scholarly journals Primary hyperparathyroidism and Zollinger Ellison syndrome during pregnancy: a case report

2021 ◽  
Vol 2021 ◽  
Author(s):  
Dalal Ali ◽  
Patrick Divilly ◽  
Ruth Prichard ◽  
Dermot O’Toole ◽  
Donal O’Shea ◽  
...  
Keyword(s):  
Primary Hyperparathyroidism ◽  
Unplanned Pregnancy ◽  
Case Series ◽  
Medical Attention ◽  
High Dose ◽  
List Type ◽  
Childbearing Age ◽  
Zollinger Ellison Syndrome ◽  
In Pregnancy

Summary Multiple endocrine neoplasia type 1 (MEN1) is a rare inherited endocrine disorder with a high rate of penetrance. The incidence of MEN1 is 1/30,000 in the general population; however, it is quite rare for a patient to present for medical attention with MEN1 for the first time in pregnancy. Primary hyperparathyroidism (PHPT) is one of the most common features of MEN1. The incidence of PHPT occurring in pregnancy is 1%. Despite advances in the medical, surgical and obstetric care over the years, management of this condition during pregnancy may be challenging. It can be difficult to identify pregnant women with PHPT requiring intervention and to monitor safely. Hypercalcemia can result in significant maternal and fetal adverse outcomes including: miscarriage, intrauterine growth restriction, preterm delivery, neonatal hypocalcaemia, pre-eclampsia and maternal nephrolithiasis. Herein, we present a case study of a lady with a strong family history of MEN1, who was biochemically proven to have PHPT and evidence of Zollinger Ellison Syndrome (ZE) on endoscopy. This patient delayed her assisted pregnancy plans for in vitro fertilization (IVF) until completion of the MEN1 workup; nevertheless, she spontaneously achieved an unplanned pregnancy. As a result, she required intervention with parathyroidectomy in the second trimester of her pregnancy as her calcium level continued to rise. This case study highlights the workup, follow up and management of MEN1 presenting with PHPT and ZE in pregnancy. Learning points Women of childbearing age who are suspected to have a diagnosis of primary hyperparathyroidism ideally should have genetic testing and avoid pregnancy until definitive plans are in place. Zollinger Ellison syndrome in pregnancy means off-label use of high dose of proton pump inhibitors (PPI). Use of PPI in pregnancy is considered to be safe based on retrospective studies. Omeprazole, however, is FDA class C drug because of lack of large prospective studies or large case series during pregnancy. Calcium supplements in the form of calcium carbonate must be converted to calcium chloride by gastric acid in order to be absorbed, however, patients rendered achlorhydric as a result of PPI use will have impaired absorption of calcium. Therefore, use of calcium citrate might be considered a better option in this case.

scholarly journals Management of primary hyperparathyroidism in pregnancy: a case series

2019 ◽  
Vol 2019 ◽  
Author(s):  
Aisling McCarthy ◽  
Sophie Howarth ◽  
Serena Khoo ◽  
Julia Hale ◽  
Sue Oddy ◽  
...  
Keyword(s):  
Primary Hyperparathyroidism ◽  
Serum Calcium ◽  
Case Series ◽  
Definitive Treatment ◽  
List Type ◽  
Second Trimester ◽  
Management Options ◽  
Neonatal Complications ◽  
Fetal Complications ◽  
In Pregnancy

Summary Primary hyperparathyroidism (PHPT) is characterised by the overproduction of parathyroid hormone (PTH) due to parathyroid hyperplasia, adenoma or carcinoma and results in hypercalcaemia and a raised or inappropriately normal PTH. Symptoms of hypercalcaemia occur in 20% of patients and include fatigue, nausea, constipation, depression, renal impairment and cardiac arrythmias. In the most severe cases, uraemia, coma or cardiac arrest can result. Primary hyperparathyroidism in pregnancy is rare, with a reported incidence of 1%. Maternal and fetal/neonatal complications are estimated to occur in 67 and 80% of untreated cases respectively. Maternal complications include nephrolithiasis, pancreatitis, hyperemesis gravidarum, pre-eclampsia and hypercalcemic crises. Fetal complications include intrauterine growth restriction; preterm delivery and a three to five-fold increased risk of miscarriage. There is a direct relationship between the degree of severity of hypercalcaemia and miscarriage risk, with miscarriage being more common in those patients with a serum calcium greater than 2.85 mmol/L. Neonatal complications include hypocalcemia. Herein, we present a case series of three women who were diagnosed with primary hyperparathyroidism in pregnancy. Case 1 was diagnosed with multiple endocrine neoplasia type 1 (MEN1) in pregnancy and required a bilateral neck exploration and subtotal parathyroidectomy in the second trimester of her pregnancy due to symptomatic severe hypercalcaemia. Both case 2 and case 3 were diagnosed with primary hyperparathyroidism due to a parathyroid adenoma and required a unilateral parathyroidectomy in the second trimester. This case series highlights the work-up and the tailored management approach to patients with primary hyperparathyroidism in pregnancy. Learning points: Primary hyperparathyroidism in pregnancy is associated with a high incidence of associated maternal fetal and neonatal complications directly proportionate to degree of maternal serum calcium levels. Parathyroidectomy is the definitive treatment for primary hyperparathyroidism in pregnancy and was used in the management of all three cases in this series. It is recommended when serum calcium is persistently greater than 2.75 mmol/L and or for the management of maternal or fetal complications of hypercalcaemia. Surgical management, when necessary is ideally performed in the second trimester. Primary hyperparathyroidism is genetically determined in ~10% of cases, where the likelihood is increased in those under 40 years, where there is relevant family history and those with other related endocrinopathies. Genetic testing is a useful diagnostic adjunct and can guide treatment and management options for patients diagnosed with primary hyperparathyroidism in pregnancy, as described in case 1 in this series, who was diagnosed with MEN1 syndrome. Women of reproductive age with primary hyperparathyroidism need to be informed of the risks and complications associated with primary hyperparathyroidism in pregnancy and pregnancy should be deferred and or avoided until curative surgery has been performed and calcium levels have normalised.

