-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/13/2024] [Accepted: 01/24/2025] [Indexed: 02/17/2025]
Abstract
In the CNHR study, 35% of postmenopausal women had osteoporosis by BMD or fragility fracture, and 4% had both. Three men ≥ 50 had osteoporosis by BMD or fragility fracture (33.3%; n = 3/9). This suggests that close follow-up of skeletal health is necessary in postmenopausal women, and men ≥ 50 with chronic HypoPT. PURPOSE Chronic hypoparathyroidism (HypoPT) has been associated with decreased bone turnover and abnormalities in bone mineral density (BMD), microarchitecture, and strength. Current guidelines do not recommend systematic evaluation of skeletal health in patients with chronic HypoPT. Our study assessed skeletal health in pre- and postmenopausal women with chronic HypoPT and adult men. METHODS This prospective study enrolled adults with chronic HypoPT from the Canadian National Hypoparathyroidism Registry. Clinical characteristics, bone fractures, biochemistry, and serum bone biomarkers were assessed at baseline. Skeletal health evaluation included assessments of fragility fractures, BMD at lumbar spine (LS), femoral neck (FN), total hip (TH), 1/3 radial sites, trabecular bone score (TBS), and bone biomarkers. RESULTS We present the baseline data of the patients enrolled in the registry. We analyzed a total of 101 patients: 18 men, 35 premenopausal, and 48 postmenopausal women. The mean (SD) age at the onset of HypoPT was 40.7 (16.8) years, and the average disease duration was 11.2 (8.6) years. The most common etiology was postsurgical (74.3% vs. 25.7% non-surgical). Most patients received calcium supplements (89%) and active vitamin D (80%) at baseline. No fragility fractures or low BMD were reported in premenopausal women. However, BMD at LS, FN, TH, and TBS were significantly lower in postmenopausal compared to premenopausal women. CONCLUSIONS Overall, 35% of postmenopausal women had osteoporosis by BMD or prior fragility fracture, and 4% had both. Three men ≥ 50 years had osteoporosis by BMD or fragility fracture (33.3%; n = 3/9). This study suggests that close follow-up of skeletal health is necessary in postmenopausal women with chronic HypoPT and men ≥ 50 years.
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Affiliation(s)
- Aliya A Khan
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada.
- Bone Research and Education Centre, Oakville, Canada.
- Divisions of Endocrinology and Metabolism, McMaster University, 3075 Suite #223 Hospital Gate, Oakville, ON, L6M 1M1, Canada.
| | - Hajar AbuAlrob
- Department of Health Research Methodology, McMaster University, Hamilton, ON, Canada
| | - Dalal S Ali
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada
| | | | | | - Habiba Afifi
- Bone Research and Education Centre, Oakville, Canada
| | - Manoela Braga
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada
| | - Alice Cheng
- Department of Medicine, University of Toronto, Toronto, ON, Canada
| | - Jouma Malhem
- Bone Research and Education Centre, Oakville, Canada
| | - Adam Millar
- The Division of Endocrinology and Metabolism, University of Toronto, Toronto, ON, Canada
| | | | | | | | - Ally Prebtani
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada
| | - Zubin Punthakee
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada
| | | | - Sarah Khan
- Bone Research and Education Centre, Oakville, Canada
| | | | | | - James Edward Massey Young
- Division of Otolaryngology-Head and Neck SurgeryDepartment of Surgery, McMaster University, Hamilton, ON, Canada
| | - Maria Luisa Brandi
- F.I.R.M.O. Onlus Italian Foundation for the Research On Bone Diseases, Florence, Italy
- Donatello Bone Clinic, Villa Donatello Hospital, Florence, Italy
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Carney P, Cutler GB, Schneider K, Zhang F, DiMarchi R. MBX 2109, A Once-Weekly Parathyroid Hormone Replacement Therapy Prodrug: Phase 1, First-In-Human, Randomized Trial. J Clin Endocrinol Metab 2025; 110:940-950. [PMID: 39574220 PMCID: PMC11913096 DOI: 10.1210/clinem/dgae808] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/06/2024] [Revised: 09/06/2024] [Accepted: 11/20/2024] [Indexed: 01/14/2025]
Abstract
CONTEXT Hypoparathyroidism denotes parathyroid hormone (PTH) deficiency and impaired mineral metabolism. MBX 2109, a novel prodrug yielding a biologically active PTH peptide agonist (PTH[1-32], extended by a fatty acylated Lys33), is being developed as a long-acting, once-weekly PTH replacement therapy. OBJECTIVE Here, we report the safety, pharmacokinetics (PK), and pharmacodynamics (PD) of MBX 2109 in healthy volunteers. METHODS This phase 1, randomized, double-blind, placebo-controlled, multiple ascending-dose study (NCT05158335) enrolled healthy adults, who were randomly assigned 4:1 to receive MBX 2109 (200, 400, 600, and 900 μg; n = 8) or placebo (n = 2) by subcutaneous administration once weekly for 4 doses (days 1, 8, 15, and 22). The primary end point was safety and tolerability. Key secondary end points were PK and PD. RESULTS Overall, 40 participants (MBX 2109 n = 32, placebo n = 8) were randomly assigned (mean age, 43.3 years; 22.5% female). Treatment-emergent adverse events (TEAEs) occurred in 50% to 88% of MBX 2109 groups and in 25% of placebo participants. In the MBX 2109 groups, no severe or serious TEAEs were observed. Injection-site reaction was the most common treatment-related TEAE. The half-lives were 79 to 95 hours for MBX 2109 and 184 to 213 hours for the fatty-acylated biologically active PTH peptide, which showed dose- and time-dependent exposure increases. CONCLUSION The sustained-action PTH prodrug MBX 2109 was well tolerated with no unexpected, off-target safety issues. The long half-life and flat exposure profile of MBX 2109's biologically active PTH agonist supports once-weekly administration. MBX 2109 doses were identified for future studies.
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Affiliation(s)
- Patricia Carney
- Clinical Operations, MBX Biosciences Inc, Carmel, IN 46032, USA
| | | | - Kristi Schneider
- Clinical Development, MBX Biosciences Inc, Carmel, IN 46032, USA
| | - Fa Zhang
- Department of Chemistry, Indiana University, Bloomington, IN 47405, USA
| | - Richard DiMarchi
- Department of Chemistry, Indiana University, Bloomington, IN 47405, USA
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Yang YY, Deng YH, Sun LH, Rejnmark L, Wang L, Pietschmann P, Glüer CC, A Khan A, Minisola S, Liu JM. Hypoparathyroidism: Similarities and differences between Western and Eastern countries. Osteoporos Int 2025; 36:391-402. [PMID: 39777494 DOI: 10.1007/s00198-024-07352-6] [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: 06/05/2024] [Accepted: 12/11/2024] [Indexed: 01/11/2025]
Abstract
BACKGROUD Hypoparathyroidism (hypoPT) is characterized by acute and chronic complications due to insufficient parathyroid hormone (PTH) production or action. Several management guidelines have been developed, but mostly based on evidence from Western countries. Data from Eastern countries have not been systematically compared with those from Western countries. METHODS Literatures regarding to the epidemiology, genetics, risk factors, clinical manifestations and therapies for hypoPT in Easten and Western countries, including China, South Korea, Japan, India, and USA, Canada, Italy, and etc., were searched through PubMed and CNKI. This review was officially endorsed by European Calcified Tissue Society (ECTS) board. RESULTS Postoperative hypoPT is the major form of hypoPT in both Western and Eastern countries. The genetic profiles and clinical features of hypoPT are similar in Eastern and Western countries. The most commonly used medications in Eastern countries are calcium and native vitamin D or active vitamin D analogues, similar to their Western counterparts. While PTH replacement therapy is not available and approved to use in most Eastern countries. CONCLUSION Physicians and surgeons should follow the guidelines on the management of thyroid nodules, taking more care of protecting parathyroid glands during surgery. The cross-talk between East and West in the management of hypoPT should be continued. Direct comparisons of the management strategies in patients with hypoPT between Eastern and Wester countries regarding to the morbidity, mortality, quality of life, optimal dosage, efficacies and side-effects of conventional therapies or newer medications, as well as pharmacogenetics and pharmacoeconomics, would be valuable.
