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Luong TV, Le LD, Nguyen NVD, Dang HNN. Persistent challenges in the diagnosis of acute pancreatitis due to primary hyperparathyroidism during pregnancy. World J Gastroenterol 2025; 31:100973. [PMID: 39991684 PMCID: PMC11755251 DOI: 10.3748/wjg.v31.i7.100973] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/05/2024] [Revised: 12/01/2024] [Accepted: 01/02/2025] [Indexed: 01/20/2025] Open
Abstract
In this manuscript, we provide critical commentary on the systematic review by Augustin et al, which investigated acute pancreatitis induced by primary hyperparathyroidism during pregnancy. Although this is an infrequent complication, it poses severe risks to both maternal and fetal health. Due to its infrequent occurrence in clinical practice, this review is based on an analysis of individual case reports over the past 55 years. While this is not the first study to utilize this sampling method for primary hyperparathyroidism-induced acute pancreatitis, it is unique in that it has a sufficiently large sample size with statistically significant results. Our discussion focuses on the diagnostic challenges associated with this condition, which are grounded in the mechanisms of parathyroid hormone secretion and variations in serum calcium levels. We also address the limitations of the current review and suggest potential strategies to increase diagnostic accuracy and improve health outcomes for both mothers and fetuses during pregnancy.
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Affiliation(s)
- Thang Viet Luong
- Department of Internal Medicine, University of Medicine and Pharmacy, Hue University, Hue 530000, Viet Nam
| | - Linh Duy Le
- Department of Internal Medicine, University of Medicine and Pharmacy, Hue University, Hue 530000, Viet Nam
| | - Nam Van Duc Nguyen
- Department of Internal Medicine, University of Medicine and Pharmacy, Hue University, Hue 530000, Viet Nam
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Lemaitre M, Picart C, Gueorguieva I, Charbit J, Edouard T, Linglart A, Luton D, Chanson P. Chapter 7: The different forms of primary hyperparathyroidism at different ages of life: Childhood, pregnancy, lactation, old age. ANNALES D'ENDOCRINOLOGIE 2025; 86:101696. [PMID: 39818293 DOI: 10.1016/j.ando.2025.101696] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/18/2025]
Abstract
Primary hyperparathyroidism is rare in children. A germline mutation is identified in half of all children with primary hyperparathyroidism (70% of newborns and infants, and 40% of children and adolescents). The clinical manifestations of primary hyperparathyroidism in children are highly variable (often absent in newborns, rather severe and symptomatic in children and adolescents) and depend on the genetic cause, as well as the severity, rapidity of onset and duration of hypercalcemia. Morphological investigation and treatment of children and adolescents follow the same rules as for adults. Surgery must be carried out by a surgeon expert in this pathology in children. Primary hyperparathyroidism is rarely diagnosed during pregnancy. Pregnancy-related changes in phosphocalcic homeostasis can sometimes mask its symptomatology, which explains why it is most often asymptomatic and/or undiagnosed due to low levels of hypercalcemia and/or the attribution of certain symptoms (e.g. vomiting) to pregnancy itself. Maternal-fetal morbidity associated with primary hyperparathyroidism during pregnancy, historically considered significant, is in fact rare and depends on maternal calcium levels. Treatment (conservative or surgical) must be adapted to the term of pregnancy, the severity of symptoms and maternal-fetal risks. Primary hyperparathyroidism is common in the elderly. Although the diagnostic approach is comparable to that in younger patients, some clinical specificities of this population need to be considered. In view of the high prevalence of osteoporotic fractures and their consequences for morbidity and mortality in the elderly, surgery is the preferred option in case of osteoporosis. In 2024, the neuropsychological symptoms and cardiovascular impairment associated with primary hyperparathyroidism do not justify parathyroidectomy. Parathyroidectomy is much less frequent than in the younger population, although it remains the first-line treatment, especially as its safety and efficacy have been widely demonstrated.
