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Pal R, Mukherjee S, Prasad TN, Bhadada SK. Pregnancy with primary hyperparathyroidism. Best Pract Res Clin Endocrinol Metab 2025; 39:101983. [PMID: 40023680 DOI: 10.1016/j.beem.2025.101983] [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] [Indexed: 03/04/2025]
Abstract
Primary hyperparathyroidism (PHPT) in pregnancy is rare. The physiological changes that occur in pregnancy often tend to mask the symptoms of PHPT, thereby making diagnosis challenging. If left undiagnosed, PHPT can lead to significant feto-maternal morbidity, which, primarily depends on maternal serum calcium levels. Maternal serum calcium > 11.4 mg/dl increases the risk of incident maternal and fetal complications. The diagnosis of PHPT in pregnancy is based on the documentation of parathyroid hormone-dependent hypercalcemia. Ultrasonography can be safely used to localize the culprit parathyroid lesions; other imaging modalities entailing the risk of exposure to ionizing radiation should preferably be avoided. Treatment involves parathyroid surgery (preferably performed in the second trimester) and/or medical management (hydration, use of calcium-lowering drugs like calcitonin and/or cinacalcet) and should be tailored to the term of pregnancy, severity of hypercalcemia, potential maternal-foetal risks involved, available surgical expertise and patient's choices.
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Affiliation(s)
- Rimesh Pal
- Department of Endocrinology, Postgraduate Institute of Medical Education and Research (PGIMER), Chandigarh 160012, India.
| | - Soham Mukherjee
- Department of Endocrinology, Postgraduate Institute of Medical Education and Research (PGIMER), Chandigarh 160012, India.
| | - Trupti N Prasad
- Department of Endocrinology, TNMC and BYL Nair Hospital, Mumbai 400008, India.
| | - Sanjay Kumar Bhadada
- Department of Endocrinology, Postgraduate Institute of Medical Education and Research (PGIMER), Chandigarh 160012, India.
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Eli S, Gal SG, Adnan Z. Comparison between surgical and non-surgical management of primary hyperparathyroidism during pregnancy: a systematic review. Endocrine 2024; 86:101-108. [PMID: 38918290 PMCID: PMC11445326 DOI: 10.1007/s12020-024-03930-0] [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: 03/27/2024] [Accepted: 06/11/2024] [Indexed: 06/27/2024]
Abstract
PURPOSE The management of primary hyperparathyroidism (PHPT) during pregnancy may be surgical or conservative. This study compared adverse outcomes between surgical and non-surgical treatments. Additionally, the study investigated the correlation between serum calcium values and complication rates. METHODS A systematic review of retrospective studies, case series, and case reports. Biochemical parameters, interventions, and outcomes of each pregnancy were recorded. The study population comprised two groups: the non-surgical and surgical groups. Adverse outcomes were categorized as maternal, obstetric, or neonatal. RESULTS The surgical and non-surgical groups consisted of 163 and 185 patients, respectively. A positive correlation was observed between the mean maternal gestational calcium value and both maternal and obstetric complication. Neonatal complications were more prevalent in patients treated conservatively across all maternal calcium values (p < 0.001). No significant differences were observed in maternal outcomes and overall obstetric outcomes between the study groups, albeit a higher mean serum calcium value in the surgical group (12.3 mg/dL) compared with the non-surgical group (11.1 mg/dL). CONCLUSIONS Given the significantly lower neonatal adverse outcomes in the surgical group compared to the non-surgical group, along with non-inferior maternal and obstetric outcomes in the surgical group, the overall data of this study suggest that parathyroidectomy is favorable to non-surgical management even in cases of mild hypercalcemia.
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Affiliation(s)
- Shezifi Eli
- Bar-Ilan University, The Azrieli Faculty of Medicine, Safed, Israel
- Laniado Hospital, Netanya, Israel
| | - Shlomo Gozlan Gal
- Department of Physiology and Cell Biology, Ben-Gurion University of the Negev, Beer-Sheva, Israel
| | - Zaina Adnan
- Bar-Ilan University, The Azrieli Faculty of Medicine, Safed, Israel.
- Division of Endocrinology and Metabolism, Clalit Medical Health Care Services, Haifa and Western Galilee District, Zvulon Medical Center, Haifa, Israel.
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3
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Foster E, Maalouf NM. Use of Cinacalcet for the Management of Primary Hyperparathyroidism in Pregnancy. JCEM CASE REPORTS 2024; 2:luae117. [PMID: 39011404 PMCID: PMC11247167 DOI: 10.1210/jcemcr/luae117] [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: 04/16/2024] [Indexed: 07/17/2024]
Abstract
Primary hyperparathyroidism in pregnancy is uncommon. Consequently, there are no randomized controlled studies that address treatment of primary hyperparathyroidism in pregnancy, and the efficacy and safety of medical management with cinacalcet in this setting is unknown. We report a case of a 28-year-old woman with primary hyperparathyroidism and hypercalcemia that worsened during her third trimester of pregnancy. Cinacalcet led to achievement of normocalcemia, allowing the delay of parathyroidectomy until after delivery of the baby. We also review the published literature on cinacalcet use in the management of primary hyperparathyroidism during pregnancy. Cinacalcet is typically reserved for pregnant patients with severe and symptomatic hypercalcemia, primarily serving as a last resort to delay parathyroidectomy until either the second trimester or the postpartum period.
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Affiliation(s)
- Elissa Foster
- Nephrology Division, Baylor University Medical Center, Dallas, TX 75246, USA
| | - Naim M Maalouf
- Charles and Jane Pak Center for Mineral Metabolism and Clinical Research, UT Southwestern Medical Center, Dallas, TX 75390-8885, USA
- Department of Internal Medicine, UT Southwestern Medical Center, Dallas, TX 75390-8885, USA
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Trahan MJ, Antinora C, Czuzoj-Shulman N, Benjamin A, Abenhaim HA. Obstetrical and neonatal outcomes among pregnancies complicated by hyperparathyroidism. J Matern Fetal Neonatal Med 2023; 36:2170748. [PMID: 36775282 DOI: 10.1080/14767058.2023.2170748] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/14/2023]
Abstract
PURPOSE Severe hypercalcemia resulting from hyperparathyroidism may result in adverse perinatal outcomes. The objective of this study was to evaluate maternal and neonatal outcomes among pregnant women with hyperparathyroidism using a population database. METHODS A retrospective cohort study was conducted using data from the Healthcare Cost and Utilization Project-Nationwide Inpatient Sample from 1999-2015. ICD-9 codes were used to identify women diagnosed with hyperparathyroidism during pregnancy. Perinatal outcomes between pregnant women with and without hyperparathyroidism were compared. Multivariate logistic regression, controlling for age, race, income, insurance type, hospital location, and comorbidities, evaluated the effect of hyperparathyroidism on perinatal outcomes. RESULTS Of 13,792,544 deliveries included over the study period, 368 were to women with hyperparathyroidism. The overall incidence of hyperparathyroidism was 2.7/100,000 births, increasing from 1.6 to 5.2/100,000 births over the study period (p < 0.0001). Women with hyperparathyroidism were older and had more comorbidities, such as obesity, and pre-gestational hypertension and diabetes. Relative to the comparison group, women with hyperparathyroidism were more likely to deliver preterm, OR 1.69 (95% CI 1.24-2.29), to develop preeclampsia, 3.14 (2.30-4.28), and to deliver by cesarean, 1.69 (1.36-2.09). Infants born to mothers with hyperparathyroidism were more likely to be growth restricted, 1.83 (1.08-3.07), and to be diagnosed with a congenital anomaly, 4.21 (2.09-8.48). CONCLUSION Hyperparathyroidism during pregnancy is associated with a significant increase in adverse perinatal outcomes, including preeclampsia, preterm delivery, fetal growth restriction, and congenital anomalies. As such, pregnancies among women with hyperparathyroidism should be considered high-risk, and specialized care is recommended in order to minimize maternal and neonatal morbidity.
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Affiliation(s)
- Marie-Julie Trahan
- Department of Obstetrics and Gynecology, McGill University, Montreal, Quebec, Canada
| | | | | | - Alice Benjamin
- Department of Obstetrics and Gynecology, McGill University, Montreal, Quebec, Canada
| | - Haim A Abenhaim
- Department of Obstetrics and Gynecology, McGill University, Montreal, Quebec, Canada.,Department of Obstetrics and Gynecology, Division of Maternal-Fetal Medicine, Jewish General Hospital, Montreal, Quebec, Canada
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Zhong H, Liao Q, Liu J. Expert consensus on multidisciplinary approach to the diagnosis and treatment of primary hyperparathyroidism in pregnancy in China. Endocrine 2023; 82:282-295. [PMID: 37221429 DOI: 10.1007/s12020-023-03392-w] [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: 03/15/2023] [Accepted: 04/29/2023] [Indexed: 05/25/2023]
Abstract
Primary hyperparathyroidism in pregnancy is a rare disease that can have detrimental effects on both maternal and fetal/neonatal outcomes. The physiological changes that occur during pregnancy can complicate the diagnosis, imaging examinations, and treatment of this disorder. To enhance our understanding and management of primary hyperparathyroidism in pregnancy, experts from various fields, including endocrinology, obstetrics, surgery, ultrasonography, nuclear medicine, pediatrics, nephrology, and general practice in China, collaborated to develop a consensus addressing the critical aspects of the diagnosis and treatment of primary hyperparathyroidism in pregnancy with a multidisciplinary team approach. This consensus provides valuable guidance for healthcare professionals in managing this condition, ultimately improving outcomes for both mothers and their babies.
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Affiliation(s)
- Huiping Zhong
- Department of Obstetrics and Gynecology, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, 200025, China.
| | - Quan Liao
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, 100730, China.
| | - Jianmin Liu
- Department of Endocrine and Metabolic Diseases, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, 200025, China.
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Diaz Dilernia F, Estefan M, Zanotti G. Pathological femoral neck fracture in a male patient due to non-simultaneous bilateral transient hip osteoporosis: A case report: Statements and Declarations. J Clin Orthop Trauma 2023; 42:102207. [PMID: 37529549 PMCID: PMC10388589 DOI: 10.1016/j.jcot.2023.102207] [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: 09/15/2022] [Revised: 04/11/2023] [Accepted: 06/24/2023] [Indexed: 08/03/2023] Open
Abstract
We report a case of a 31-year-old man who developed a non-simultaneous bilateral transient hip osteoporosis (THO) complicated with an atraumatic undisplaced pathological femoral neck fracture (FNF) of his left hip. He was successfully treated with internal fixation on the left hip, and a follow-up Magnetic Resonance Imaging evidenced satisfactory resolution on the right side. Diagnosis and management of THO are usually challenging as symptoms before pathological FNFs might be misdiagnosed. This case highlights the importance of suspecting pathological FNFs when pain persists despite conservative treatment. Finally, surgical treatment is strongly recommended if a pathological FNF is confirmed. Level of evidence Level IV.