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Affiliation(s)
- Yu-Ying Yang
- Department of Endocrine and Metabolic Diseases, Shanghai Institute of Endocrine and Metabolic Diseases, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
- National Clinical Research Center for Metabolic Diseases (Shanghai), Key Laboratory for Endocrine and Metabolic Diseases of the National Health Commission of the PR China, Shanghai National Center for Translational Medicine, Shanghai, China
| | - Yan-Hua Deng
- Department of Endocrine and Metabolic Diseases, Shanghai Institute of Endocrine and Metabolic Diseases, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
- National Clinical Research Center for Metabolic Diseases (Shanghai), Key Laboratory for Endocrine and Metabolic Diseases of the National Health Commission of the PR China, Shanghai National Center for Translational Medicine, Shanghai, China
| | - Li-Hao Sun
- Department of Endocrine and Metabolic Diseases, Shanghai Institute of Endocrine and Metabolic Diseases, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
- National Clinical Research Center for Metabolic Diseases (Shanghai), Key Laboratory for Endocrine and Metabolic Diseases of the National Health Commission of the PR China, Shanghai National Center for Translational Medicine, Shanghai, China
| | - Lars Rejnmark
- Department of Endocrinology and Internal Medicine, Aarhus University Hospital, 8200, Aarhus N, Denmark
| | - Ling Wang
- Department of Radiology, Beijing Jishuitan Hospital, Capital Medical University, Beijing, China
| | - Peter Pietschmann
- Division of Cellular and Molecular Pathophysiology, Department of Pathophysiology and Allergy Research, Center for Pathophysiology, Infectiology and Immunology, Medical University of Vienna, Waehringer Guertel 18-20, 1090, Vienna, Austria
| | - Claus-Christian Glüer
- Section Biomedical Imaging, Department of Radiology and Neuroradiology, North Competence Center, University Medical Center Schleswig-Holstein Kiel, Kiel University, Molecular Imaging, Kiel, Germany
| | - Aliya A Khan
- Divisions of Endocrinology and Metabolism and Geriatrics, McMaster University, Hamilton, ON, L8S 4L8, Canada.
| | - Salvatore Minisola
- Department of Clinical, Internal, Anesthesiological and Cardiovascular Sciences, Sapienza University of Rome, 00161, Rome, Italy.
| | - Jian-Min Liu
- Department of Endocrine and Metabolic Diseases, Shanghai Institute of Endocrine and Metabolic Diseases, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China.
- National Clinical Research Center for Metabolic Diseases (Shanghai), Key Laboratory for Endocrine and Metabolic Diseases of the National Health Commission of the PR China, Shanghai National Center for Translational Medicine, Shanghai, China.
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Zhao Q, Wang L, Xiang H, Qiu L. Reducing early pregnancy loss with vitamin D 3: an analysis of serum 1,25-(OH)D 3 modulation and miscarriage risk. J Clin Biochem Nutr 2025; 76:164-178. [PMID: 40151409 PMCID: PMC11936736 DOI: 10.3164/jcbn.24-147] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/28/2024] [Accepted: 09/11/2024] [Indexed: 03/29/2025] Open
Abstract
This case-control study of 124 early-pregnant women found that daily supplementation with 400 IU of vitamin D3 significantly increased serum 1,25-(OH)D3 levels and was associated with a lower incidence of threatened miscarriage. The study suggests that vitamin D3 supplementation could reduce the risk of early miscarriage and improve pregnancy outcomes by modulating immune responses and hormonal stability.
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Affiliation(s)
- Qian Zhao
- Department of Obstetrics and Gynecology in Hangzhou First People’s Hospital Tonglu Hospital in Zhejiang Province, Hangzhou, No. 899 Meilin Road, Chengnan Street, Tonglu, Hangzhou 311500, Zhejiang Province, China
| | - Liangying Wang
- Department of Obstetrics and Gynecology in Hangzhou First People’s Hospital Tonglu Hospital in Zhejiang Province, Hangzhou, No. 899 Meilin Road, Chengnan Street, Tonglu, Hangzhou 311500, Zhejiang Province, China
| | - Hongqin Xiang
- Department of Obstetrics and Gynecology in Hangzhou First People’s Hospital Tonglu Hospital in Zhejiang Province, Hangzhou, No. 899 Meilin Road, Chengnan Street, Tonglu, Hangzhou 311500, Zhejiang Province, China
| | - Luling Qiu
- Department of Obstetrics and Gynecology in Hangzhou First People’s Hospital Tonglu Hospital in Zhejiang Province, Hangzhou, No. 899 Meilin Road, Chengnan Street, Tonglu, Hangzhou 311500, Zhejiang Province, China
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Palermo A, Naciu AM, Donovan YKT, Tabacco G, Zavatta G. PTH Substitution Therapy for Chronic Hypoparathyroidism: PTH 1-84 and Palopegteriparatide. Curr Osteoporos Rep 2025; 23:12. [PMID: 39987371 DOI: 10.1007/s11914-025-00905-6] [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] [Accepted: 02/12/2025] [Indexed: 02/24/2025]
Abstract
PURPOSE OF REVIEW to describe and compare the efficacy and safety of the main PTH treatments, namely PTH(1-84) and palopegteriparatide, for the management of hypoparathyroidism. RECENT FINDINGS neither PTH (1-84) nor PTH(1-34) have been shown a clear and consistent favorable impact on the 24 h urinary calcium excretion normalization, while the positive effect on quality of life is still debated. Recently, the Food & Drug Administration and the European Medicines Agency approved palopegteriparatide as the first true replacement therapy for hypoPT management. Palopegteriparatide is a prodrug of PTH(1-34), administered once daily, and designed to provide continuous exposure to released PTH over a 24-h dosing period. According to phase II and phase III studies, palopegteriparatide seems to fill the gaps identified in existing therapies for hypoPT. Palopegteriparatide is the first real replacement therapy for the management of hypoparathyroidism and seems to fill the gaps identified in existing therapies for hypoPT.
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Affiliation(s)
- Andrea Palermo
- Unit of Metabolic Bone and Thyroid Disorders, Fondazione Policlinico Universitario Campus Bio-Medico, Rome, Italy.