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Affiliation(s)
- Madleen Lemaitre
- Service d'endocrinologie, diabétologie, métabolisme, nutrition, hôpital Huriez, institut génomique européen pour le diabète, université de Lille, CHU de Lille, 59000 Lille, France
| | - Clotilde Picart
- Service de gériatrie, hôpital Ambroise-Paré, AP-HP, Boulogne-Billancourt, France
| | - Iva Gueorguieva
- Service de pédiatrie, hôpital Jeanne-de-Flandres, université de Lille, CHU de Lille, 59000 Lille, France
| | - Judith Charbit
- Gerontology Department, Broca Hospital, Université Paris-Cité, AP-HP, Paris, France
| | - Thomas Edouard
- UMR 1301-Inserm 5070-CNRS EFS, unité d'endocrinologie, maladies osseuses et génétique, hôpital des Enfants, institut RESTORE, université Paul-Sabatier, CHU de Toulouse, 31000 Toulouse, France
| | - Agnès Linglart
- Inserm, Endocrine Physiology and Physiopathology, service d'endocrinologie et de diabétologie pédiatrique, centre de référence des maladies rares du métabolisme du calcium et du phosphate, hôpital Bicêtre, université Paris-Saclay, Assistance publique-Hôpitaux de Paris, 94270 Le Kremlin-Bicêtre, France
| | - Dominique Luton
- Service de gynécologie-obstétrique, hôpital Bicêtre, université Paris-Saclay, Assistance publique-Hôpitaux de Paris, 94270 Le Kremlin-Bicêtre, France
| | - Philippe Chanson
- Inserm, Endocrine Physiology and Physiopathology, service d'endocrinologie et des maladies de la reproduction, centre de référence des maladies rares de l'hypophyse HYPO, hôpital Bicêtre, université Paris-Saclay, Assistance publique-Hôpitaux de Paris, 94270 Le Kremlin-Bicêtre, France.
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Sabbah N. Acute hypercalcemic crisis: A narrative review with a focus on pregnancy. ANNALES D'ENDOCRINOLOGIE 2024; 85:604-613. [PMID: 38880126 DOI: 10.1016/j.ando.2024.01.012] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 11/09/2023] [Revised: 01/16/2024] [Accepted: 01/20/2024] [Indexed: 06/18/2024]
Abstract
A hypercalcemic crisis is a rare therapeutic emergency. However, it should not be overlooked, particularly during pregnancy, as it is associated with significant maternal and fetal morbidity and mortality. The most frequent etiology, including in pregnant women, is primary hyperparathyroidism. Knowledge of calcium-phosphate metabolism during pregnancy is important for understanding and interpreting the clinicopathological abnormalities observed in parathyroid pathology. Despite the expert consensus statement on parathyroid pathology issued by the European Society of Endocrinology, management of hypercalcemic crises remains poorly codified, particularly in pregnant women. Diagnostic examinations and hypocalcemia treatments are generally not recommended during pregnancy; however, it may be necessary to optimize preparation for surgery. Notably, surgery is the treatment of choice, particularly during pregnancy, when it should ideally be performed during the 2nd trimester. Therefore, a multidisciplinary approach is necessary. A consensus among European experts recommends systematic early detection of hypercalcemia during early pregnancy.
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Affiliation(s)
- Nadia Sabbah
- Endocrinology Diabetology and Nutrition Department, Centre Hospitalier de Cayenne, avenue des Flamboyants, 97300 Cayenne, French Guiana.
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Luo Y, Lv Q, Xu Z, Fang J, Pu H, Gao Y, Qian S, Chen F, Zhao X, Hou L. Case report: Microwave ablation is a safe and effective method for primary hyperparathyroidism in pregnancy. Front Med (Lausanne) 2024; 11:1204696. [PMID: 38298816 PMCID: PMC10827987 DOI: 10.3389/fmed.2024.1204696] [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: 08/01/2023] [Accepted: 01/05/2024] [Indexed: 02/02/2024] Open
Abstract
Primary hyperparathyroidism (PHPT) is a rare disease in pregnancy and endangers the health of both pregnant women and fetuses. However, the treatments are very limited for PHPT and most of them are unsatisfactory because of the peculiar state in pregnancy. The only curable method is parathyroidectomy which can be safely performed in the second trimester of pregnancy. In this case, we reported a pregnant woman with primary parathyroid adenoma presenting hypercalcemia and severe vomit at the end of first trimester. Finally, she got cured by microwave ablation at the end of first trimester and gave birth to a healthy baby boy.