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Affiliation(s)
- Fernando Diaz Dilernia
- Adult Hip and Knee Reconstructive Surgery, Division of Orthopaedic Surgery, Department of Surgery, Queen's University & Kingston Health Sciences Centre, Kingston, Ontario, Canada, K7L 2V7
| | - Martin Estefan
- Hip Surgery Unit, Institute of Orthopaedics “Carlos E. Ottolenghi”, Italian Hospital of Buenos Aires, Buenos Aires, C1199ACK, Argentina
| | - Gerardo Zanotti
- Hip Surgery Unit, Institute of Orthopaedics “Carlos E. Ottolenghi”, Italian Hospital of Buenos Aires, Buenos Aires, C1199ACK, Argentina
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A Fatal Case of Metastatic Pulmonary Calcification during the Puerperium. Int J Mol Sci 2022; 23:ijms232315131. [PMID: 36499458 PMCID: PMC9735927 DOI: 10.3390/ijms232315131] [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: 10/28/2022] [Revised: 11/28/2022] [Accepted: 11/29/2022] [Indexed: 12/05/2022] Open
Abstract
We present an unusual case of a fatal respiratory failure in a young woman developed two weeks after she gave birth at home. Circumstantial and clinical features of the case were strongly suggestive for a 'classical' septic origin of the respiratory symptoms. Autopsy, together with histopathological and immunohistochemical analyses allowed demonstrating a massive calcium redistribution consisting of an important osteolysis, especially from cranial bones and abnormal accumulation in lungs and other organs. Such physiopathology was driven by a primary hyperparathyroidism secondary to a parathyroid carcinoma as demonstrated by immunohistochemistry. This very rare case is furthermore characterised by a regular pregnancy course, ended with the birth of a healthy new-born. A complex interaction between pregnancy physiology and hyperparathyroidism might be hypothesised, determining the discrepancy between the relative long period of wellness and the tumultuous cascade occurred in the puerperium.
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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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9
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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: 200] [Impact Index Per Article: 66.7] [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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Arshad MF, Arambewela MH, Bennet WM, Sterrenburg M, Balasubramanian SP. Primary hyperparathyroidism in pregnancy: experience of a tertiary centre. Surg Today 2022; 53:470-475. [PMID: 36107253 PMCID: PMC10042935 DOI: 10.1007/s00595-022-02583-8] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/29/2021] [Accepted: 08/03/2022] [Indexed: 11/24/2022]
Abstract
Abstract
Background and purpose
The management of primary hyperparathyroidism (PHPT) during pregnancy is challenging and there is no clear consensus on whether it increases the risk of complications in pregnancy. We conducted this study to review the maternal and fetal outcomes of pregnant women treated for PHPT in a single centre.
Methods
Data on relevant clinical parameters, demographics, management strategies, maternal and fetal outcomes were collected from the medical records of pregnant patients with PHPT diagnosed between 2012 and 2019.
Results
Of 15 pregnant women with PHPT, 6 were managed medically and 9 underwent surgery. The median age at their index pregnancy was 28 years [range 19–42]. The median highest adjusted calcium level in the medical group was 2.90 [range 2.61–3.25] mmol/L vs. 3.11 [2.78–4.95] mmol/L in the surgical group. There was one miscarriage and the stillbirth of twins in the medical group, but no such outcomes in the surgical group. The median gestational ages were 39 + 3 weeks [range 24 + 2–41 + 2 weeks] and 39 + 4 weeks [range 37 + 1–39 + 5 weeks] in the medical and surgical groups, respectively. No birth was complicated by neonatal tetany or convulsions.
Conclusion
More complications developed in the pregnant PHPT patients who were managed medically than in those who underwent surgery. Surgery performed during the second trimester resulted in good outcomes. Multi-centre prospective studies are required to ascertain the risk of various complications in women with PHPT during pregnancy.
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Affiliation(s)
- Muhammad Fahad Arshad
- Oncology and Metabolism, University of Sheffield Medical School, Sheffield, S10 2RX, UK.
- Sheffield Teaching Hospitals, Royal Hallamshire Hospital, Sheffield, S10 2JF, UK.
| | - Maulee Hiromi Arambewela
- Sheffield Teaching Hospitals, Royal Hallamshire Hospital, Sheffield, S10 2JF, UK
- University of Sri Jayewardenepura, Nugegoda, Sri Lanka
| | - William M Bennet
- Sheffield Teaching Hospitals, Royal Hallamshire Hospital, Sheffield, S10 2JF, UK
| | - Monique Sterrenburg
- Sheffield Teaching Hospitals, Royal Hallamshire Hospital, Sheffield, S10 2JF, UK
| | - Saba P Balasubramanian
- Oncology and Metabolism, University of Sheffield Medical School, Sheffield, S10 2RX, UK
- Sheffield Teaching Hospitals, Royal Hallamshire Hospital, Sheffield, S10 2JF, UK
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11
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Beck AJ, Reddy VM, Sulkin T, Browne D. Management of severe and symptomatic primary hyperparathyroidism in the first trimester of unplanned pregnancy. Endocrinol Diabetes Metab Case Rep 2022; 2022:21-0203. [PMID: 36070416 PMCID: PMC9513675 DOI: 10.1530/edm-21-0203] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/23/2022] [Accepted: 08/17/2022] [Indexed: 11/08/2022] Open
Abstract
Summary Primary hyperparathyroidism (PHP) is the most common aetiology for hypercalcaemia. The incidence of PHP in pregnant women is reported to be 8/100 000 population/year. It presents a threat to the health of both mother (hyperemesis, nephrolithiasis) and fetus (fetal death, congenital malformations, and neonatal severe hypocalcaemia-induced tetany). However, there is a lack of clear guidance on the management of primary hyperparathyroidism in pregnancy. In this study, we describe the case of a 26-year-old female patient who presented with severe hypercalcaemia secondary to PHP and underwent successful parathyroid adenectomy under local anaesthesia. Learning points Primary hyperparathyroidism is a rare complication in pregnancy, but the consequences for mother and fetus can be severe. A perceived risk of general anaesthesia to the fetus in the first trimester has resulted in a general consensus to delay parathyroid surgery to the second trimester when possible - although the increased risk of fetal loss may occur before planned surgery. If the patient presents with severe or symptomatic hypercalcaemia, minimally invasive surgery under local anaesthetic should be considered regardless of the gestational age of the pregnancy.
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Affiliation(s)
- Adele J Beck
- Royal Devon University Healthcare NHS Foundation Trust, Exeter, UK
| | - Venkat M Reddy
- Royal Cornwall Hospitals NHS Trust, Endocrinology and Diabetes Mellitus, Treliske, Truro, UK
| | - Tom Sulkin
- Royal Cornwall Hospitals NHS Trust, Endocrinology and Diabetes Mellitus, Treliske, Truro, UK
| | - Duncan Browne
- Royal Cornwall Hospitals NHS Trust, Endocrinology and Diabetes Mellitus, Treliske, Truro, UK
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12
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Chung JY, Lee YS, Pyeon SY, Han SA, Huh H. Bilateral superficial cervical plexus block for parathyroidectomy during pregnancy: A case report. World J Clin Cases 2022; 10:4153-4160. [PMID: 35665113 PMCID: PMC9131236 DOI: 10.12998/wjcc.v10.i13.4153] [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: 08/06/2021] [Revised: 01/21/2022] [Accepted: 03/17/2022] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Primary hyperparathyroidism (PHPT) is the most common cause of pregnancy-related hypercalcemia. PHPT can cause maternal and fetal complications in pregnant women. General anesthesia for non-obstetric surgery in pregnant women is associated with maternal hazards and concerns regarding long-term neonatal neurocognitive effects. Surgical removal of the lesion in mid-pregnancy is currently the primary treatment option for pregnant patients with PHPT. However, the blood calcium concentration at which surgery should be considered remains under discussion due to the risk of miscarriage.
CASE SUMMARY A 31-year-old nulliparous woman at 11 wk of gestation was admitted to our hospital for parathyroidectomy. The patient had a history of intrauterine fetal death with unknown etiology at 16 wk of gestation 1 year prior. Her blood test results showed that the serum calcium level was elevated to 12.9 mg/dL, and the parathyroid hormone level was elevated to 157 pg/mL. In a neck ultrasound, it revealed a 0.8 cm × 1.5 cm sized oval, hypoechoic mass in the upper posterior of the left thyroid gland, which was compatible with parathyroid adenoma. Superficial cervical plexus block (SCPB) for parathyroidectomy was performed. After surgery, the obstetrician checked the status of the fetus, and there were no abnormal signs. Since then her calcium level returned to normal values after one week of surgery and a healthy male neonate of 2910 g was delivered vaginally at 38 wk of gestation.
CONCLUSION Our case suggests that SCPB can be an anesthetic option for parathyroidectomy during the first trimester of pregnancy.
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Affiliation(s)
- Jun-Young Chung
- Department of Anesthesiology and Pain Medicine, Kyung Hee University Hospital at Gangdong, College of Medicine, Kyung Hee University, Seoul 05278, South Korea
| | - Yo Seob Lee
- Department of Anesthesiology and Pain Medicine, Kyung Hee University Hospital at Gangdong, College of Medicine, Kyung Hee University, Seoul 05278, South Korea
| | - Seung Yeon Pyeon
- Department of Obstetrics & Gynecology, Kyung Hee University Hospital at Gangdong, College of Medicine, Kyung Hee University, Seoul 05278, South Korea
| | - Sang-Ah Han
- Department of Surgery, Kyung Hee University Hospital at Gangdong, College of Medicine, Kyung Hee University, Seoul 05278, South Korea
| | - Hyub Huh
- Department of Anesthesiology and Pain Medicine, Kyung Hee University Hospital at Gangdong, College of Medicine, Kyung Hee University, Seoul 05278, South Korea
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13
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Gonçalves JS, Queiró LC, Soares J, Correia CR. Transient neonatal hypocalcaemia caused by maternal hyperparathyroidism. BMJ Case Rep 2022; 15:e248262. [PMID: 35296496 PMCID: PMC8928309 DOI: 10.1136/bcr-2021-248262] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 02/25/2022] [Indexed: 11/04/2022] Open
Abstract
Hypocalcaemia in neonates can range from asymptomatic to a potentially life-threatening condition. We present a case of a 36 weeks gestational age boy, admitted to our neonatal intensive care unit for jitteriness, mild hypotonia and breastfeeding difficulties. By the ninth day of life, he presented with late-onset hypocalcaemia, hypomagnesaemia, low 25-OH-vitamin D and inappropriately normal parathyroid hormone. Further investigation revealed maternal hypercalcaemia with high parathyroid hormone. Maternal asymptomatic hyperparathyroidism was diagnosed and admitted as the cause of neonatal hypocalcaemia. There was a clinical improvement and calcium levels stabilisation after treatment with calcium gluconate and vitamin D3 This case highlights the importance of careful evaluation of neonatal late-onset hypocalcaemia in uncovering asymptomatic maternal hyperparathyroidism.