- Unit of Endocrinology and Diabetes, Campus Bio-Medico University, Rome, Italy.
| | - Anda Mihaela Naciu
- Unit of Metabolic Bone and Thyroid Disorders, Fondazione Policlinico Universitario Campus Bio-Medico, Rome, Italy
| | - Yu Kwang Tay Donovan
- Arden Diabetes and Endocrine Clinic, Royal Square Medical Centre, 101 Irrawaddy Road, #10-08, Novena, S329565, Singapore
| | - Gaia Tabacco
- Unit of Metabolic Bone and Thyroid Disorders, Fondazione Policlinico Universitario Campus Bio-Medico, Rome, Italy
- Department for the Promotion of Human Science and Quality of Life, San Raffaele Open University, Rome, Italy
| | - Guido Zavatta
- Department of Medical and Surgical Sciences, University of Bologna, Bologna, Italy
- Division of Endocrinology and Diabetes Prevention and Care, IRCSS Azienda Ospedaliero-Universitaria Di Bologna, Bologna, Italy
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Arkfeld CK, Mannstadt M, Mahalingaiah S. Hypoparathyroidism and assisted reproductive technology: considerations while undergoing ovarian hyperstimulation. Curr Opin Endocrinol Diabetes Obes 2024; 31:197-202. [PMID: 39287990 DOI: 10.1097/med.0000000000000887] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 09/19/2024]
Abstract
PURPOSE OF REVIEW Hypoparathyroidism (hypoPTH) is a rare disease that requires diligent adherence to treatment regimens to prevent hypocalcemia but also treatment-induced hypercalcemia and hypercalciuria. The menstrual cycle, pregnancy, and lactation can all impact calcium homeostasis but there is little known regarding the impact of ovarian stimulation. Furthermore, the limited reports suggest no clear association between menstrual phase and calcium balance among those with hypoPTH. With increasing patient utilization of assisted reproductive technology (ART), there is a need for better understanding the care required for patients with hypoparathyroidism pursuing fertility technology. RECENT FINDINGS There is currently no literature available on patients with hypoparathyroidism and the impact of controlled ovarian stimulation on calcium homeostasis. We present information regarding physiologic changes in pregnancy that impact calcium homeostasis and the first case presentation of a patient with hypoparathyroidism pursuing ART. SUMMARY This article provides the first insights and guidance when providing fertility care for patients with hypoparathyroidism.
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Affiliation(s)
- Christopher K Arkfeld
- Massachusetts General Hospital, Harvard Medical School, Division of Reproductive Endocrinology and Infertility
| | - Michael Mannstadt
- Massachusetts General Hospital, Harvard Medical School, Division of Endocrinology
| | - Shruthi Mahalingaiah
- Massachusetts General Hospital, Harvard Medical School, Division of Reproductive Endocrinology and Infertility
- Harvard T.H. Chan School of Public Health, Boston, Massachusetts, USA
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11
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Kuusela E, Kostiainen I, Ritvonen E, Ryhänen EM, Schalin-Jäntti C. Bone mineral density over ten years after primary parathyroidectomy in multiple endocrine neoplasia type 1. JBMR Plus 2024; 8:ziae129. [PMID: 39575107 PMCID: PMC11579653 DOI: 10.1093/jbmrpl/ziae129] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/14/2024] [Revised: 10/01/2024] [Accepted: 10/20/2024] [Indexed: 11/24/2024] Open
Abstract
Primary hyperparathyroidism (PHPT) associated with multiple endocrine neoplasia type 1 (MEN1) impairs bone mineral density and causes osteoporosis already in young patients. We aimed to investigate bone mineral density (BMD) in a contemporary cohort of patients with MEN1-related PHPT after long-term follow-up and compare these results with that of healthy controls. Thirty-five patients with genetically confirmed MEN1 were diagnosed with MEN1 at mean age 28.7 ± 13.6 years. Thirty-two (91.4%) underwent primary parathyroidectomy at mean age 33.3 ± 13.7 years; 12 had undergone at least 2 surgeries with on average 7.3 ± 5.9 years between the operations. BMD was assessed by DXA at the end of mean follow-up, 13.2 years after the primary parathyroidectomy and compared with that of 35 age- and gender-matched controls. More than 10 years after the first parathyroidectomy, mean BMD in patients with MEN1 is in the normal range. However, it is still significantly lower compared with healthy controls.
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Affiliation(s)
- Emma Kuusela
- Endocrinology, Abdominal Center, Helsinki University Hospital and University of Helsinki, ENDO-ERN (European Reference Network on Rare Endocrine Conditions), FIN-00290 Helsinki, Finland
| | - Iiro Kostiainen
- Endocrinology, Abdominal Center, Helsinki University Hospital and University of Helsinki, ENDO-ERN (European Reference Network on Rare Endocrine Conditions), FIN-00290 Helsinki, Finland
| | - Elina Ritvonen
- Endocrinology, Abdominal Center, Helsinki University Hospital and University of Helsinki, ENDO-ERN (European Reference Network on Rare Endocrine Conditions), FIN-00290 Helsinki, Finland
- Heart and Lung Center, Helsinki University Hospital and Helsinki University, FIN-00290 Helsinki, Finland
| | - Eeva M Ryhänen
- Endocrinology, Abdominal Center, Helsinki University Hospital and University of Helsinki, ENDO-ERN (European Reference Network on Rare Endocrine Conditions), FIN-00290 Helsinki, Finland
| | - Camilla Schalin-Jäntti
- Endocrinology, Abdominal Center, Helsinki University Hospital and University of Helsinki, ENDO-ERN (European Reference Network on Rare Endocrine Conditions), FIN-00290 Helsinki, Finland
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12
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Yang M, Pu SL, Li L, Ma Y, Qin Q, Wang YX, Huang WL, Hu HY, Zhu MF, Li CZ. Hypoparathyroidism with situs inversus totalis: A case report. World J Radiol 2024; 16:561-568. [PMID: 39494145 PMCID: PMC11525822 DOI: 10.4329/wjr.v16.i10.561] [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: 05/05/2024] [Revised: 08/31/2024] [Accepted: 09/19/2024] [Indexed: 10/28/2024] Open
Abstract
BACKGROUND Hypoparathyroidism (HP) is a rare endocrine disorder, while situs inversus totalis (SIT) is a rare condition in which the internal organs are positioned in a mirrored pattern compared to their usual positions. This case illustrates some potential shared mechanisms between HP and SIT, highlighting the importance of accurate identification and prompt first emergency, offering insights for future research. CASE SUMMARY This report discusses a case of a middle-aged patient with adolescent-onset HP with concurrent SIT. The patient experienced recurrent episodes of increased neuromuscular excitability (manifesting as spasms in the hands and feet and laryngospasms) and even periods of unconsciousness. Initially, these symptoms led to a misdiagnosis of epilepsy. Nevertheless, upon thorough examination and treatment in the general medicine ward, the correct diagnosis was established. Corresponding treatment resulted in improved management of the patient's symptoms. CONCLUSION Co-occurrence of HP and SIT may be associated with genetic mutations, chromosomal anomalies, or hereditary factors, as may other similar conditions.