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Affiliation(s)
- Yunbo Luo
- Department of Thyroid and Breast Surgery, Affiliated Hospital of North Sichuan Medical College, Nanchong, China
| | - Qi Lv
- Department of Operating Room, Affiliated Hospital of North Sichuan Medical College, Nanchong, China
| | - Zhou Xu
- Department of Thyroid and Breast Surgery, Affiliated Hospital of North Sichuan Medical College, Nanchong, China
| | - Jiang Fang
- Department of Thyroid and Breast Surgery, Affiliated Hospital of North Sichuan Medical College, Nanchong, China
| | - Hongyu Pu
- Department of Thyroid and Breast Surgery, Affiliated Hospital of North Sichuan Medical College, Nanchong, China
| | - Yanchun Gao
- Department of Thyroid and Breast Surgery, Affiliated Hospital of North Sichuan Medical College, Nanchong, China
| | - Shuangqiang Qian
- Department of Thyroid and Breast Surgery, Affiliated Hospital of North Sichuan Medical College, Nanchong, China
| | - Fei Chen
- Department of Nuclear Medicine, Affiliated Hospital of North Sichuan Medical College, Nanchong, China
| | - Xiaobo Zhao
- Department of Thyroid and Breast Surgery, Affiliated Hospital of North Sichuan Medical College, Nanchong, China
| | - Lingmi Hou
- Department of Academician (Expert) Workstation, Biological Targeting Laboratory of Breast Cancer, Breast and Thyroid Surgery, Affiliated Hospital of North Sichuan Medical College, Nanchong, China
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Amara IA, Bula-Ibula D. [Diagnosis and management of primary hyperparathyroidism during pregnancy: A systematic review and a longitudinal case study]. GYNECOLOGIE, OBSTETRIQUE, FERTILITE & SENOLOGIE 2023; 51:531-537. [PMID: 37827286 DOI: 10.1016/j.gofs.2023.10.003] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 06/01/2023] [Revised: 09/28/2023] [Accepted: 10/03/2023] [Indexed: 10/14/2023]
Abstract
OBJECTIVE There is no specific recommendation for management in pregnant women: the aim of this review, based on a clinical case study, is to clarify its development, complications, risk factor and treatment. METHODS A review of the literature was performed by consulting the Pubmed, Cochrane Library, and Science Direct databases. RESULTS Primary hyperparathyroidism is defined as excessive production of parathyroid hormone resulting in hypercalcemia. The prevalence of primary hyperparathyroidism during pregnancy is not known. Indeed, the symptomatology, related to hypercalcemia, is not very specific and easily confused with the clinical manifestations of pregnancy. The physiological changes specific to the pregnant state frequently lead to a slight hypocalcemia which may complicate the diagnosis of primary hyperparathyroidism. Primary hyperparathyroidism results from a parathyroid adenoma in the majority of cases and is detected by ultrasound during pregnancy. Primary hyperparathyroidism in pregnancy causes significant risks to both mother and fetus. The maternal complication rate is 14-67%, however, the most serious complication is hypercalcemic crisis, which requires increased surveillance in the postpartum period. Obstetrical complications are also induced by primary hyperparathyroidism, such as acute polyhydramnios, or intrauterine growth retardation. The fetal complication rate can reach 45-80% of cases with neonatal hypocalcemia as the main complication. If medical treatment is based on hyperhydration, only surgical treatment is curative. CONCLUSION Surgery should be proposed to symptomatic patients or those with high blood calcium levels, discussed in interdisciplinary committee and should be organized ideally in the second trimester to avoid maternal and fetal complications.
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Affiliation(s)
- Inesse Ait Amara
- CHU de Brugmann, place A.-Van-Gehuchten 4, 1020 Bruxelles, Belgique.