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Affiliation(s)
- Joana S Gonçalves
- Pediatrics Department, Centro Hospitalar de Lisboa Ocidental EPE, Lisboa, Portugal
| | | | - Joana Soares
- Pediatrics Department, Centro Hospitalar de Lisboa Ocidental EPE, Lisboa, Portugal
| | - Cátia R Correia
- Pediatrics Department, Centro Hospitalar de Lisboa Ocidental EPE, Lisboa, Portugal
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14
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Gumber L, Sivasankaran K, Khan SMS. Parathyroid carcinoma presenting with ventricular bigeminy in pregnancy. BMJ Case Rep 2022; 15:e247069. [PMID: 35135802 PMCID: PMC8830158 DOI: 10.1136/bcr-2021-247069] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 01/13/2022] [Indexed: 11/03/2022] Open
Abstract
Parathyroid carcinoma is very rare in pregnancy. Clinical features are similar to primary hyperparathyroidism. A 38-year-old pregnant woman had repeated hospital admissions for palpitations, headaches, dizziness and polydipsia. Blood investigations showed severe hypercalcaemia with raised parathyroid hormone and 24-hour ECG showed ventricular bigeminy and premature ventricular contractions. Neck ultrasound showed a lesion in the right thyroid lobe. Consequently, she underwent an en bloc resection of the right parathyroid and thyroid lobe at 23 weeks gestation. Histology results confirmed parathyroid cancer. This case highlights the complexities of identifying hypercalcaemia in pregnancy due to the overlapping features with common disorders of pregnancy. Early recognition and timely surgical management can prevent maternal and fetal complications. Also, the case demonstrates the value of interprofessional collaboration between different specialities in providing quality care and improving outcomes. An abridged version of this case was presented at European Congress of Endocrinology 2021.
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Affiliation(s)
- Leher Gumber
- Nottingham University Hospitals NHS Trust, Nottingham, UK
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15
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Hyperparathyroidism in Pregnancy: A Review of the Literature. Obstet Gynecol Surv 2022; 77:35-44. [PMID: 34994393 DOI: 10.1097/ogx.0000000000000977] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/25/2022]
Abstract
Importance Maternal hyperparathyroidism can be associated with significant maternal and fetal morbidity and fetal mortality. Because the maternal symptoms are typically nonspecific, the disorder may not be recognized leading to adverse pregnancy outcomes. Objective The aim of this study was to review the literature on the etiology/prevalence, pathophysiology, diagnosis, management (medical and surgical), and the maternal/neonatal complications associated with pregnancies complicated by hyperparathyroidism. Evidence Acquisition A literature search was undertaken by our university librarian using the search engines PubMed and Web of Science. Search terms used included "hyperparathyroidism" AND "pregnancy" OR "pregnancy complications" OR "maternal." The number of years searched was not limited, but the abstracts had to be in English. Results There were 309 abstracts identified, 164 of which are the basis of this review. This includes 137 articles of the 269 individual case reports in the literature since the first case report in 1947. The articles and case reports reviewed the etiology, risk factors, diagnosis, management, complications, and maternal/fetal outcomes of pregnancies complicated by hyperparathyroidism. Conclusions and Relevance Undiagnosed maternal hyperparathyroidism can result in critical maternal and fetal outcomes during pregnancy. This review highlights what is currently known about hyperparathyroidism during pregnancy to increase the awareness of this serious pregnancy disorder.
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16
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Pliakos I, Chorti A, Moysidis M, Kotsovolis G, Kaltsas T, Pana A, Ioannidis A, Papavramidis TS. Parathyroid adenoma in pregnancy: A case report and systematic review of the literature. Front Endocrinol (Lausanne) 2022; 13:975954. [PMID: 36325457 PMCID: PMC9618884 DOI: 10.3389/fendo.2022.975954] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/22/2022] [Accepted: 09/27/2022] [Indexed: 12/02/2022] Open
Abstract
OBJECTIVE Primary hyperparathyroidism is a common disorder of the parathyroid glands. Parathyroid adenoma (PA) in pregnancy is a relatively rare disease, whose diagnosis and treatment is a challenging task. The aim of the present study is to present a new case of parathyroid adenoma during pregnancy and to give a detailed account of all reported cases of parathyroid adenoma during pregnancy in the literature. STUDY DESIGN A bibliographic research was performed, and characteristics of parathyroid adenomas in pregnancy such as age, gestational week at diagnosis, ionized calcium levels, genetic testing result, symptomatology, radiological method of localization, treatment method, gestational week at operation, and maternal/fetal complications were recorded. RESULTS A 34-year-old woman at her 25 weeks' gestation was diagnosed with parathyroid adenoma and was referred to our Surgical Department due to contraindication for conservative treatment. A parathyroidectomy was performed, and the maternal and fetal postoperative period was uneventful. Two hundred eleven cases of parathyroid adenoma in pregnancy were recorded in the literature, and statistical analysis was performed. The median gestational week at diagnosis was 21 ± 9.61 weeks. The mean level of ionized calcium was 2.69 mmol/l [SD = 0.75 (2.55-2.84 95% CI)]. Most cases were familiar (72.4%), while surgery was the preferred treatment option (67.3%). The majority of cases were asymptomatic (21.7%), and the main radiological method applied for localization was ultrasound (63.4%). CONCLUSION Parathyroid adenoma in pregnancy is a rare condition. The early diagnosis is of great importance as surgical treatment at the second trimester of pregnancy outweighs the maternal and fetal risks.
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Affiliation(s)
- I. Pliakos
- Department of Minimal Invasive Endocrine Surgery, Kyanous Stavros, Euromedica Clinic, Thessaloniki, Greece
| | - A. Chorti
- 1st Propedeutic Department of Surgery, AHEPA University Hospital, Faculty of Health Science, Medical School, Aristotle University of Thessaloniki, Thessaloniki, Greece
| | - Moysis Moysidis
- Department of Minimal Invasive Endocrine Surgery, Kyanous Stavros, Euromedica Clinic, Thessaloniki, Greece
| | - G. Kotsovolis
- Department of Minimal Invasive Endocrine Surgery, Kyanous Stavros, Euromedica Clinic, Thessaloniki, Greece
| | - T. Kaltsas
- Department of Minimal Invasive Endocrine Surgery, Kyanous Stavros, Euromedica Clinic, Thessaloniki, Greece
| | - A. Pana
- 1st Department of Obstetrics and Gynecology, Papageorgiou Hospital, Faculty of Health Science, Medical School, Aristotle University of Thessaloniki, Thessaloniki, Greece
| | - A. Ioannidis
- 1st Propedeutic Department of Surgery, AHEPA University Hospital, Faculty of Health Science, Medical School, Aristotle University of Thessaloniki, Thessaloniki, Greece
| | - TS. Papavramidis
- Department of Minimal Invasive Endocrine Surgery, Kyanous Stavros, Euromedica Clinic, Thessaloniki, Greece
- 1st Propedeutic Department of Surgery, AHEPA University Hospital, Faculty of Health Science, Medical School, Aristotle University of Thessaloniki, Thessaloniki, Greece
- *Correspondence: TS. Papavramidis,
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Abstract
Hypercalcemic disorders are rare in pregnant women and are usually due to primary hyperparathyroidism. Clinical manifestations of hypercalcemia are nonspecific and can be masked by the physiologic changes of pregnancy. Furthermore, routine antenatal screening does not include serum calcium measurement and a hypercalcemia diagnosis may therefore be delayed until term or even after delivery. Timely recognition and appropriate interventions are essential to decrease maternal and fetal complications. Conservative measures are appropriate in the presence of mild hypercalcemia. Parathyroidectomy remains the mainstay of treatment for primary hyperparathyroidism with significant hypercalcemia not responding to conservative measures.
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Affiliation(s)
- Karel Dandurand
- Division of Endocrinology and Metabolism, McMaster University, Bone Research and Education Centre, 3075 Hospital Gate, Unit 223, Oakville, ON L6M 1M1, Canada
| | - Dalal S Ali
- Division of Endocrinology and Metabolism, McMaster University, Bone Research and Education Centre, 3075 Hospital Gate, Unit 223, Oakville, ON L6M 1M1, Canada
| | - Aliya A Khan
- Division of Endocrinology and Metabolism, McMaster University, Bone Research and Education Centre, 3075 Hospital Gate, Unit 223, Oakville, ON L6M 1M1, Canada.
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18
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Islam AK. Advances in the diagnosis and the management of primary hyperparathyroidism. Ther Adv Chronic Dis 2021; 12:20406223211015965. [PMID: 34178298 PMCID: PMC8202248 DOI: 10.1177/20406223211015965] [Citation(s) in RCA: 10] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/08/2020] [Accepted: 04/20/2021] [Indexed: 12/26/2022] Open
Abstract
The parathyroid glands, one of the last organs to be discovered, are responsible for maintaining calcium homeostasis, and they continue to present the clinician with diagnostic and management challenges that are reviewed herein. Primary hyperparathyroidism (PHPT) comprises the vast majority of pathology of the parathyroid glands. The classic variant, presenting with elevated calcium and parathyroid hormone levels, has been studied extensively, but the current body of literature has added to our understanding of normocalcemic and normohormonal variants of PHPT, as well as syndromic forms of PHPT. All variants can lead to bone loss, kidney stones, declining renal function, and a variety of neurocognitive, gastrointestinal, and musculoskeletal complaints, although the majority of PHPT today is asymptomatic. Surgery remains the definitive treatment for PHPT, and advances in screening, evolving indications for surgery, new imaging modalities, and improvements in intra-operative methods have greatly changed the landscape. Surgery continues to produce excellent results in the hands of an experienced parathyroid surgeon. For those patients who are not candidates for surgery, therapeutic advances in medical management allow for improved control of the hypercalcemic state. Parathyroid cancer is extremely rare; the diagnosis is often made intra-operatively or on final pathology, and recurrence is common. The mainstay of treatment is normalization of serum calcium via surgery and medical adjuncts.