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Affiliation(s)
- Mao Yang
- Department of General Medicine, First People’s Hospital of Zunyi (Third Affiliated Hospital of Zunyi Medical University), Zunyi 563000, Guizhou Province, China
| | - Sheng-Lan Pu
- Department of General Medicine, First People’s Hospital of Zunyi (Third Affiliated Hospital of Zunyi Medical University), Zunyi 563000, Guizhou Province, China
| | - Ling Li
- Department of General Medicine, First People’s Hospital of Zunyi (Third Affiliated Hospital of Zunyi Medical University), Zunyi 563000, Guizhou Province, China
| | - Yu Ma
- Department of Nutrition, First People’s Hospital of Zunyi (Third Affiliated Hospital of Zunyi Medical University), Zunyi 563000, Guizhou Province, China
| | - Qin Qin
- Department of General Medicine, First People’s Hospital of Zunyi (Third Affiliated Hospital of Zunyi Medical University), Zunyi 563000, Guizhou Province, China
| | - Yan-Xia Wang
- Department of General Medicine, First People’s Hospital of Zunyi (Third Affiliated Hospital of Zunyi Medical University), Zunyi 563000, Guizhou Province, China
| | - Wen-Long Huang
- Department of General Medicine, First People’s Hospital of Zunyi (Third Affiliated Hospital of Zunyi Medical University), Zunyi 563000, Guizhou Province, China
| | - Hong-Ya Hu
- Department of General Medicine, First People’s Hospital of Zunyi (Third Affiliated Hospital of Zunyi Medical University), Zunyi 563000, Guizhou Province, China
| | - Mei-Feng Zhu
- Department of General Medicine, First People’s Hospital of Zunyi (Third Affiliated Hospital of Zunyi Medical University), Zunyi 563000, Guizhou Province, China
| | - Chun-Zhu Li
- Department of General Medicine, First People’s Hospital of Zunyi (Third Affiliated Hospital of Zunyi Medical University), Zunyi 563000, Guizhou Province, China
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13
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Díez JJ, Anda E, Pérez-Corral B, Paja M, Alcazar V, Sánchez-Ragnarsson C, Orois A, Romero-Lluch AR, Sambo M, Oleaga A, Caballero Á, Alhambra MR, Urquijo V, Delgado-Lucio AM, Fernández-García JC, Doulatram-Gamgaram VK, Dueñas-Disotuar S, Martín T, Peinado M, Sastre J. Prevalence of chronic postsurgical hypoparathyroidism not adequately controlled: an analysis of a nationwide cohort of 337 patients. Front Endocrinol (Lausanne) 2024; 15:1464515. [PMID: 39387052 PMCID: PMC11461294 DOI: 10.3389/fendo.2024.1464515] [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: 07/14/2024] [Accepted: 09/05/2024] [Indexed: 10/12/2024] Open
Abstract
Purpose The identification of patients with chronic hypoparathyroidism who are adequately (AC) or not adequately controlled (NAC) has clinical interest, since poor disease control is related to complications and mortality. We aimed to assess the prevalence of NAC patients in a cohort of subjects with postsurgical hypoparathyroidism. Methods We performed a multicenter, retrospective, cohort study including patients from 16 Spanish hospitals with chronic hypoparathyroidism lasting ≥3 years. We analyzed disease control including biochemical profile and clinical wellness. For biochemical assessment we considered three criteria: criterion 1, normal serum calcium, phosphorus and calcium x phosphorus product; criterion 2, the above plus estimated glomerular filtration rate ≥60 ml/min/1.73 m2; and criterion 3, the above plus normal 24-hour urinary calcium excretion. A patient was considered AC if he or she met the biochemical criteria and was clinically well. Results We included 337 patients with postsurgical hypoparathyroidism (84.3% women, median age 45[36-56] years, median time of follow-up 8.9[6.0-13.0] years). The proportions of NAC patients with criteria 1, 2 and 3 were, respectively, 45.9%, 49.2% and 63.1%. Patients who had dyslipidemia at the time of diagnosis presented a significantly higher risk of NAC disease (criterion 3; OR 7.05[1.44-34.45]; P=0.016). NAC patients (criterion 2) had a higher proportion of subjects with incident chronic kidney disease and eye disorders, and NAC patients (criterion 3) had a higher proportion of incident chronic kidney disease, nephrolithiasis and dyslipidemia than AC patients. Conclusion The present study shows a strikingly high prevalence of NAC patients in the clinical practice of Spanish endocrinologists. Results suggest that NAC disease might be associated with some prevalent and incident comorbidities.
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Affiliation(s)
- Juan J. Díez
- Department of Endocrinology, Hospital Universitario Puerta de Hierro Majadahonda, Instituto de Investigación Sanitaria Puerta de Hierro Segovia de Arana, Majadahonda, Spain
- Department of Medicine, Universidad Autónoma de Madrid, Madrid, Spain
| | - Emma Anda
- Department of Endocrinology, Hospital Universitario de Navarra, Pamplona, Spain
| | - Begoña Pérez-Corral
- Department of Endocrinology, Complejo Asistencial Universitario de León, León, Spain
| | - Miguel Paja
- Department of Endocrinology, Hospital Universitario de Basurto, Universidad del País Vasco, Universidad del País Vasco (UPV)/EHU, Bilbao, Spain
| | - Victoria Alcazar
- Department of Endocrinology, Hospital Severo Ochoa, Leganés, Spain
| | - Cecilia Sánchez-Ragnarsson
- Department of Endocrinology, Hospital Universitario Central de Asturias, Instituto de Investigación Sanitaria del Principado de Asturias, Oviedo, Spain
| | - Aida Orois
- Department of Endocrinology and Nutrition, Hospital Clínic, Barcelona, Spain
| | - Ana R. Romero-Lluch
- Department of Endocrinology, Hospital Universitario Virgen del Rocío, Sevilla, Spain
| | - Marcel Sambo
- Department of Endocrinology, Hospital General Universitario Gregorio Marañón, Madrid, Spain
| | - Amelia Oleaga
- Department of Endocrinology, Hospital Universitario de Basurto, Universidad del País Vasco, Universidad del País Vasco (UPV)/EHU, Bilbao, Spain
| | - Águeda Caballero
- Department of Endocrinology, Hospital Universitario de Canarias, Tenerife, Spain
| | - María R. Alhambra
- Department of Endocrinology, Hospital Universitario Reina Sofía, Córdoba, Spain
| | - Virginia Urquijo
- Department of Endocrinology, Hospital Universitario de Cruces, Bilbao, Spain
| | | | - José C. Fernández-García
- Department of Endocrinology, Hospital Regional Universitario de Málaga, Instituto de Investigación Biomédica de Málaga, Universidad de Málaga, Málaga, Spain
| | - Viyey K. Doulatram-Gamgaram
- Department of Endocrinology, Hospital Regional Universitario de Málaga, Instituto de Investigación Biomédica de Málaga, Universidad de Málaga, Málaga, Spain
| | - Suset Dueñas-Disotuar
- Department of Endocrinology, Hospital Universitario Virgen del Rocío, Sevilla, Spain
| | - Tomás Martín
- Department of Endocrinology, Hospital Universitario Virgen Macarena, Sevilla, Spain
| | - Mercedes Peinado
- Department of Endocrinology, Hospital Universitario Virgen Macarena, Sevilla, Spain
| | - Julia Sastre
- Department of Endocrinology, Hospital Universitario de Toledo, Toledo, Spain
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14
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Santa Ritta Barreira CE, Kowalski LP, Dias FL, Farias TPDE, Santos IC, Vartanian JG, Beltrão AMF, Feitosa H, Neto RG, Oliveira AF, Ribeiro R, Vanderlei FAB, Duarte FHG, Filho PEP, Pinheiro RN. Guideline From the Brazilian Society of Surgical Oncology and Brazilian College of Surgeons in Preventing and Managing Acute Hypoparathyroidism After Thyroid Surgery. J Surg Oncol 2024; 130:705-713. [PMID: 39315485 DOI: 10.1002/jso.27910] [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: 08/16/2024] [Accepted: 08/18/2024] [Indexed: 09/25/2024]
Abstract
The risk of hypoparathyroidism and hypocalcemia is a critical concern in thyroid surgery. Preserving parathyroid gland vascularization during surgery is essential for effective prevention. Preoperative and postoperative management, including calcium and Vitamin D supplementation, is paramount. Measurement of parathyroid hormone levels after surgery is the best predictor of hypoparathyroidism. This guideline offers recommendations for the prevention, diagnosis, and treatment of acute hypoparathyroidism and hypocalcemia after thyroid surgery.