| | - Diana Bula-Ibula
- CHU de Brugmann, place A.-Van-Gehuchten 4, 1020 Bruxelles, Belgique
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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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Bollerslev J, Rejnmark L, Zahn A, Heck A, Appelman-Dijkstra NM, Cardoso L, Hannan FM, Cetani F, Sikjaer T, Formenti AM, Björnsdottir S, Schalin-Jäntti C, Belaya Z, Gibb F, Lapauw B, Amrein K, Wicke C, Grasemann C, Krebs M, Ryhänen E, Makay Ö, Minisola S, Gaujoux S, Bertocchio JP, Hassan-Smith Z, Linglart A, Winter EM, Kollmann M, Zmierczak HG, Tsourdi E, Pilz S, Siggelkow H, Gittoes N, Marcocci C, Kamenický P, the 2021 PARAT Working Group. European Expert Consensus on Practical Management of Specific Aspects of Parathyroid Disorders in Adults and in Pregnancy: Recommendations of the ESE Educational Program of Parathyroid Disorders. Eur J Endocrinol 2022; 186:R33-R63. [PMID: 34863037 PMCID: PMC8789028 DOI: 10.1530/eje-21-1044] [Citation(s) in RCA: 94] [Impact Index Per Article: 31.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/13/2021] [Accepted: 12/03/2021] [Indexed: 11/17/2022]
Abstract
This European expert consensus statement provides recommendations for the diagnosis and management of primary hyperparathyroidism (PHPT), chronic hypoparathyroidism in adults (HypoPT), and parathyroid disorders in relation to pregnancy and lactation. Specified areas of interest and unmet needs identified by experts at the second ESE Educational Program of Parathyroid Disorders (PARAT) in 2019, were discussed during two virtual workshops in 2021, and subsequently developed by working groups with interest in the specified areas. PHPT is a common endocrine disease. However, its differential diagnosing to familial hypocalciuric hypercalcemia (FHH), the definition and clinical course of normocalcemic PHPT, and the optimal management of its recurrence after surgery represent areas of uncertainty requiring clarifications. HypoPT is an orphan disease characterized by low calcium concentrations due to insufficient PTH secretion, most often secondary to neck surgery. Prevention and prediction of surgical injury to the parathyroid glands are essential to limit the disease-related burden. Long-term treatment modalities including the place for PTH replacement therapy and the optimal biochemical monitoring and imaging surveillance for complications to treatment in chronic HypoPT, need to be refined. The physiological changes in calcium metabolism occurring during pregnancy and lactation modify the clinical presentation and management of parathyroid disorders in these periods of life. Modern interdisciplinary approaches to PHPT and HypoPT in pregnant and lactating women and their newborns children are proposed. The recommendations on clinical management presented here will serve as background for further educational material aimed for a broader clinical audience, and were developed with focus on endocrinologists in training.
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Affiliation(s)
- Jens Bollerslev
- Faculty of Medicine, University of Oslo, Oslo, Norway
- Section of Specialized Endocrinology, Department of Endocrinology, Medical Clinic, Oslo University Hospital, Oslo, Norway
- Correspondence should be addressed to J Bollerslev Email
| | - Lars Rejnmark
- Department of Endocrinology and Internal Medicine, Aarhus University Hospital, Aarhus, Denmark
| | - Alexandra Zahn
- Schön-Klinik Hamburg, Department of Endocrine Surgery, Hamburg, Germany
| | - Ansgar Heck
- Faculty of Medicine, University of Oslo, Oslo, Norway
- Section of Specialized Endocrinology, Department of Endocrinology, Medical Clinic, Oslo University Hospital, Oslo, Norway
| | - Natasha M Appelman-Dijkstra
- Division of Endocrinology, Department of Medicine, Leiden University Medical Center (LUMC), Leiden, the Netherlands
| | - Luis Cardoso
- Centro Hospitalar e Universitário de Coimbra, i3S – Instituto de Investigação e Inovação em Saúde da Universidade do Porto, Porto, Portugal
| | - Fadil M Hannan
- Nuffield Department of Women’s and Reproductive Health, University of Oxford, Oxford, UK
| | - Filomena Cetani
- Department of Clinical and Experimental Medicine, University of Pisa, Pisa, Italy
| | - Tanja Sikjaer
- Department of Endocrinology and Internal Medicine, Aarhus University Hospital, Aarhus, Denmark
| | - Anna Maria Formenti
- Institute of Endocrine and Metabolic Sciences, Vita-Salute San Raffaele University, IRCCS San Raffaele Hospital, Milan, Italy
| | - Sigridur Björnsdottir
- Department of Endocrinology, Metabolism and Diabetes, Karolinska University Hospital, Stockholm, Sweden
| | - Camilla Schalin-Jäntti
- Endocrinology, Abdominal Center, University of Helsinki and Helsinki University Hospital, Helsinki, Finland
| | - Zhanna Belaya
- The National Medical Research Centre for Endocrinology, Moscow, Russia
| | - Fraser Gibb
- Edinburgh Centre for Endocrinology & Diabetes, Royal Infirmary of Edinburgh, Edinburgh, UK