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Affiliation(s)
- Ana Kashfia Islam
- Department of Surgery, UT Southwestern Medical Center, 5323 Harry Hines Boulevard, Dallas, TX 75390-9159, USA
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19
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Xu Y, Yu Y. Primary hyperparathyroidism presenting as a brown tumor with hypercalcemia crisis in a second-trimester pregnant woman: A case report. Medicine (Baltimore) 2021; 100:e25968. [PMID: 34011079 PMCID: PMC8137077 DOI: 10.1097/md.0000000000025968] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/06/2021] [Accepted: 04/28/2021] [Indexed: 01/05/2023] Open
Abstract
INTRODUCTION Primary hyperparathyroidism (PHPT) in pregnancy is rare and unrecognized because the maternal physiological adaptations blurs the symptoms. There is no standard treatment strategy for maternal PHPT. Early diagnosis and interventions can prevent catastrophic consequences to the mother and fetus. PATIENT CONCERNS A 31-year-old Chinese woman was admitted, due to a lump on the left lower leg for 4 months. The patient complained of mild pain in the left lower leg following exercise that could be relieved after a short rest. The patient was at 18 weeks of gestation, and the growth of the fetus was normal. The patient has a 3-year history of hypercalcemia and a 2-year history of nephrolithiasis. No family history of hypercalcemia and endocrine tumors were present. DIAGNOSIS Laboratory tests demonstrated high serum calcium level of 3.84 mmol/L, parathyroid hormone 1393 pg/mL, alkaline phosphatase 488 μ/L. Ultrasound showed a 22.4 mm × 7.8 mm solid nodule in the left lower lobe of the thyroid gland. Based on these findings, the patient was diagnosed with PHPT. INTERVENTIONS The patient accepted continuous renal replacement to reduce ironized calcium level. Parathyroidectomy was performed at the 19th week of gestation. Threatened abortion occurred 2 days after the surgery, and magnesium sulfate was used to prevent the abortion. Calcium gluconate, calcium carbonate and vitamin D3 were used to treat the hypocalcemia that occurred 5 days after the surgery. OUTCOMES Pathology examination demonstrated the parathyroid adenoma. Abortion was prevented using magnesium sulfate and hypocalcemia was cured with calcium gluconate, calcium carbonate and vitamin D3. At 38-week of gestation, the patient (ionized calcium level: 2.16 mmol/L) delivered a healthy female baby weighing 2700 g with 10/10 Apgar. Till now, both the mother and infant showed no complications. CONCLUSION Maternal PHPT is rare and challenging to diagnose, causing life-threatening complications to mother and fetus. Any decision regarding surgery for a pregnant woman with primary hyperparathyroidism is more complex than in men or nonpregnant women. The decision should be made based on the severity of hypercalcemia and symptoms.
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Affiliation(s)
- Yun Xu
- International Medicine Department
| | - Yingying Yu
- The Second Affiliated Hospital Zhejiang University School of Medicine, Hangzhou, China
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20
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Tsourdi E, Anastasilakis AD. Parathyroid Disease in Pregnancy and Lactation: A Narrative Review of the Literature. Biomedicines 2021; 9:475. [PMID: 33925967 PMCID: PMC8145477 DOI: 10.3390/biomedicines9050475] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/13/2021] [Revised: 04/18/2021] [Accepted: 04/20/2021] [Indexed: 12/27/2022] Open
Abstract
Pregnancy and lactation are characterized by sophisticated adaptations of calcium homeostasis, aiming to meet fetal, neonatal, and maternal calcium requirements. Pregnancy is primarily characterized by an enhancement of intestinal calcium absorption, whereas during lactation additional calcium is obtained through resorption from the maternal skeleton, a process which leads to bone loss but is reversible following weaning. These maternal adaptations during pregnancy and lactation may influence or confound the presentation, diagnosis, and management of parathyroid disorders such as primary hyperparathyroidism or hypoparathyroidism. Parathyroid diseases are uncommon in these settings but can be severe when they occur and may affect both maternal and fetal health. This review aims to delineate the changes in calcium physiology that occur with pregnancy and lactation, describe the disorders of calcium and parathyroid physiology that can occur, and outline treatment strategies for these diseases in the above settings.
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Affiliation(s)
- Elena Tsourdi
- Center for Healthy Aging, Department of Medicine III, Technische Universität Dresden Medical Center, 01307 Dresden, Germany
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21
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Vetchinnikova ON, Prokopenko EI, Voronkova IA. [Adenoma of the parathyroid gland in a young woman with chronic kidney disease: primary or tertiary hyperparathyroidism?]. TERAPEVT ARKH 2020; 92:78-82. [PMID: 33346483 DOI: 10.26442/00403660.2020.10.000557] [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: 11/23/2020] [Accepted: 11/23/2020] [Indexed: 11/22/2022]
Abstract
Clinical observation of a young woman with chronic kidney disease IV stage and hyperparathyroidism is presented. Ultrasound and99mTc-sestamibi scintigraphy of the anterior surface of the neck visualized a tumor of the left upper parathyroid gland. In a histological examination of distant education was diagnosed a solid parathyroid adenoma. The difficulty of differential diagnosis between primary and secondary/tertiary hyperparathyroidism in chronic kidney disease is discussed.
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Affiliation(s)
| | | | - I A Voronkova
- Vladimirsky Moscow Regional Research Clinical Institute.,Endocrinology Research Centre
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22
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Atypical pregnancy-associated osteoporosis of the patella: a case report. CURRENT ORTHOPAEDIC PRACTICE 2020. [DOI: 10.1097/bco.0000000000000910] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022]
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23
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Bansal S, Kaushik RM, Kaushik R, Modi S, Raghuvanshi S, Kusum A. Primary hyperparathyroidism presenting as severe hypercalcemia with acute pancreatitis in pregnancy. Gynecol Endocrinol 2020; 36:469-472. [PMID: 31793366 DOI: 10.1080/09513590.2019.1698028] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023] Open
Abstract
Primary hyperparathyroidism presenting first time with severe hypercalcemia is rare in pregnancy. We report a case of primary hyperparathyroidism due to a cystic parathyroid adenoma presenting as severe hypercalcemia with acute pancreatitis in second trimester of pregnancy. Acute pancreatitis was managed by conservative treatment. Hypercalcemia failed to respond to medical management and ultimately responded to ultrasound-guided ethanol ablation of parathyroid adenoma. The delivery was uneventful and patient continues to remain normocalcemic during follow up. As such, ethanol ablation of parathyroid adenoma may be considered during pregnancy in case of failure of response to medical management and when surgical removal of parathyroid adenoma is not safe.
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Affiliation(s)
- S Bansal
- Department of General Medicine, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Dehradun, India
| | - R M Kaushik
- Department of General Medicine, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Dehradun, India
| | - R Kaushik
- Department of General Medicine, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Dehradun, India
| | - S Modi
- Department of General Medicine, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Dehradun, India
| | - S Raghuvanshi
- Department of Radiodiagnosis, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Dehradun, India
| | - A Kusum
- Department of Pathology, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Dehradun, India
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24
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Abstract
Endocrine emergencies in pregnancy can be life threatening and are associated with increased morbidity for both the mother and fetus. Thyroid storm, diabetic ketoacidosis, and hypercalcemic crisis require a high clinical suspicion, rapid treatment, and multidisciplinary care to ensure best outcomes. Critical care consultation and intensive care unit admission are often warranted. Fetal testing may initially be concerning; however often improves with correction of the underlying metabolic derangement(s) and delivery is generally avoided until maternal status improves.
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25
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Abstract
Hyperparathyroidism is a rare disease during pregnancy, which has increased risks, including miscarriage and fetal growth restriction. However, the diagnosis of hyperparathyroidism is frequently not recognised or delayed as symptoms are non-specific and calcium is not routinely measured. With a thorough medical history and clinical suspicion, early diagnosis and treatment can reduce the risk of some pregnancy complications. A 35-year-old woman presented at 13/40 with hyperemesis gravidarum. She had elevated serum calcium and a parathyroid lesion on ultrasound. She underwent a parathyroidectomy with rapid normalisation of her calcium. Histopathology confirmed a parafibromin-deficient parathyroid tumour, suggestive of hyperparathyroidism jaw tumour syndrome. At 30/40, she presented with pre-eclampsia (hypertension, hyper-reflexia, proteinuria and intrauterine growth restriction) and had a caesarean section at 30+1/40, delivering a male infant, 897 g (fifth percentile). She had a prior 12-month history of chronic constipation and nephrolithiasis but was not investigated further despite elevated calcium (2.82 mmol/L).
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Affiliation(s)
- Catherine Davis
- Faculty of Medicine and Health, The University of Sydney Northern Clinical School, St Leonards, New South Wales, Australia
| | - Tanya Nippita
- Faculty of Medicine and Health, The University of Sydney Northern Clinical School, St Leonards, New South Wales, Australia.,Department of Obstetrics and Gynaecology, Royal North Shore Hospital, Sydney, New South Wales, Australia
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26
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Sharma SG, Levine SN, Yatavelli RK, Shaha MA, Nathan CAO. Parathyroidectomy in First Trimester of Pregnancy. J Endocr Soc 2020; 4:bvaa015. [PMID: 32133431 PMCID: PMC7049289 DOI: 10.1210/jendso/bvaa015] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/01/2019] [Indexed: 11/24/2022] Open
Abstract
Primary hyperparathyroidism is not commonly diagnosed during pregnancy. For pregnant women with mild, asymptomatic disease, surgery can be avoided unless the degree of hypercalcemia becomes more severe or they develop complications. However, there are no evidence-based guidelines to assist clinicians regarding the management of primary hyperparathyroidism during pregnancy. When surgery is deemed necessary during pregnancy, the second trimester is generally considered to be the optimal time. We report the case of a 31-year-old female G1P0 who presented at 6 weeks gestation with symptoms of nausea, vomiting, polyuria, and corrected calcium of 14.8 mg/dL. Due to the extreme degree of hypercalcemia and refractory to medical treatment, it was decided that surgery could not be delayed until the second trimester. At 7w3d gestational age the patient had resection of a 37 gram, 5 × 4 × 3 cm right inferior parathyroid adenoma.