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Affiliation(s)
- Carlos Eduardo Santa Ritta Barreira
- Hospital DF STAR, Cirurgia de Cabeça e Pescoço, Brasília, DF, Brazil
- Hospital Sírio Libanês Brasília, Cirurgia de Cabeça e Pescoço, Brasília, DF, Brazil
| | - Luiz Paulo Kowalski
- Departamento de Cirurgia de Cabeça e Pescoço e Otorrinolaringologia, AC Camargo Cancer Center, São Paulo, SP, Brazil
- Departamento de Cirurgia de CabeCça e Pescoço, Faculdade de Medicina da Universidade de São Paulo, São Paulo, SP, Brazil
- Instituto De Câncer do Estado de São Paulo (ICESP), Faculdade de Medicina da Universidade de São Paulo (HCFMUSP), São Paulo, SP, Brazil
| | - Fernando Luiz Dias
- Instituto Nacional de Câncer (INCA), Seção de Cirurgia de Cabeça e Pescoço, Rio de Janeiro, RJ, Brazil
- Curso de Pós-graduação em Cirurgia de Cabeça e Pescoço, Pontifícia Universidade Católica do Rio de Janeiro, Rio de Janeiro, Brazil
| | - Terence Pires D E Farias
- Instituto Nacional de Câncer (INCA), Seção de Cirurgia de Cabeça e Pescoço, Rio de Janeiro, RJ, Brazil
- Curso de Pós-graduação em Cirurgia de Cabeça e Pescoço, Pontifícia Universidade Católica do Rio de Janeiro, Rio de Janeiro, Brazil
| | - Izabella Costa Santos
- Instituto Nacional de Câncer (INCA), Seção de Cirurgia de Cabeça e Pescoço, Rio de Janeiro, RJ, Brazil
- Curso de Pós-graduação em Cirurgia de Cabeça e Pescoço, Pontifícia Universidade Católica do Rio de Janeiro, Rio de Janeiro, Brazil
| | - José Guilherme Vartanian
- Departamento de Cirurgia de Cabeça e Pescoço e Otorrinolaringologia, AC Camargo Cancer Center, São Paulo, SP, Brazil
| | | | - Heládio Feitosa
- Departamento de Cirurgia Oncológica, Instituto do Câncer do Ceará, Fortaleza, Ceará, Brazil
| | - Ruy Gomes Neto
- Departamento de Cirurgia Geral e Especializada, Universidade Federal do Estado do Rio de Janeiro, Rio de Janeiro, Brazil
| | | | - Reitan Ribeiro
- Departamento de Cirurgia Oncológica, Hospital Erasto Gaertner, Curitiba, Brazil
| | | | | | - Pedro Eder Portari Filho
- Departamento de Cirurgia Geral e Especializada, Universidade Federal do Estado do Rio de Janeiro, Rio de Janeiro, Brazil
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15
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di Filippo L, Bilezikian JP, Canalis E, Terenzi U, Giustina A. New insights into the vitamin D/PTH axis in endocrine-driven metabolic bone diseases. Endocrine 2024; 85:1007-1019. [PMID: 38632163 DOI: 10.1007/s12020-024-03784-6] [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: 02/17/2024] [Accepted: 03/13/2024] [Indexed: 04/19/2024]
Abstract
BACKGROUND Endocrine regulation of bone metabolisms is the focus of the "Skeletal Endocrinology" series of meetings. AIMS To report on the outcome of the discussion on the role of vitamin D/PTH axis in endocrine osteopathies held during the 10th Skeletal Endocrinology Meeting which took place in Stresa (Italy) in March 2023. OUTCOMES Vitamin D/PTH axis has relevant influence on several outcomes in the general population and in patients affected by endocrinopathies such as hypoparathyroidism and secreting pituitary adenomas. CONCLUSIONS Assessing the status of the vitamin D/PTH axis and using vitamin D and PTH as therapeutic agents is mandatory in several endocrine-related bone metabolic conditions.
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Affiliation(s)
- Luigi di Filippo
- Institute of Endocrine and Metabolic Sciences, San Raffaele Vita Salute University and IRCCS San Raffaele Hospital, Milan, Italy
| | - John P Bilezikian
- Department of Medicine, Endocrinology Division, Vagelos College of Physicians and Surgeons Columbia University, New York, NY, USA
| | - Ernesto Canalis
- Department of Orthopaedic Surgery, UConn Health, Farmington, Connecticut, USA; Department of Medicine, UConn Health, Farmington, Connecticut, USA; UConn Musculoskeletal Institute, UConn Health, Farmington, CT, USA
| | - Umberto Terenzi
- Institute of Endocrine and Metabolic Sciences, San Raffaele Vita Salute University and IRCCS San Raffaele Hospital, Milan, Italy
| | - Andrea Giustina
- Institute of Endocrine and Metabolic Sciences, San Raffaele Vita Salute University and IRCCS San Raffaele Hospital, Milan, Italy.
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16
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Brod M, Pfeiffer KM, Beck JF, Smith A. Measuring treatment impacts on symptoms in adults with hypoparathyroidism: findings from the PaTHway trial. J Patient Rep Outcomes 2024; 8:94. [PMID: 39136801 PMCID: PMC11322464 DOI: 10.1186/s41687-024-00757-1] [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: 01/08/2024] [Accepted: 07/03/2024] [Indexed: 08/16/2024] Open
Abstract
BACKGROUND Hypoparathyroidism is a rare endocrine disease frequently associated with serious physical and cognitive symptoms. This study's purpose was to understand the impacts of the phase 3 PaTHway clinical trial treatment, TransCon PTH, on patients' overall, physical, and cognitive hypoparathyroidism signs/symptoms and what patients consider meaningful improvement. METHODS Individual telephone exit interviews were conducted with patients who recently completed the PaTHway trial blinded period. Using a semi-structured interview guide, interviews focused on trial treatment impact on hypoparathyroidism symptoms following the symptom list in the Hypoparathyroidism Patient Experience Scale-Symptom (HPES-Symptom). Meaningful changes in hypoparathyroidism symptoms were assessed with the Patient Global Impression of Severity (PGIS) and Patient Global Impression of Change (PGIC) measures. Interviewees were probed on the meaningfulness of reported changes in symptoms from prior to starting trial treatment to the past 2 weeks/current time. Interviews were audiotaped and transcribed. Transcripts were coded for emerging concepts and themes/subthemes covered in the interview guide based on an adapted grounded theory approach. RESULTS Nineteen adults with hypoparathyroidism participated in interviews in the United States (n = 13, 68.4%) and Canada (n = 6, 31.6%). Marked improvements in physical and cognitive symptoms were described among trial treatment group respondents. The majority of participants who reported experiencing hypoparathyroidism physical symptoms pre-trial indicated symptom improvement with treatment, including muscle twitching (100%, n = 15), low energy (92.9%, n = 13), feeling tired (92.3%, n = 12), muscle weakness (92.9%, n = 13), tingling without numbness (84.6%, n = 11), trouble sleeping (92.3%, n = 12), muscle cramping (92.3%, n = 12), tingling with numbness (92.3%, n = 12), muscle spasms (100%, n = 12), and pain (90.9%, n = 10). Most participants who reported experiencing cognitive symptoms pre-trial reported symptom improvement with treatment, including difficulty finding the right words (86.7%, n = 13), difficulty concentrating (93.3%, n = 14), trouble remembering (92.9%, n = 13), trouble thinking clearly (85.7%, n = 12), and difficulty understanding information (83.3%, n = 10). Those in the placebo group reported limited or no improvement. The vast majority of participants affirmed that the improvements they experienced in symptom frequency on the PGIS/PGIC and HPES-Symptom were meaningful. CONCLUSIONS Findings indicate that TransCon PTH treatment improved participants' physical and cognitive hypoparathyroidism symptoms in meaningful ways, while reducing the daily burden associated with conventional therapy. TRIAL REGISTRATION NCT04701203 Registered: 06 January 2021. https://clinicaltrials.gov/study/NCT04701203?term=NCT04701203&rank=1 .