| | - Bruno Lapauw
- Department of Endocrinology, Ghent University Hospital, Ghent, Belgium
| | - Karin Amrein
- Division of Endocrinology and Diabetology, Medical University of Graz, Graz, Austria
| | - Corinna Wicke
- Thyroid Center, Luzerner Kantonsspital, Luzern, Switzerland
| | - Corinna Grasemann
- Division of Rare Diseases, Department of Pediatrics, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
| | - Michael Krebs
- Division of Endocrinology and Metabolism, Department of Internal Medicine III, Medical University of Vienna, Vienna, Austria
| | - Eeva Ryhänen
- Endocrinology, Abdominal Center, University of Helsinki and Helsinki University Hospital, Helsinki, Finland
| | - Özer Makay
- Division of Endocrine Surgery, Department of General Surgery, Ege University Hospital, Izmir, Turkey
| | - Salvatore Minisola
- Department of Internal Medicine and Medical Disciplines, Sapienza University of Rome, Rome, Italy
| | - Sébastien Gaujoux
- Department of Digestive, Hepatobiliary and Endocrine Surgery, Paris Descartes University, Cochin Hospital, Paris, France
| | - Jean-Philippe Bertocchio
- Assistance Publique-Hôpitaux de Paris (AP-HP), Pitié-Salpêtrière Hospital, Nephrology Department, Boulevard de l’Hôpital, Paris, France
| | - Zaki Hassan-Smith
- Department of Endocrinology, University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK
| | - Agnès Linglart
- Université de Paris Saclay, AP-HP, Centre de Référence des Maladies Rares du Métabolisme du Calcium et du Phosphate, Filière OSCAR, Service d’Endocrinologie et Diabète de l’Enfant, Hôpital Bicêtre Paris Saclay, Le Kremlin Bicêtre, France
| | - Elizabeth M Winter
- Division of Endocrinology, Department of Medicine, Leiden University Medical Center (LUMC), Leiden, the Netherlands
| | - Martina Kollmann
- Department of Obstetrics and Gynecology, Medical University of Graz, Graz, Austria
| | - Hans-Georg Zmierczak
- Reference Centre for Rare Bone, Calcium and Phosphate Disorders – University Hospital Ghent, Ghent, Belgium
| | - Elena Tsourdi
- Center for Healthy Aging, Department of Medicine III, Technische Universität Dresden Medical Center, Dresden, Germany
| | - Stefan Pilz
- Division of Endocrinology and Diabetology, Medical University of Graz, Graz, Austria
| | - Heide Siggelkow
- Endokrinologikum Göttingen, Georg-August-University Göttingen, Göttingen, Germany
| | - Neil Gittoes
- Department of Endocrinology, University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK
| | - Claudio Marcocci
- Department of Clinical and Experimental Medicine, University of Pisa, Pisa, Italy
| | - Peter Kamenický
- Université Paris-Saclay, Inserm, Physiologie et Physiopathologie Endocriniennes, Assistance Publique-Hôpitaux de Paris, Hôpital Bicêtre, Service d’Endocrinologie et des Maladies de la Reproduction, Centre de Référence des Maladies Rares du Métabolisme du Calcium et du Phosphate, Le Kremlin-Bicêtre, France
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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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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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10
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Nastos C, Paspala A, Mavroeidi I, Stavratis F, Lampadiari V, Kalantaridou S, Peppa M, Pikoulis E. Surgical management of primary hyperparathyroidism during pregnancy: a systematic review of the literature. Gynecol Endocrinol 2021; 37:1086-1095. [PMID: 34044722 DOI: 10.1080/09513590.2021.1932801] [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: 10/21/2022] Open
Abstract
OBJECTIVE Gestational primary hyperparathyroidism (PHPT) is an endocrinological disorder with serious outcomes for both women and neonates. The aim of our study was to present the current evidence concerning the perioperative outcomes of pregnant women with PHPT who underwent parathyroidectomy during pregnancy. METHODS A meticulous systematic review of the literature published before February 2020 and all studies which presented perioperative and pregnancy outcomes off pregnant women who underwent parathyroidectomy for PHPT, were included. RESULTS A total of 53 were finally included, which reported 92 pregnant women who had parathyroidectomy during their pregnancy. A total of 46 patients were hospitalized due to significant complications of PHPT before their parathyroidectomy. With regards to surgical approach, 52.2% of patients underwent minimally invasive parathyroidectomy (MIP), while bilateral neck exploration (BNE) was 41.3% of cases. Only 4 women was not cured, whereas transient hypocalcemia was occurred in 18 patients. All cases proceeded to deliveries of healthy neonates, after their parathyroidectomy. CONCLUSIONS Parathyroidectomy during pregnancy is a safe and effective treatment option with minimum complications and probably should be considered as the treatment of choice in specific group of pregnant women with PHPT.