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Affiliation(s)
- Sonia G Sharma
- Department of Medicine, Division of Endocrinology, Diabetes and Metabolism, Louisiana State University Health Sciences Center, Shreveport, Louisiana
| | - Steven N Levine
- Department of Medicine, Division of Endocrinology, Diabetes and Metabolism, Louisiana State University Health Sciences Center, Shreveport, Louisiana
| | - Rajini K Yatavelli
- Department of Medicine, Division of Endocrinology, Diabetes and Metabolism, Louisiana State University Health Sciences Center, Shreveport, Louisiana
| | - Manish A Shaha
- Department of Otolaryngology-Head and Neck Surgery, Louisiana State University Health Sciences Center, Shreveport, Louisiana
| | - Cherie Ann O Nathan
- Department of Otolaryngology-Head and Neck Surgery, Louisiana State University Health Sciences Center, Shreveport, Louisiana
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27
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Sprenger-Mähr H, Zitt E, Kronbichler A, Cejna M, Lhotta K. A hemodialysis patient with bone disease after pregnancy: a case report. BMC Nephrol 2019; 20:425. [PMID: 31752733 PMCID: PMC6873679 DOI: 10.1186/s12882-019-1603-8] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/01/2019] [Accepted: 10/28/2019] [Indexed: 11/10/2022] Open
Abstract
BACKGROUND Pregnancy is rare in women on hemodialysis. Recommendations for the treatment of secondary hyperparathyroidism (sHPT) and preservation of bone health in pregnant dialysis patients are lacking. CASE PRESENTATION We present the case of a young woman with end-stage kidney disease (ESKD) due to lupus nephritis, who developed multiple brown tumors while on hemodialysis during her second pregnancy. During her first pregnancy sHPT was well controlled and no skeletal complications occurred. Before the second pregnancy she developed severe sHPT. During pregnancy, dialysis time was increased to 24 h per week, the patient was given oral calcitriol, and the dialysate calcium concentration was set at 1.5 mmol/l. In week 20 the patient complained about bone pain in her left hip. Magnetic resonance imaging revealed a cystic lesion compatible with a brown tumor. The baby was delivered in the 36th week by cesarean section. Further assessment identified multiple brown tumors of her skeleton, including the acetabulum, tibia, ribs, skull, thoracic spine and thumb. She required multiple orthopedic surgeries. Three months after pregnancy, etelcalcetide was started, which brought about a gradual improvement in her sHPT. CONCLUSIONS This case demonstrates that the combination of pregnancy and severe sHPT in dialysis patients can have deleterious consequences for bone health.
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Affiliation(s)
- Hannelore Sprenger-Mähr
- Department of Internal Medicine III, Academic Teaching Hospital Feldkirch, Carinagasse 47, Feldkirch, Austria
| | - Emanuel Zitt
- Department of Internal Medicine III, Academic Teaching Hospital Feldkirch, Carinagasse 47, Feldkirch, Austria
| | - Andreas Kronbichler
- Department of Internal Medicine IV, Medical University of Innsbruck, Innsbruck, Austria
| | - Manfred Cejna
- Institute for Diagnostic and Interventional Radiology, Academic Teaching Hospital Feldkirch, Feldkirch, Austria
| | - Karl Lhotta
- Department of Internal Medicine III, Academic Teaching Hospital Feldkirch, Carinagasse 47, Feldkirch, Austria.
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28
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Malheiro E, Chabbert-Buffet N, Talbot JN, Périé S. Hyperparathyroidism in pregnancy and 99mTc-MIBI scintigraphy. Eur Ann Otorhinolaryngol Head Neck Dis 2019; 136:501-503. [DOI: 10.1016/j.anorl.2019.05.009] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
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29
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Haciyanlı M, Özlem Gür EÖ, Genç H, Gücek Haciyanlı S, Tatar F, Acar T, Karaisli S. Minimally invasive parathyroidectomy using intraoperative ultrasonographic localization for primary hyperparathyroidism in pregnancy: report of two cases. Turk J Surg 2019; 35:231-235. [PMID: 32550334 DOI: 10.5578/turkjsurg.4330] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/08/2018] [Accepted: 01/08/2019] [Indexed: 11/15/2022]
Abstract
Primary hyperparathyroidism (pHPT) in pregnancy is a rare entity associated with increased maternal and fetal mortality and morbidity. Diagnosis of pHPT is challenging in pregnancy. Approximately 80% of the cases are asymptomatic, while the most common symptoms are nausea, vomiting, polyuria, polydypsia, and cloudy vision in symptomatic patients. Since the most common cause of pHPT in pregnancy is adenoma, such in the general population, focused anterior or lateral approach is recommended due to shorter operation time, less risk for the fetus, and lower complication risk. Performing intraoperative ultrasonography to do the incision just over the adenoma provides quicker access to the adenoma and intraoperative parathormone assay confirms the surgical cure. Laryngeal mask anesthesia causes lesser sore throat, laryngospasm, coughing, and rapid recovery as compared to endotracheal intubation anesthesia. This study aimed to present the management of two pregnant patients diagnosed with pHPT and who underwent minimally invasive parathyroidectomy under intraoperative ultrasonography and laryngeal mask anesthesia at the second trimester of gestation. To the best of our knowledge, parathyroidectomy under laryngeal mask anesthesia in pregnancy has never been described before.
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Affiliation(s)
- Mehmet Haciyanlı
- Clinic of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
| | - Emine Özlem Özlem Gür
- Clinic of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
| | - Hüdai Genç
- Clinic of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
| | - Selda Gücek Haciyanlı
- Clinic of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
| | - Fatma Tatar
- Clinic of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
| | - Turan Acar
- Clinic of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
| | - Serkan Karaisli
- Clinic of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
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Leere JS, Vestergaard P. Calcium Metabolic Disorders in Pregnancy: Primary Hyperparathyroidism, Pregnancy-Induced Osteoporosis, and Vitamin D Deficiency in Pregnancy. Endocrinol Metab Clin North Am 2019; 48:643-655. [PMID: 31345528 DOI: 10.1016/j.ecl.2019.05.007] [Citation(s) in RCA: 12] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
Physiologic changes during pregnancy include calcium, phosphate, and calciotropic hormone status. Calcium metabolic disorders are rare in pregnancy and management with close calcium and vitamin D control and supplementation. Primary hyperparathyroidism is mostly asymptomatic and does not affect conception or pregnancy. It requires control of plasma calcium levels. Surgical intervention may be indicated. Data on severe cases are missing. Osteoporosis in or before pregnancy is rare but usually diagnosed from fractures. Medical treatment other than supplementation is contraindicated. Vitamin D deficiency is common and may affect conception and increase complications. Current evidence does not prove vitamin D supplements effective in improving outcomes.
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Affiliation(s)
- Julius Simoni Leere
- Department of Clinical Medicine and Endocrinology, Aalborg University, Aalborg University Hospital, Aalborg, Denmark; Department of Endocrinology, Aalborg University Hospital, Mølleparkvej 4, Aalborg 9000, Denmark.
| | - Peter Vestergaard
- Department of Endocrinology, Aalborg University Hospital, Mølleparkvej 4, Aalborg 9000, Denmark; Steno Diabetes Center North Jutland, Aalborg, Denmark
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Yang J, Dong MJ, Chen F. A rare lethal case of severe acute necrotizing pancreatitis due to a parathyroid adenoma in a third-trimester pregnant woman. BMC Endocr Disord 2019; 19:82. [PMID: 31357974 PMCID: PMC6664735 DOI: 10.1186/s12902-019-0409-9] [Citation(s) in RCA: 6] [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] [Received: 04/30/2019] [Accepted: 07/15/2019] [Indexed: 02/07/2023] Open
Abstract
BACKGROUND Primary hyperparathyroidism (PHPT), which is mostly caused by a parathyroid adenoma, is fairly common in postmenopausal women but is relatively rare in pregnant women. PHPT-induced pancreatitis during pregnancy is associated with significant maternal and foetal morbidity and mortality. Diagnosis is challenging because of non-specific symptoms and changes in maternal calcium homeostasis. Information about the optimal treatment strategy for the prevention of catastrophic consequences to the mother and foetus is limited. Here, we describe a rare lethal case of severe acute necrotizing pancreatitis due to a parathyroid adenoma in a woman in her third trimester of pregnancy. CASE PRESENTATION A previously healthy 24-year-old Chinese woman at 37 weeks of gestation presented with persisting epigastric pain, nausea and bilious vomiting for 1 day. PHPT-induced acute necrotizing pancreatitis was diagnosed on the basis of her serum calcium, parathyroid levels and imaging results. A caesarean section and parathyroidectomy were performed at 1 day and 11 days after admission, respectively. Histological examination confirmed a right inferior parathyroid adenoma with a size of 2.0 × 1.5 cm. Following the parathyroidectomy, the patient had eucalcaemia and presented normal parathyroid hormone (PTH) levels. Although the foetus was normal, the patient died of multiple organ failure due to severe pancreatitis. CONCLUSIONS PHPT-induced acute necrotizing pancreatitis is a rare clinical entity and life-threatening condition to both the mother and the foetus during pregnancy. Early diagnosis can be challenging and is crucial. Appropriate treatment according to the patient's condition may effectively reduce maternal and foetal mortality.
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Affiliation(s)
- Jun Yang
- Department of Nuclear Medicine, the First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, 310003 People’s Republic of China
| | - Meng-jie Dong
- Department of Nuclear Medicine, the First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, 310003 People’s Republic of China
| | - Feng Chen
- Department of Radiology, the First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, 310003 People’s Republic of China
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Zhang YJ, Shi LX, Xu Y, Zheng X, Li JW, Wu Q, Li SX, He YH. Optical quantum weak measurement coupled with UV spectrophotometry for sensitively and non-separatedly detecting enantiopurity. OPTICS EXPRESS 2019; 27:9330-9342. [PMID: 31045086 DOI: 10.1364/oe.27.009330] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Received: 11/26/2018] [Accepted: 03/06/2019] [Indexed: 06/09/2023]
Abstract
Based on the theories of quantum weak measurement, we built a set of linear common-path optical weak measurement systems in frequency domain for detecting chiral molecules. The polarization resolution with this system to detect the optical rotation of chirality molecules is nearly two orders of magnitude higher than that of conventional polarizers. Combined with ultraviolet spectroscopy, the purity of the proline enantiomers mixture was detected. The purity resolution can reach to 0.14%, which is comparable to the liquid chromatography. Weak measurement combined with ultraviolet spectroscopy to non-separatedly detect the purity of chiral enantiomers has great application potential in the pharmaceutical industry.