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Affiliation(s)
- Meryl Brod
- The Brod Group, 219 Julia Ave., Mill Valley, CA, 94941, USA.
| | | | - Jane F Beck
- The Brod Group, 219 Julia Ave., Mill Valley, CA, 94941, USA
| | - Alden Smith
- Ascendis Pharma, Inc., 1000 Page Mill Road, Palo Alto, CA, 94304, USA
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17
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Soubry E, David K, Swillen A, Vergaelen E, Docx Op de Beeck M, Hulsmans M, Charleer S, Decallonne B. Endocrine manifestations in adults with 22q11.2 deletion syndrome: a retrospective single-center cohort study. J Endocrinol Invest 2024; 47:1827-1836. [PMID: 38308768 PMCID: PMC11196336 DOI: 10.1007/s40618-023-02276-0] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/19/2023] [Accepted: 12/07/2023] [Indexed: 02/05/2024]
Abstract
INTRODUCTION AND OBJECTIVE Patients with the 22q11.2 deletion syndrome (22q11DS) frequently display cardiological and psychiatric diseases, but are also at increased risk for endocrine manifestations. The aim of this study was to evaluate the screening, prevalence, and management of hypoparathyroidism and thyroid disease in patients with 22q11DS, to evaluate the metabolic profile, and to compare these results with current literature and guidelines. DESIGN We performed a retrospective study of patients with genetically confirmed 22q11DS, followed at the center for human genetics of the University Hospitals Leuven, resulting in a cohort of 75 patients. Medical history, medication, and laboratory results concerning hypoparathyroidism, thyroid dysfunction, and the metabolic profile were collected. RESULTS Of the total cohort, 26 patients (35%) had at least one hypocalcaemic episode. During hypocalcaemia, parathyroid hormone (PTH) was measured in only 12 patients with 11 having normal or low PTH, confirming a diagnosis of hypoparathyroidism. Recurrent episodes of hypocalcaemia occurred in seventeen patients (23%). Adherence to the guidelines was low, with 13% of patients having a yearly serum calcium evaluation, 12% receiving daily calcium supplements, and 20% receiving non-active vitamin D. Hypothyroidism was present in 31 patients (44%) and hyperthyroidism in 6 patients (8%). Information on body mass index (BMI) was available in 52 patients (69%), of which 38% were obese (BMI ≥ 30 kg/m2). CONCLUSION Hypoparathyroidism, hypothyroidism, and obesity are common endocrine manifestations in patients with 22q11DS but are probably underdiagnosed and undertreated, indicating the need for multidisciplinary follow-up including an endocrinologist.
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Affiliation(s)
- E Soubry
- Department of Endocrinology, University Hospitals Leuven, Louvain, Belgium
| | - K David
- Department of Endocrinology, University Hospitals Leuven, Louvain, Belgium
| | - A Swillen
- Department of Genetics, University Hospitals Leuven, Louvain, Belgium
| | - E Vergaelen
- Department of Psychiatry, University Hospitals Leuven, Louvain, Belgium
| | | | - M Hulsmans
- Department of Endocrinology, University Hospitals Leuven, Louvain, Belgium
| | - S Charleer
- Department of Endocrinology, University Hospitals Leuven, Louvain, Belgium
| | - B Decallonne
- Department of Endocrinology, University Hospitals Leuven, Louvain, Belgium.
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18
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Towler DA. Parathyroid hormone-PTH1R signaling in cardiovascular disease and homeostasis. Trends Endocrinol Metab 2024; 35:648-660. [PMID: 38429163 PMCID: PMC11233248 DOI: 10.1016/j.tem.2024.02.005] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/16/2023] [Revised: 02/03/2024] [Accepted: 02/05/2024] [Indexed: 03/03/2024]
Abstract
Primary hyperparathyroidism (pHPT) afflicts our aging population with an incidence approaching 50 per 100 000 patient-years at a female:male ratio of ~3:1. Decisions surrounding surgical management are currently driven by age, hypercalcemia severity, presence of osteoporosis, renal insufficiency, or hypercalciuria with or without nephrolithiasis. Cardiovascular (CV) disease (CVD) is not systematically considered. This is notable since the parathyroid hormone (PTH) 1 receptor (PTH1R) is biologically active in the vasculature, and adjusted CV mortality risk is increased almost threefold in individuals with pHPT who do not meet contemporary recommendations for surgical cure. We provide an overview of epidemiology, pharmacology, and physiology that highlights the need to: (i) identify biomarkers that establish a healthy 'set point' for CV PTH1R signaling tone; (ii) better understand the pharmacokinetic-pharmacodynamic (PK-PD) relationships of PTH1R ligands in CV homeostasis; and (iii) incorporate CVD risk assessment into the management of hyperparathyroidism.
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Affiliation(s)
- Dwight A Towler
- Department of Internal Medicine - Endocrine Division, Charles and Jane Pak Center for Mineral Metabolism and Clinical Research, UT Southwestern Medical Center, Dallas, TX 75390, USA.
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19
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Lindsay Mart F, Winer KK, Johnson K, Wasserman H. Initiation of Continuous rhPTH Infusion With Insulin Pump in an Inpatient Setting. JCEM CASE REPORTS 2023; 1:luad136. [PMID: 37954834 PMCID: PMC10634629 DOI: 10.1210/jcemcr/luad136] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 07/19/2023] [Indexed: 11/14/2023]
Abstract
Hypoparathyroidism is one of the few remaining hormonal insufficiencies not treated with replacement of its missing hormone. Conventional therapy involves multiple daily oral doses of calcium, active vitamin D, and magnesium, which is not only cumbersome for patients, but carries risk of nephrocalcinosis and is inadequate in patients with enteral malabsorption. Subcutaneous parathyroid hormone 1-34 (PTH[1-34]) has been tested as a hormonal replacement therapy for treatment of hypoparathyroidism. PTH(1-34) delivered by continuous infusion via insulin pump decreases or eliminates the need for oral medications, stabilizes serum and urine calcium at normal levels with minimal fluctuation, and significantly reduces PTH doses. In this case report, we describe the clinical application of PTH(1-34) via insulin pump in an adolescent with autoimmune polyendocrinopathy syndrome type 1 (APS1). Transition to a PTH pump reduced hospital admissions for calcium abnormalities and allowed our patient to discontinue all scheduled daily conventional therapy.