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Affiliation(s)
- Constantinos Nastos
- 3rd Department of Surgery, National and Kapodistrian University of Athens, Attikon University Hospital, Athens, Greece
| | - Anna Paspala
- 3rd Department of Surgery, National and Kapodistrian University of Athens, Attikon University Hospital, Athens, Greece
| | - Ioanna Mavroeidi
- Endocrine Unit, 2nd Propaedeutic Department of Internal Medicine & Research Institute, National & Kapodistrian University of Athens, Attikon University Hospital, Athens, Greece
| | - Fotios Stavratis
- 3rd Department of Surgery, National and Kapodistrian University of Athens, Attikon University Hospital, Athens, Greece
| | - Vaia Lampadiari
- 2nd Propaedeutic Department of Internal Medicine & Research Institute, National & Kapodistrian University of Athens, Attikon University Hospital, Athens, Greece
| | - Sophia Kalantaridou
- 3rd Department of Gynaecology and Obstetrics, Attikon Hospital, Medical School, National and Kapodistrian University of Athens, Athens, Greece
| | - Melpomeni Peppa
- Endocrine Unit, 2nd Propaedeutic Department of Internal Medicine & Research Institute, National & Kapodistrian University of Athens, Attikon University Hospital, Athens, Greece
| | - Emmanuil Pikoulis
- 3rd Department of Surgery, National and Kapodistrian University of Athens, Attikon University Hospital, Athens, Greece
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11
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Pal R, Bhadada SK, Gupta N, Behera A, Aggarwal N, Aggarwal A, Raviteja KV, Saikia UN, Kaur G, Arvindbhai SM, Walia R. Primary hyperparathyroidism in pregnancy: observations from the Indian PHPT registry. J Endocrinol Invest 2021; 44:1425-1435. [PMID: 33037580 DOI: 10.1007/s40618-020-01441-z] [Citation(s) in RCA: 14] [Impact Index Per Article: 3.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/14/2020] [Accepted: 09/29/2020] [Indexed: 01/12/2023]
Abstract
PURPOSE To present the data on primary hyperparathyroidism (PHPT) in pregnancy from India obtained from a large database maintained over 15 years. METHODS We retrieved data of all women with gestational PHPT from the Indian PHPT registry between July 2005 and January 2020, and compared their clinical, biochemical, and other characteristics with age-matched non-pregnant women with PHPT. RESULTS Out of 386 women, eight had gestational PHPT (2.1%). The common presenting manifestations were acute pancreatitis (50%) and renal stone disease (50%); two were asymptomatic. Five women (62.5%) had a history of prior miscarriages. Seven patients (88%) had preeclampsia during the present gestation. Serum calcium and intact parathyroid hormone (iPTH) were not statistically different from the age-matched non-pregnant PHPT group. Six patients with mild-to-moderate hypercalcemia were medically managed with hydration with/without cinacalcet while one patient underwent percutaneous ethanol ablation of the parathyroid adenoma; none underwent surgery during pregnancy. Mean serum calcium maintained from treatment initiation till delivery was 10.5 ± 0.4 mg/dl. One patient had spontaneous preterm delivery at 36 weeks; the remaining patients had normal vaginal delivery at term. None had severe preeclampsia/eclampsia. Fetal outcomes included low birth weight in three newborns (37.5%); two of them had hypocalcemic seizures. CONCLUSION The prevalence of gestational PHPT was 2.1% in this largest Indian PHPT cohort, which is higher than that reported from the West (< 1%). Gestational PHPT can lead to preeclampsia and miscarriage. Pregnant PHPT patients with mild-to-moderate hypercalcemia can be managed with hydration/cinacalcet; however, long-term safety data and large-scale randomized controlled trials are required.