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Calina D, Docea AO, Golokhvast KS, Sifakis S, Tsatsakis A, Makrigiannakis A. Management of Endocrinopathies in Pregnancy: A Review of Current Evidence. INTERNATIONAL JOURNAL OF ENVIRONMENTAL RESEARCH AND PUBLIC HEALTH 2019; 16:ijerph16050781. [PMID: 30836653 PMCID: PMC6427139 DOI: 10.3390/ijerph16050781] [Citation(s) in RCA: 18] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 01/28/2019] [Revised: 02/27/2019] [Accepted: 02/27/2019] [Indexed: 12/29/2022]
Abstract
Pregnancy in women with associated endocrine conditions is a therapeutic challenge for clinicians. These disorders may be common, such us thyroid disorders and diabetes, or rare, including adrenal and parathyroid disease and pituitary dysfunction. With the development of assisted reproductive techniques, the number of pregnancies with these conditions has increased. It is necessary to recognize symptoms and correct diagnosis for a proper pharmacotherapeutic management in order to avoid adverse side effects both in mother and fetus. This review summarizes the pharmacotherapy of these clinical situations in order to reduce maternal and fetal morbidity.
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Affiliation(s)
- Daniela Calina
- Department of Clinical Pharmacy, University of Medicine and Pharmacy of Craiova, 200349 Craiova, Romania.
| | - Anca Oana Docea
- Department of Toxicology, University of Medicine and Pharmacy of Craiova, 200349 Craiova, Romania.
| | | | - Stavros Sifakis
- Department of Obstetrics and Gynecology, Mitera Maternity Hospital, 71110 Heraklion, Crete, Greece.
| | - Aristides Tsatsakis
- Department of Forensic Sciences and Toxicology, Faculty of Medicine, University of Crete, 71110 Heraklion, Crete, Greece.
| | - Antonis Makrigiannakis
- Department of Obstetrics and Gynecology, Medical School, University of Crete, 71110 Heraklion, Crete, Greece.
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Rigg J, Gilbertson E, Barrett HL, Britten FL, Lust K. Primary Hyperparathyroidism in Pregnancy: Maternofetal Outcomes at a Quaternary Referral Obstetric Hospital, 2000 Through 2015. J Clin Endocrinol Metab 2019; 104:721-729. [PMID: 30247615 DOI: 10.1210/jc.2018-01104] [Citation(s) in RCA: 34] [Impact Index Per Article: 5.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/21/2018] [Accepted: 09/17/2018] [Indexed: 12/14/2022]
Abstract
CONTEXT Primary hyperparathyroidism (PHPT) in pregnancy has historically been associated with substantial maternofetal morbidity and mortality rates. The optimal treatment and timing of surgical intervention in pregnancy remain contested. OBJECTIVE To compare maternofetal outcomes of medically and surgically treated patients with PHPT in pregnancy. DESIGN Retrospective chart review. SETTING Quaternary referral hospital. PATIENTS Women with PHPT in pregnancy treated between 1 January 2000 and 31 December 2015. INTERVENTIONS Medical therapy or parathyroid surgery. MAIN OUTCOMES MEASURED Timing of diagnosis; maternal corrected serum calcium concentrations; gestation, indication and mode of delivery; complications attributable to PHPT; birth weight; and admission to the neonatal intensive care unit (NICU). RESULTS Twenty-two pregnancies were managed medically, and six patients underwent parathyroidectomy in pregnancy (five in trimester 2, and one at 32 weeks gestation). Most patients treated medically either had a corrected serum calcium concentration <2.85 mmol/L in early pregnancy or had PHPT diagnosed in trimester 3. Of viable medically managed pregnancies, 30% were complicated by preeclampsia, and preterm delivery occurred in 66% of this group. All preterm neonates required admission to the NICU for complications related to prematurity. All surgically treated patients delivered their babies at term, and there were no complications of parathyroid surgery. CONCLUSION Maternofetal outcomes have improved relative to that reported in early medical literature in patients treated medically and surgically, but the rates of preeclampsia and preterm delivery were higher in medically treated patients. The study was limited by its retrospective design and small sample sizes.
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MESH Headings
- Adult
- Antihypertensive Agents/therapeutic use
- Calcium/blood
- Female
- Humans
- Hyperparathyroidism, Primary/blood
- Hyperparathyroidism, Primary/complications
- Hyperparathyroidism, Primary/therapy
- Infant, Newborn
- Infant, Premature, Diseases/epidemiology
- Infant, Premature, Diseases/etiology
- Infant, Premature, Diseases/therapy
- Infusions, Intravenous
- Intensive Care Units, Neonatal/statistics & numerical data
- Pamidronate/therapeutic use
- Parathyroidectomy/statistics & numerical data
- Pre-Eclampsia/epidemiology
- Pre-Eclampsia/etiology
- Pregnancy
- Premature Birth/epidemiology
- Premature Birth/etiology
- Referral and Consultation/statistics & numerical data
- Rehydration Solutions/administration & dosage
- Retrospective Studies
- Time Factors
- Young Adult
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Affiliation(s)
- Jane Rigg
- Royal Brisbane and Women's Hospital, Herston, Queensland, Australia
- The University of Queensland, Royal Brisbane Clinical Unit, Herston, Queensland, Australia
| | - Elise Gilbertson
- Sunshine Coast University Hospital, Birtinya, Queensland, Australia
- The University of Queensland, Sunshine Coast Clinical Unit, Sunshine Coast Health Institute, Birtinya, Queensland, Australia
| | - Helen L Barrett
- Royal Brisbane and Women's Hospital, Herston, Queensland, Australia
- The University of Queensland, Royal Brisbane Clinical Unit, Herston, Queensland, Australia
| | - Fiona L Britten
- Royal Brisbane and Women's Hospital, Herston, Queensland, Australia
- The University of Queensland, Royal Brisbane Clinical Unit, Herston, Queensland, Australia
| | - Karin Lust
- Royal Brisbane and Women's Hospital, Herston, Queensland, Australia
- The University of Queensland, Royal Brisbane Clinical Unit, Herston, Queensland, Australia
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35
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Zhang Y, Ding JW, Yu LY, Luo DC, Sun JL, Lei ZK, Wang ZH. Twin pregnancy with triple parathyroid adenoma: A case report and review of literature. World J Clin Cases 2018; 6:466-471. [PMID: 30294612 PMCID: PMC6163144 DOI: 10.12998/wjcc.v6.i11.466] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/20/2018] [Revised: 07/27/2018] [Accepted: 08/06/2018] [Indexed: 02/05/2023] Open
Abstract
Primary hyperparathyroidism (PHPT) is rare during pregnancy. A case of twin pregnancy with three simultaneous parathyroid adenomas at the same time has not been reported. Multiple parathyroid lesions are difficult to diagnose, as pregnant women who insist upon continuing a pregnancy are not able to undergo 99mTc-sestamibi scintigraphy, so cases of PHPT are easily unobserved and often can have serious consequences for the patient and the fetus. Therefore, we reported a case of a 28-year-old woman mid-pregnancy with twins, who had hypercalcemia and was eventually diagnosed with twin pregnancy with PHPT due to a triple parathyroid adenoma, had good pregnancy outcomes after undergoing surgery in mid-pregnancy. Twin pregnancy with PHPT due to a triple parathyroid adenoma, as presented in this case, is very rare and surgery in mid-pregnancy is demonstrated here as safe. Intraoperative parathormone monitoring was and remains key to a successful operation.
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Affiliation(s)
- Yu Zhang
- Department of Oncology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
| | - Jin-Wang Ding
- Department of Oncology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
| | - Ling-Ying Yu
- Department of Endocrinology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
| | - Ding-Cun Luo
- Department of Oncology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
| | - Jian-Liang Sun
- Department of Anesthesiology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
| | - Zhi-Kai Lei
- Department of Ultrasound Branch, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
| | - Zhi-Hua Wang
- Department of Obstetrics and Gynecology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
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36
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Refardt J, Farina P, Hoesli I, Meier C. Hypercalcemic crisis in third trimenon: evaluating the optimal treatment strategy. Gynecol Endocrinol 2018; 34:833-836. [PMID: 29658374 DOI: 10.1080/09513590.2018.1462314] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 10/17/2022] Open
Abstract
Hypercalcemia due to primary hyperparathyroidism during pregnancy is a rare condition and associated with increased morbidity and mortality for the mother and the unborn child. Whereas parathyroidectomy is favored during the second trimester, no clear recommendations exist for its management during the third trimenon. We here report the case of a 26-year-old woman in the 29th week of her first pregnancy, who was admitted to our clinic with hypertension, intra-uterine growth retardation and polyhydramnios. Severe hypercalcemia due to primary hyperparathyroidism was diagnosed (total calcium 3.34 mmol/l; PTH 216 pg/ml), but no enlarged parathyroid gland could be localized by ultrasound. Treatment with calcitonin and cinacalcet could not control hypercalcemia. Therefore explorative surgery was performed and a single parathyroid adenoma was resected, resulting in normalization of serum calcium levels. The surgical procedure was tolerated well by the mother and fetus. Hypercalcemia-induced hypertension and polyhydramnios ameliorated before C-section was performed two weeks later and unrelated to the intervention. This case report underlines the importance of early diagnosis and treatment of primary hyperparathyroidism during pregnancy. If diagnosed in the third trimenon, an interdisciplinary approach is crucial. If medical treatment fails to sufficiently control hypercalcemia, surgical parathyroid exploration should be considered even in cases of unsuccessful localization of adenomatous parathyroid glands.