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Affiliation(s)
- Faith Lindsay Mart
- Division of Endocrinology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA
| | - Karen K Winer
- National Institute of Child Health and Human Development, National Institutes of Health, Bethesda, MD 20817, USA
| | - Karla Johnson
- Division of Endocrinology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA
| | - Halley Wasserman
- Division of Endocrinology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA
- Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH 45267, USA
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20
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Bolster MB, Cahalane AM, Grinspoon SK, Miloslavsky EM. Case 12-2023: A 44-Year-Old Woman with Muscle Weakness and Myalgia. N Engl J Med 2023; 388:1513-1520. [PMID: 37075144 DOI: 10.1056/nejmcpc2211375] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 04/21/2023]
Affiliation(s)
- Marcy B Bolster
- From the Departments of Medicine (M.B.B., S.K.G., E.M.M.) and Radiology (A.M.C.), Massachusetts General Hospital, and the Departments of Medicine (M.B.B., S.K.G., E.M.M.) and Radiology (A.M.C.), Harvard Medical School - both in Boston
| | - Alexis M Cahalane
- From the Departments of Medicine (M.B.B., S.K.G., E.M.M.) and Radiology (A.M.C.), Massachusetts General Hospital, and the Departments of Medicine (M.B.B., S.K.G., E.M.M.) and Radiology (A.M.C.), Harvard Medical School - both in Boston
| | - Steven K Grinspoon
- From the Departments of Medicine (M.B.B., S.K.G., E.M.M.) and Radiology (A.M.C.), Massachusetts General Hospital, and the Departments of Medicine (M.B.B., S.K.G., E.M.M.) and Radiology (A.M.C.), Harvard Medical School - both in Boston
| | - Eli M Miloslavsky
- From the Departments of Medicine (M.B.B., S.K.G., E.M.M.) and Radiology (A.M.C.), Massachusetts General Hospital, and the Departments of Medicine (M.B.B., S.K.G., E.M.M.) and Radiology (A.M.C.), Harvard Medical School - both in Boston
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21
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Gamarra E, Retta F, Lucatello B, Ragazzoni F, Camponovo C, Deandrea M, Trimboli P. Continuous subcutaneous rhPTH infusion for managing difficult chronic hypoparathyroidism. A systematic review. Endocrine 2023:10.1007/s12020-023-03355-1. [PMID: 37017857 DOI: 10.1007/s12020-023-03355-1] [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: 02/13/2023] [Accepted: 03/12/2023] [Indexed: 04/06/2023]
Abstract
PURPOSE Standard treatment for chronic hypoparathyroidism is represented by long-life per os supplementation of calcium and vitamin D. Since 90s, exogenous PTH is also available, but a not negligible number of patients experience a poor control. Starting from the experience with pumps in diabetes, it has been hypothesized that the infusion of PTH through pump might result in a better disease control. The aim of this systematic review is to summarize the published data about continuous subcutaneous PTH infusion in chronic hypoPTH patients and achieve conclusions for clinical practice. METHODS A comprehensive computer literature search of the PubMed/MEDLINE, Embase, and Scopus databases was conducted by two authors independently (last search on November 30, 2022). All findings were summarized and critically discussed. RESULTS We included 14 of the 103 retrieved articles, 2 RCTs, 8 case reports, and 4 case series, published between 2008 and 2022. Of the total 40 patients, 17 were adults, and 23 pediatric. The etiology was postsurgical in 50% of cases and genetic in the other 50%. All had a failure of standard care and a rapid improvement of clinical and biochemical parameters on PTH pump therapy, without severe adverse events. CONCLUSIONS Based on literature, pump PTH infusion may represent an effective, safe, and feasible option for patients with chronic hypoparathyroidism refractory to standard therapy. From a clinical perspective, careful patient selection, a skilled healthcare team, the assessment of the local setting and the collaboration with pump suppliers are essential.
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Affiliation(s)
- Elena Gamarra
- Servizio di Endocrinologia e Diabetologia, Ente Ospedaliero Cantonale (EOC), Bellinzona, Switzerland.
| | - Francesca Retta
- UO Endocrinologia, Diabetologia e Malattie del metabolismo, AO Ordine Mauriziano Torino, Torino, Italy
| | - Barbara Lucatello
- UO Endocrinologia, Diabetologia e Malattie del metabolismo, AO Ordine Mauriziano Torino, Torino, Italy
| | - Federico Ragazzoni
- UO Endocrinologia, Diabetologia e Malattie del metabolismo, AO Ordine Mauriziano Torino, Torino, Italy
| | - Chiara Camponovo
- Servizio di Endocrinologia e Diabetologia, Ente Ospedaliero Cantonale (EOC), Bellinzona, Switzerland
| | - Maurilio Deandrea
- UO Endocrinologia, Diabetologia e Malattie del metabolismo, AO Ordine Mauriziano Torino, Torino, Italy
| | - Pierpaolo Trimboli
- Servizio di Endocrinologia e Diabetologia, Ente Ospedaliero Cantonale (EOC), Bellinzona, Switzerland
- Facoltà di Scienze Biomediche, Università della Svizzera Italiana (USI), Lugano, Switzerland
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22
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Cipriani C, Cianferotti L. Vitamin D in hypoparathyroidism: insight into pathophysiology and perspectives in clinical practice. Endocrine 2023:10.1007/s12020-023-03354-2. [PMID: 37000405 DOI: 10.1007/s12020-023-03354-2] [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: 02/24/2023] [Accepted: 03/12/2023] [Indexed: 04/01/2023]
Abstract
Hypoparathyroidism (HypoPT) is a rare endocrine disorder characterized by the absence or insufficient parathyroid hormone production resulting in chronic hypocalcemia. Complications of HypoPT include perturbation of several target organs. The conventional treatment consists of the administration of active vitamin D, namely calcitriol. Regarding vitamin D status, few data are available, mostly in HypoPT subjects supplemented with parent vitamin D. In addition, perturbation of vitamin D metabolism has been poorly investigated, as well as the contribution of altered vitamin D status on the clinical expression of the disease. The most recent consensus on the management of chronic HypoPT suggests the baseline evaluation of serum 25-hydroxy-vitamin D [25(OH)D] and supplementation with parent vitamin D with the aim to achieve and maintain serum 25(OH)D levels in the range of 30-50 ng/mL. The rationale for using supplementation with parent vitamin D (either ergocalciferol or cholecalciferol) in HypoPT would be to provide sufficient 25(OH)D substrate to the residual 1-α-hydroxylase activity, thus ensuring its conversion to active vitamin D in renal and extra-renal tissues. More data from experimental and clinical studies are needed for better assessing how these mechanisms may significantly influence metabolic control in HypoPT and eventually skeletal and extra-skeletal manifestation of the disease. Finally, future data will clarify how the currently available parent vitamin D compounds (ergocalciferol, cholecalciferol, calcifediol) would perform in addressing these specific issues.
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Affiliation(s)
- Cristiana Cipriani
- Department of Clinical, Internal, Anesthesiological and Cardiovascular Sciences, Sapienza University of Rome, Viale del Policlinico 155, 00161, Rome, Italy.