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Affiliation(s)
- R Pal
- Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India
| | - S K Bhadada
- Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India.
| | - N Gupta
- Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India
| | - A Behera
- Department of General Surgery, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India
| | - N Aggarwal
- Department of Gynecology and Obstetrics, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India
| | - A Aggarwal
- Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India
| | - K V Raviteja
- Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India
| | - U N Saikia
- Department of Histopathology, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India
| | - G Kaur
- Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India
| | - S M Arvindbhai
- Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India
| | - R Walia
- Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, 160012, India
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12
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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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13
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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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14
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Horton WB, Stumpf MM, Coppock JD, Lancaster L, Dalkin AC, Liu Z, Chisholm CA, Smith PW, Kirk SE. Gestational Primary Hyperparathyroidism Due to Ectopic Parathyroid Adenoma: Case Report and Literature Review. J Endocr Soc 2017; 1:1150-1155. [PMID: 29264569 PMCID: PMC5686630 DOI: 10.1210/js.2017-00172] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/29/2017] [Accepted: 06/26/2017] [Indexed: 02/07/2023] Open
Abstract
Gestational primary hyperparathyroidism (GPHPT) is a rare condition with fewer than 200 cases reported. We present the case of a 21-year-old woman who presented at 10 weeks’ gestation with severe hypercalcemia. Laboratory investigation was consistent with primary hyperparathyroidism. Neck ultrasound did not reveal any parathyroid enlargement. Due to the persistence of severe hypercalcemia, she was treated with 4 weeks of cinacalcet therapy, which was poorly tolerated due to nausea and vomiting. At 14 weeks’ gestation, she underwent neck exploration with right lower, left upper, and partial right upper parathyroid gland excision. Intra- and postoperative parathyroid hormone (PTH) and calcium levels remained elevated. After a thorough discussion of risks/benefits, the patient requested further treatment. A parathyroid sestamibi scan (PSS) revealed an ectopic adenoma in the left mediastinum. The adenoma was removed via video-assisted thorascopic parathyroidectomy with intraoperative PTH declining to nearly undetectable levels. She ultimately delivered a physically and developmentally normal infant at 37 weeks’ gestation. Appropriate treatment of severe GPHPT may prevent the maternal and fetal complications of hypercalcemia. This case, in which cinacalcet therapy and PSS were used, adds to the body of literature regarding treatment of severe GPHPT.
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Affiliation(s)
- William B Horton
- Division of Endocrinology and Metabolism, Department of Medicine, University of Virginia Health System, Charlottesville, Virginia 22903
| | - Meaghan M Stumpf
- Division of Endocrinology and Metabolism, Department of Medicine, University of Virginia Health System, Charlottesville, Virginia 22903
| | - Joseph D Coppock
- Department of Pathology, University of Virginia Health System, Charlottesville, Virginia 22903
| | - Luke Lancaster
- Division of Nuclear Medicine, Department of Radiology and Medical Imaging, University of Virginia Health System, Charlottesville, Virginia 22903
| | - Alan C Dalkin
- Division of Endocrinology and Metabolism, Department of Medicine, University of Virginia Health System, Charlottesville, Virginia 22903
| | - Zhenqi Liu
- Division of Endocrinology and Metabolism, Department of Medicine, University of Virginia Health System, Charlottesville, Virginia 22903
| | - Christian A Chisholm
- Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, University of Virginia Health System, Charlottesville, Virginia 22903
| | - Philip W Smith
- Division of General Surgery, Department of Surgery, University of Virginia Health System, Charlottesville, Virginia 22903
| | - Susan E Kirk
- Division of Endocrinology and Metabolism, Department of Medicine, University of Virginia Health System, Charlottesville, Virginia 22903
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