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Affiliation(s)
- Julie Refardt
- a Division of Endocrinology, Diabetes and Metabolism , University Hospital Basel , Switzerland
| | - Patricia Farina
- b Department of Obstetrics and Gynecology , University Hospital Basel , Switzerland
| | - Irene Hoesli
- b Department of Obstetrics and Gynecology , University Hospital Basel , Switzerland
| | - Christian Meier
- a Division of Endocrinology, Diabetes and Metabolism , University Hospital Basel , Switzerland
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37
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Primary Hyperparathyroidism in Pregnancy: Successful Parathyroidectomy during First Trimester. Case Rep Endocrinol 2018; 2018:5493917. [PMID: 30174966 PMCID: PMC6106801 DOI: 10.1155/2018/5493917] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/16/2018] [Accepted: 07/31/2018] [Indexed: 01/18/2023] Open
Abstract
Primary hyperparathyroidism in pregnancy can result in significant maternal and fetal complications. When indicated, prompt parathyroidectomy in the early second trimester is considered the treatment of choice. Pregnant patients with primary hyperparathyroidism who have an indication for parathyroidectomy during the first trimester represent a therapeutic challenge. We present the case of a 32-year-old primigravida who presented with symptomatic hypercalcemia from her primary hyperparathyroidism. She remained symptomatic despite aggressive conservative management and underwent parathyroidectomy in her first trimester with excellent outcomes.
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38
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Primary Hyperparathyroidism in Pregnancy: A Multicenter Case Series of 4 Patients from Northern India. J Obstet Gynaecol India 2018; 69:31-35. [PMID: 30956488 DOI: 10.1007/s13224-018-1148-y] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/09/2017] [Accepted: 06/14/2018] [Indexed: 11/27/2022] Open
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Koren R, Neeman O, Koren S, Benbassat CA. Humoral hypercalcemia of pregnancy treated with bisphosphonates. ARCHIVES OF ENDOCRINOLOGY AND METABOLISM 2018; 62:125-128. [PMID: 29694631 PMCID: PMC10118686 DOI: 10.20945/2359-3997000000016] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Received: 04/02/2017] [Accepted: 08/27/2017] [Indexed: 11/23/2022]
Abstract
Hypercalcemia can be hazardous during pregnancy, most cases being due to primary hyperparathyroidism. We report a case of hypercalcemia with suppressed PTH levels necessitating treatment with bisphosphonates during pregnancy. A 38-year-old woman at the 26th week gestation was admitted because of symptomatic hypercalcemia. She did not take any medication that could influence her calcium levels. Physical examination was unremarkable. Laboratory tests on admission were: calcium 12.7 mg/dL (8.5-10.5 mg/dL), phosphorus 1.8 mg/dL (2.5-4.5 mg/dL) and PTH on 3 consecutive tests 1.2, 1.3 and 1.2 pg/mL (15-65 pg/mL). Her 24h urine calcium was 900 mg, 25-OH-D 40 ng/mL (30-58 ng/mL) and 1,25-OH-D 99 pg/mL (80-146 for women in the third trimester). Abdominal ultrasound revealed multiple hypervascular liver lesions consistent with hemangiomas by MRI. Breast and neck ultrasound were normal, and chest CT revealed few non-significant 0.3-0.7 cm pulmonary nodules with no change after an interval of 3 months. She was treated with isotonic saline, loop diuretics and calcitonin. Despite this treatment, calcium levels remained high (14.1 mg/dL), and pamidronate was initiated. On 35th week gestation, she underwent a cesarean section complicated by hypocalcemia of the newborn. Eight weeks after delivery, her calcium levels are 9.4 mg/dL and PTH 18 mg/dL. According to the extensive workup and the post-partum normalization of PTH and calcium levels, we conclude that excessive secretion of placental PTHrP was the cause of hypercalcemia in this patient. No significant adverse effect of bisphosphonate on the mother or baby were seen at the short term follow up.
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Affiliation(s)
- Ronit Koren
- Department of Internal Medicine A, Assaf Harofeh Medical Center, Zerifin, Israel
| | - Ortal Neeman
- Division of Maternal Fetal Medicine, Department of Obstetrics and Gynecology, Assaf Harofeh Medical Center, Zerifin, Israel
| | - Shlomit Koren
- Sackler Faculty of Medicine, Tel-Aviv University, Tel-Aviv, Israel
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40
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Zeng H, Li Z, Zhang X, Wang N, Tian Y, Wang J. Anesthetic management of primary hyperparathyroidism during pregnancy: A case report. Medicine (Baltimore) 2017; 96:e9390. [PMID: 29390544 PMCID: PMC5758246 DOI: 10.1097/md.0000000000009390] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/31/2022] Open
Abstract
RATIONALE Primary hyperparathyroidism (PHPT) during pregnancy is rare. Nevertheless, hypercalcemia secondary to gestational PHPT may be masked by physiological changes in calcium homeostasis during pregnancy. Gestational PHPT constitutes a serious danger to mother and fetus. Surgery is the only curative treatment when conservative treatment could not control the condition. Due to the lack of guidelines concerning PHPT during pregnancy, the optimal anesthetic management of PHPT during pregnancy needs to be individualized. Patient concerns: We report a case of PHPT with successful surgical treatment under combined cervical plexus block and general anesthesia. DIAGNOSIS She was diagnosed with hypercalcemia, PHPT, a possible parathyroid adenoma, and a 19-week intrauterine pregnancy. INTERVENTIONS The patient underwent heparin-free hemodialysis before the surgery in the nephrology department in the presence of a cardiologist. She then received a successful parathyroidectomy under combined bilateral superficial cervical plexus block and general anesthesia in her 19th week of pregnancy. OUTCOMES She was released from the hospital with no maternal or fetal complications on postoperative day 9. A healthy baby boy was uneventfully born at 37 weeks of gestation. LESSONS We suggest that surgical removal of the lesion after lowering the blood calcium concentration in mid-pregnancy is currently the optimal treatment option for pregnant patients with PHPT. Furthermore, multidisciplinary perioperative management is particularly important.
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41
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Gonzalo García I, Robles Fradejas M, Martín Macías MDLA, Biain Ciganda A, Bustinza Beaskoetxea Z, Ruiz Pérez E, Fernández Matia G, Martínez Guisasola J. Primary hyperparathyroidism in pregnancy treated with cinacalcet: a case report. J OBSTET GYNAECOL 2017; 38:132-134. [PMID: 28760052 DOI: 10.1080/01443615.2017.1325862] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/19/2022]
Affiliation(s)
- Irene Gonzalo García
- a Department of Obstetrics and Gynecology, Prenatal Diagnosis Unit , Burgos University Hospital , Burgos , Spain
| | - Mónica Robles Fradejas
- a Department of Obstetrics and Gynecology, Prenatal Diagnosis Unit , Burgos University Hospital , Burgos , Spain
| | | | - Ana Biain Ciganda
- a Department of Obstetrics and Gynecology, Prenatal Diagnosis Unit , Burgos University Hospital , Burgos , Spain
| | - Zuriñe Bustinza Beaskoetxea
- a Department of Obstetrics and Gynecology, Prenatal Diagnosis Unit , Burgos University Hospital , Burgos , Spain
| | - Enrique Ruiz Pérez
- b Department of Endocrinology , Burgos University Hospital , Burgos , Spain
| | | | - Javier Martínez Guisasola
- a Department of Obstetrics and Gynecology, Prenatal Diagnosis Unit , Burgos University Hospital , Burgos , Spain
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Harbeck B, Rahvar AH, Danneberg S, Schütt M, Sayk F. Life-threatening endocrine emergencies during pregnancy - management and therapeutic features. Gynecol Endocrinol 2017; 33:510-514. [PMID: 28361555 DOI: 10.1080/09513590.2017.1307959] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/28/2022] Open
Abstract
Endocrine emergencies during pregnancy may be life-threatening events for both mother and fetus. Besides pregnancy-associated endocrine disorders, several pre-existing endocrinopathies such as type-1 diabetes and Grave's disease or adrenal failure may acutely deteriorate during pregnancy. Since "classical" signs are often modified by pregnancy, early diagnosis and management may be hampered. In addition, laboratory tests show altered physiologic ranges and pharmacologic options are limited while therapeutic goals are mostly tighter than in the non-pregnant patient. Though subclinical endocrinopathies are more frequent and worth consideration due to their related adverse sequelae, this article focuses on endocrine emergencies complicating pregnancy.
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Affiliation(s)
- Birgit Harbeck
- a Department of Medicine I , University of Lübeck , Lübeck , Germany and
| | | | - Sven Danneberg
- a Department of Medicine I , University of Lübeck , Lübeck , Germany and
| | - Morten Schütt
- a Department of Medicine I , University of Lübeck , Lübeck , Germany and
| | - Friedhelm Sayk
- b Department of Medicine II , University of Lübeck , Lübeck , Germany
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43
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Asadipooya K, Graves L, Greene LW. Transient osteoporosis of the hip: review of the literature. Osteoporos Int 2017; 28:1805-1816. [PMID: 28314897 DOI: 10.1007/s00198-017-3952-0] [Citation(s) in RCA: 67] [Impact Index Per Article: 8.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/08/2016] [Accepted: 01/30/2017] [Indexed: 11/26/2022]
Abstract
Transient osteoporosis of the hip (TOH) is a temporary clinical condition of unknown etiology which usually resolves with conservative therapy though may be complicated by fracture or progression to avascular necrosis (AVN). TOH may be slightly more prevalent in men but when it occurs in women, it is most often seen in the latter part of pregnancy. Though fracture is a rare complication of TOH when it occurs, it is most often associated with TOH occurring in pregnancy. Magnetic resonance imaging (MRI) is the best method to diagnosis TOH. Low signal intensity on T1-weighted images, high signal intensity on T2-weighted images, and homogenous pattern of edema (the femoral head and/or neck) with normal subchondral area are in favor of TOH. A shortened course to recovery is reported by use of bisphosphonates, calcitonin, or teriparatide. Based on reported cases, core decompression is not superior to medical therapy. Transient osteoporosis of the hip, which often has no known etiology, usually resolves with conservative therapy but may predispose the patient to fracture or avascular necrosis. Diagnostic method of choice is magnetic resonance imaging. Bisphosphonates, calcitonin, or teriparatide are reported as a useful approach to reduce duration of recovery.