| | - Luisella Cianferotti
- Department of Experimental and Clinical Biomedical Sciences "Mario Serio", University of Florence, Viale GB Morgagni 50, Florence, 50134, Italy
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23
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Van Uum S, Shrayyef M, M'Hiri I, Dandurand K, Ali DS, Bilezikian JP, Collins MT, Mannstadt M, Rubin MR, Siggelkow H, Tabacco G, Tay YKD, Vokes T, Winer KK, Yao L, Guyatt G, Rejnmark L, Khan AA. Initial Assessment and Monitoring of Patients with Chronic Hypoparathyroidism: A Systematic Current Practice Survey. J Bone Miner Res 2022; 37:2630-2641. [PMID: 36066096 DOI: 10.1002/jbmr.4698] [Citation(s) in RCA: 13] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/22/2022] [Revised: 08/26/2022] [Accepted: 08/31/2022] [Indexed: 11/05/2022]
Abstract
Chronic hypoparathyroidism (HypoPT) is associated with significant morbidity and impaired quality of life (QoL). The goals of management for chronic HypoPT include improvement in QoL and the prevention of both hypo- and hypercalcemia symptoms and long-term complications. Several groups have provided consensus statements and guidelines on the management of HypoPT; however, due to limited evidence, these recommendations have largely been based on literature reviews, expert opinion, and consensus statements. The objective of this study was to use a systematic approach to describe current practice for the initial assessment and follow-up of patients with chronic HypoPT. We developed a survey asking experts in the field to select the responses that best reflect their current practice. The survey found no differences in responses between nonsurgical and postsurgical patient assessment. For new patients, respondents usually performed an assessment of serum lab profile (calcium [either albumin-adjusted or ionized], magnesium, creatinine, phosphate, 25-hydroxyvitamin D), 24-hour urine (creatinine, calcium), and a renal ultrasound to evaluate for the presence of nephrocalcinosis or nephrolithiasis. For follow-up patients, most respondents perform blood tests and urine tests every 6 months or less frequently. The reported clinical practice patterns for monitoring for complications of chronic HypoPT vary considerably among respondents. Based on the responses in this systematic expert practice survey, we provide practice suggestions for initial assessment and follow-up of patients with chronic HypoPT. In addition, we highlight areas with significant variation in practice and identify important areas for future research. © 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)
- Stan Van Uum
- Department of Medicine, Western University, London, ON, Canada
| | | | - Iman M'Hiri
- Bone Research and Education Centre, Oakville, ON, Canada
| | - Karel Dandurand
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada
| | - Dalal S Ali
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada
| | - John P Bilezikian
- Vagelos College of Physicians and Surgeons, Columbia University, New York, NY, USA
| | - Michael T Collins
- National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD, USA
| | - Michael Mannstadt
- Endocrine Unit, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA
| | - Mishaela R Rubin
- Vagelos College of Physicians and Surgeons, Columbia University, New York, NY, USA
| | - Heide Siggelkow
- Clinic of Gastroenterology, Gastrointestinal Oncology and Endocrinology, University Medical Center Goettingen, Goettingen, Germany.,MVZ Endokrinologikum Goettingen, Goettingen, Germany
| | - Gaia Tabacco
- Unit of Endocrinology and Diabetes, Campus Bio-Medico University of Rome, Rome, Italy
| | - Yu-Kwang Donovan Tay
- Department of Medicine, Sengkang General Hospital, Singhealth and Duke-NUS Medical School, Singapore, Singapore
| | | | - Karen K Winer
- Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Bethesda, MD, USA
| | - Liang Yao
- 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
| | - Lars Rejnmark
- Dept of Endocrinology and Internal Medicine, Aarhus University hospital, Aarhus, Denmark
| | - Aliya A Khan
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada
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24
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Khan AA, Bilezikian JP, Brandi ML, Clarke BL, Gittoes NJ, Pasieka JL, Rejnmark L, Shoback DM, Potts JT, Guyatt GH, Mannstadt M. Evaluation and Management of Hypoparathyroidism Summary Statement and Guidelines from the Second International Workshop. J Bone Miner Res 2022; 37:2568-2585. [PMID: 36054621 DOI: 10.1002/jbmr.4691] [Citation(s) in RCA: 51] [Impact Index Per Article: 17.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/11/2022] [Revised: 08/18/2022] [Accepted: 08/24/2022] [Indexed: 11/10/2022]
Abstract
This clinical practice guideline addresses the prevention, diagnosis, and management of hypoparathyroidism (HypoPT) and provides evidence-based recommendations. The HypoPT task forces included four teams with a total of 50 international experts including representatives from the sponsoring societies. A methodologist (GG) and his team supported the taskforces and conducted the systematic reviews. A formal process following the Grading of Recommendations, Assessment, Development and Evaluation (GRADE) methodology and the systematic reviews provided the structure for seven of the guideline recommendations. The task force used a less structured approach based on narrative reviews for 20 non-GRADEd recommendations. Clinicians may consider postsurgical HypoPT permanent if it persists for >12 months after surgery. To predict which patients will not develop permanent postsurgical HypoPT, we recommend evaluating serum PTH within 12 to 24 hours post total thyroidectomy (strong recommendation, moderate quality evidence). PTH > 10 pg/mL (1.05 pmol/L) virtually excludes long-term HypoPT. In individuals with nonsurgical HypoPT, genetic testing may be helpful in the presence of a positive family history of nonsurgical HypoPT, in the presence of syndromic features, or in individuals younger than 40 years. HypoPT can be associated with complications, including nephrocalcinosis, nephrolithiasis, renal insufficiency, cataracts, seizures, cardiac arrhythmias, ischemic heart disease, depression, and an increased risk of infection. Minimizing complications of HypoPT requires careful evaluation and close monitoring of laboratory indices. In patients with chronic HypoPT, the panel suggests conventional therapy with calcium and active vitamin D metabolites as first-line therapy (weak recommendation, low-quality evidence). When conventional therapy is deemed unsatisfactory, the panel considers the use of PTH. © 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)
- Aliya A Khan
- Department of Medicine, Division of Endocrinology and Metabolism, McMaster University, Hamilton, ON, Canada
| | - John P Bilezikian
- Vagelos College of Physicians and Surgeons, Columbia University, New York, NY, USA
| | | | - Bart L Clarke
- Mayo Clinic Division of Endocrinology, Diabetes, Metabolism, and Nutrition, Rochester, MN, USA
| | - Neil J Gittoes
- Centre for Endocrinology Diabetes & Metabolism, Queen Elizabeth Hospital, Birmingham, UK
| | - Janice L Pasieka
- Department of Surgery and Oncology, Cumming School of Medicine, University of Calgary, Calgary, AB, Canada
| | - Lars Rejnmark
- Department of Endocrinology and Internal Medicine, Aarhus University Hospital, Aarhus, Denmark
| | - Dolores M Shoback
- Endocrine Research Unit, San Francisco Veterans Affairs Medical Center, University of California, San Francisco, CA, USA
| | - John T Potts
- Endocrine Unit, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA
| | - Gordon H Guyatt
- Department of Health Research Methods, Evidence and Impact, McMaster University, Hamilton, ON, Canada
| | - Michael Mannstadt
- Endocrine Unit, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA
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25
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Khan AA, Bilezikian JP, Brandi ML, Clarke BL, Potts JJ, Mannstadt M. The Second International Workshop on the Evaluation and Management of Hypoparathyroidism. J Bone Miner Res 2022; 37:2566-2567. [PMID: 36375811 DOI: 10.1002/jbmr.4671] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/02/2022] [Accepted: 08/08/2022] [Indexed: 11/16/2022]
Affiliation(s)
- Aliya A Khan
- Division of Endocrinology and Metabolism, McMaster University, Hamilton, Canada
| | - John P Bilezikian
- Division of Endocrinology, Department of Medicine, Vagelos College of Physicians and Surgeons, Columbia University, New York, NY, USA
| | - Maria Luisa Brandi
- Fondazione Italiana sulla Ricerca sulle Malattie dell'Osso (FIRMO Foundation), Florence, Italy
| | - Bart L Clarke
- Division of Endocrinology, Diabetes, Metabolism, and Nutrition, Mayo Clinic, Rochester, MN, USA
| | - John J Potts
- Endocrine Unit, Department of Medicine, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA
| | - Michael Mannstadt
- Endocrine Unit, Department of Medicine, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA
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26
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Clarke BL. Hypoparathyroidism: update of guidelines from the 2022 International Task Force. ARCHIVES OF ENDOCRINOLOGY AND METABOLISM 2022; 66:604-610. [PMID: 36382749 PMCID: PMC10118814 DOI: 10.20945/2359-3997000000549] [Citation(s) in RCA: 8] [Impact Index Per Article: 2.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Subscribe] [Scholar Register] [Indexed: 11/18/2022]
Abstract
The 2022 International Task Force guidelines for chronic hypoparathyroidism will be published within several months in the Journal of Bone and Mineral Research. These guidelines update the original guidelines published in 2016, and include new information from literature published since then. Chronic postsurgical hypoparathyroidism is now defined as lasting for at least 12 months after surgery, rather than 6 months. Chronic postsurgical hypoparathyroidism may be predicted by serum PTH <10 pg/mL in the first 12-24 hours after surgery. The most common symptoms and complications of chronic hypoparathyroidism based on the literature are summarized in detail. How to monitor and manage patients with hypoparathyroidism is described in detail where recommendations can be given. These guidelines are intended to frame the diagnosis and care of patients with chronic hypoparathyroidism for at least the next five years.
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27
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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: 176] [Impact Index Per Article: 58.7] [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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