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Affiliation(s)
- K Asadipooya
- Fellow in Endocrinology and Metabolism, NYU School of Medicine, New York, NY, USA
- Division of Endocrinology and Metabolism, NYU Langone Medical Center, 522 1st Ave, Smilow 707B, New York, NY, 10016-6402, USA
| | - L Graves
- Division of Metabolism, Endocrinology and Genetics, Department of Internal Medicine, Endocrinology and Genetics MS 2024, University of Kansas Medical Center, 3901 Rainbow Boulevard, Kansas City, KS, 66160, USA.
| | - L W Greene
- Medicine (Endocrinology) and ObGyn, Co-Director of Osteoporosis and Metabolic Bone Disease, Department of Medicine, NYU School of Medicine, New York, NY, USA
- Division of Endocrinology and Metabolism, NYU Langone Medical Center, 650 First Avenue, 7th floor, New York, NY, 10016, USA
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Hu Y, Cui M, Sun Z, Su Z, Gao X, Liao Q, Zhao Y. Clinical Presentation, Management, and Outcomes of Primary Hyperparathyroidism during Pregnancy. Int J Endocrinol 2017; 2017:3947423. [PMID: 29147113 PMCID: PMC5632860 DOI: 10.1155/2017/3947423] [Citation(s) in RCA: 16] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/14/2017] [Revised: 07/26/2017] [Accepted: 08/08/2017] [Indexed: 12/24/2022] Open
Abstract
BACKGROUND Primary hyperparathyroidism (pHPT) in pregnancy is a rare event, but it poses a significant risk to mothers and fetuses. The optimal treatment strategy remains controversial. METHODS We present a consecutive series of twelve pregnant women with pHPT. RESULTS Twelve women were diagnosed with pHPT during pregnancy or in the postpartum period. Four of them presented no symptoms or mild symptoms. Four patients experienced serious complications, including hypercalcaemic crisis, acute pancreatitis, and eclampsia. Another four patients were identified postpartum as the result of neonatal convulsion with hypocalcaemia. Minimally invasive parathyroidectomy (MIP) under cervical plexus block was successfully performed in 11 of them during pregnancy or postpartum. The serum levels of ionized calcium and intact parathyroid hormone (iPTH) were much higher in patients with severe complications in this cohort than those in the group of patients with no symptoms or mild symptoms and patients who were diagnosed postpartum. CONCLUSIONS MIP under cervical plexus block might be a safe and effective treatment for pregnant women with pHPT. Even though both conservative and surgical treatments are applicable for most mothers and fetuses with asymptomatic and mild hyperparathyroidism, serious complications may have catastrophic consequences for both.
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Affiliation(s)
- Ya Hu
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
| | - Ming Cui
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
| | - Zhengyi Sun
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
| | - Zhe Su
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
| | - Xiang Gao
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
| | - Quan Liao
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
| | - Yupei Zhao
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
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Vera L, Oddo S, Di Iorgi N, Bentivoglio G, Giusti M. Primary hyperparathyroidism in pregnancy treated with cinacalcet: a case report and review of the literature. J Med Case Rep 2016; 10:361. [PMID: 27998296 PMCID: PMC5175373 DOI: 10.1186/s13256-016-1093-2] [Citation(s) in RCA: 31] [Impact Index Per Article: 3.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/03/2016] [Accepted: 10/10/2016] [Indexed: 12/29/2022] Open
Abstract
BACKGROUND The efficacy and safety of various modes of medical treatment for primary hyperparathyroidism in pregnancy are largely unknown. CASE PRESENTATION We report the case of a 34-year-old white woman with primary hyperparathyroidism symptomatic for nephrolithiasis. Her serum calcium was 3.15 mmol/l and parathyroid hormone was 109.0 ng/L. Neck imaging found no pathological parathyroid tissue. Cinacalcet and cholecalciferol were started. She became pregnant 17 months later. The calcimimetic was stopped. During pregnancy, she was admitted for hydration administered intravenously two to three times per week. In her 24th week of pregnancy, cinacalcet was restarted. In her 32nd week, a cesarean section was carried out as planned. CONCLUSIONS Only three cases of primary hyperparathyroidism in women on cinacalcet therapy in pregnancy have been published in the literature. In the present case, hydration was useful in controlling serum calcium. Cinacalcet therapy helped to control serum calcium.
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Affiliation(s)
- Lara Vera
- Department of Internal Medicine, Genoa University, Viale Benedetto XV 6, 16132, Genoa, Italy
| | - Silvia Oddo
- Department of Internal Medicine, Genoa University, Viale Benedetto XV 6, 16132, Genoa, Italy
| | | | | | - Massimo Giusti
- Department of Internal Medicine, Genoa University, Viale Benedetto XV 6, 16132, Genoa, Italy.
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Woods GN, Saitman A, Gao H, Clarke NJ, Fitzgerald RL, Chi NW. A Young Woman With Recurrent Gestational Hypercalcemia and Acute Pancreatitis Caused by CYP24A1 Deficiency. J Bone Miner Res 2016; 31:1841-1844. [PMID: 27105398 PMCID: PMC5071127 DOI: 10.1002/jbmr.2859] [Citation(s) in RCA: 25] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/25/2016] [Revised: 04/11/2016] [Accepted: 04/20/2016] [Indexed: 12/29/2022]
Abstract
The CYP24A1 gene encodes a mitochondrial 24-hydroxylase that inactivates 1,25(OH)2 D. Loss-of-function mutations in CYP24A1 cause hypercalcemia, nephrolithiasis and nephrocalcinosis. We describe a woman with CYP24A1 deficiency and recurrent gestational hypercalcemia. Her first pregnancy, at age 20, resulted with the intrauterine demise of twin fetuses. Postpartum, she developed severe hypercalcemia (14 mg/dL), altered mental status, and acute pancreatitis. Her PTH was suppressed (6 pg/mL) and her 1,25(OH)2 D was elevated (165 and 195 pg/mL on postpartum day 1 and 5, respectively). Between one and three months postpartum, her serum calcium decreased from 11.4 to 10.2 mg/dL while her 1,25(OH)2 D level decreased from 83 to 24 pg/mL. Her 24-hour urine calcium was 277 mg. Six months postpartum, she became pregnant again. At 14 weeks, her albumin-corrected calcium level was 10.4 mg/dL and her 1,25(OH)2 D level exceeded 200 pg/mL. To establish the diagnosis of CYP24A1 deficiency, we showed her 24,25(OH)2 D level to be undetectable (<2 ng/mL). Exon sequencing of the CYP24A1 gene revealed a homozygous, 8-nucleotide deletion in exon 8, causing an S334V substitution and premature termination due to a frame shift (c.999_1006del, p.Ser334Valfs*9). To prevent hypercalcemia, she was advised to discontinue prenatal vitamins, avoid sun exposure and calcium-rich foods, and start omeprazole and a calcium binder (250 mg K-Phos-neutral with meals). Despite these measures, both hypercalcemia (11.5 mg/dL) and acute pancreatitis recurred. Labor was induced and a healthy, normocalcemic boy was delivered. In the absence of lactation, maternal hypercalcemia resolved within 2 months. This report shows that CYP24A1-deficient subjects may be normocalcemic at baseline. Hypercalcemia may be unmasked by pregnancy through the routine use of calciferol-containing prenatal vitamins, increased 1-alpha hydroxylation of VitD by the placenta and maternal kidney, and production of PTHrP by the uteroplacental unit. CYP24A1 deficiency should be considered in patients with unexplained vitamin D-mediated hypercalcemia. © 2016 American Society for Bone and Mineral Research.
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Affiliation(s)
- Gina N Woods
- Department of Medicine, University of California, San Diego, CA, USA.
| | - Alec Saitman
- Department of Pathology, University of California, San Diego, CA, USA
| | - Hanlin Gao
- Fulgent Diagnostics, Temple City, CA, USA
| | - Nigel J Clarke
- Mass Spectrometry R&D Department, Quest Diagnostics Nichols Institute, San Juan Capistrano, CA, USA
| | | | - Nai-Wen Chi
- Department of Medicine, University of California, San Diego, CA, USA
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Preeclampsia: A Possible Complication of Primary Hyperparathyroidism. Case Rep Obstet Gynecol 2016; 2016:7501263. [PMID: 27340578 PMCID: PMC4909923 DOI: 10.1155/2016/7501263] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/07/2016] [Revised: 04/27/2016] [Accepted: 05/05/2016] [Indexed: 12/26/2022] Open
Abstract
Background. Primary hyperparathyroidism is rare in pregnancy. An association between primary hyperparathyroidism and preeclampsia has been reported in few cases worldwide. Case. A 28-year-old woman (gravida 2, para 0, and abortus 1) in her 27th week of gestation was hospitalized due to a high reading of blood pressure (194/115 mmHg) that was not accompanied by any symptoms or signs of preeclampsia. Incidentally, she was found to have a high adjusted calcium and serum parathyroid hormone (PTH) level during admission. Ultrasonographic examination of the neck revealed the presence of parathyroid adenoma. She was scheduled for surgical excision after receiving an intravenous hydration. Fetal ultrasonography revealed a growth restricted fetus with normal biophysical profile. On the sixth day of hospitalization, the patient complained of headache and epigastric pain, with elevated BP and proteinuria. The fetal nonstress test was "nonreassuring." Subsequently, she had an emergency cesarean delivery and surgical removal of the adenoma. The mother and her newborn were then transferred to intensive care, where their clinical course was unremarkable. The mother was discharged after 3 days, while the neonate stayed for close observation for 60 days. Conclusion. Early recognition of primary hyperparathyroidism among women with preeclampsia is important to prevent maternal and fetal morbidity and mortality.
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Abstract
The metabolic pathways that contribute to maintain serum calcium concentration in narrow physiological range include the bone remodeling process, intestinal absorption and renal tubule resorption. Dysbalance in these regulations may lead to hyper- or hypocalcemia. Hypercalcemia is a potentionally life-threatening and relatively common clinical problem, which is mostly associated with hyperparathyroidism and/or malignant diseases (90 %). Scarce causes of hypercalcemia involve renal failure, kidney transplantation, endocrinopathies, granulomatous diseases, and the long-term treatment with some pharmaceuticals (vitamin D, retinoic acid, lithium). Genetic causes of hypercalcemia involve familial hypocalciuric hypercalcemia associated with an inactivation mutation in the calcium sensing receptor gene and/or a mutation in the CYP24A1 gene. Furthermore, hypercalcemia accompanying primary hyperparathyroidism, which develops as part of multiple endocrine neoplasia (MEN1 and MEN2), is also genetically determined. In this review mechanisms of hypercalcemia are discussed. The objective of this article is a review of hypercalcemia obtained from a Medline bibliographic search.
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Affiliation(s)
- I. ŽOFKOVÁ
- Institute of Endocrinology, Prague, Czech Republic
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49
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Clinical Manifestations of Primary Hyperparathyroidism. Updates Surg 2016. [DOI: 10.1007/978-88-470-5758-6_6] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
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