Primary hyperparathyroidism in pregnancy: a case series and literature review

2016 ◽  
Vol 32 (10) ◽  
pp. 783-786 ◽  
Author(s):  
Naile Gokkaya ◽  
Adem Gungor ◽  
Arzu Bilen ◽  
Habib Bilen ◽  
Darina Gviniashvili ◽  
...  
Keyword(s):  
Literature Review ◽  
Primary Hyperparathyroidism ◽  
Case Series ◽  
In Pregnancy

scholarly journals Primary hyperparathyroidism in pregnancy: a case series

2015 ◽  
Author(s):  
Naile Gokkaya ◽  
Adem Gungor ◽  
Arzu Bilen ◽  
Habib Bilen
Keyword(s):  
Primary Hyperparathyroidism ◽  
Case Series ◽  
In Pregnancy

scholarly journals Pre-Eclampsia Strikes: Could It Be Related This Gland?

2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A783-A784
Author(s):  
Jason Lofters ◽  
Nia Flemming ◽  
Latoya Gayle ◽  
Ro-Kaye Simmonds ◽  
Oluwafeyi Adedoyin ◽  
...  
Keyword(s):  
Weight Gain ◽  
Primary Hyperparathyroidism ◽  
Serum Calcium ◽  
Case Reports ◽  
Case Series ◽  
Stable Condition ◽  
Fetal Lung ◽  
Gradual Improvement ◽  
Normal Urine ◽  
In Pregnancy

Abstract Introduction: Primary hyperparathyroidism (PHPT) is rarely diagnosed in pregnancy and if left untreated has the potential to lead to serious maternal and neonatal complications. We describe a case of PHPT with associated complicated pre-eclampsia. Clinical Case29-year-old primigravida admitted at 33 + 6 weeks with fatigue, 10lbs weight gain and elevated BP. Labs revealed potassium 2.9 (3.5-5.2mmol/L), corrected serum calcium (Ca)11.62 (8.4-10.2mg/dL), ionized calcium 1.69 (1.15-1.33mmol/L), PTH 163.9 (15-65pg/mL) and vitamin D 24.6 (30-100ng/mL). Other labs were normal. Urine studies showed 315mg/24h proteinuria and urine calcium of 129.5 mg/24hrs (100-300mg/24hrs). She was started on magnesium sulphate along with labetalol for BP control, given betamethasone for stimulation of fetal lung maturity as well as potassium repletion. Hypercalcemia (HCa) was initially managed with fluids and Lasix intravenously. At 34 + 2 weeks she developed SOB, orthopnea, headaches with new 9lbs weight gain over 5 days and sustained BP elevation. Urgent C-section was done for pre-eclampsia with severe features. Post-operatively, she suffered from postpartum hemorrhage, managed with transfusion of packed red cells and transient placement of a Bakri balloon. Her HCa worsened with Ca 12.56 and cinacalcet was started after delivery. This coincided with gradual improvement of her BP and Ca to 10.8. She declined additional work-up and was discharged in stable condition. Clinical LessonPHPT often goes undiagnosed in pregnancy, with symptoms of fatigue and constipation mimicking common complaints of pregnancy. Studies have also suggested that up to 25% of patients with PHPT during pregnancy present with hypertension and pre-eclampsia and that there is an association between preeclampsia and the presence of parathyroid adenomas. The pathophysiology is unclear but is thought to be due to endothelial dysfunction triggered by hypercalcemia as well as abnormal placentation. No clear guidelines exist for the management of PHPT during pregnancy, with observation and rehydration being the preferred initial options. The use of cinacalcet as well as curative surgical parathyroidectomy when Ca levels persist >11 in the second trimester have also been described. Our patient presented similarly, with severe pre-eclampsia needing urgent C-section, further complicated by persistent severe HCa. Early diagnosis of PHPT, along with treatment including cinacalcet improved her Ca. It is therefore important that PHPT be considered in patients presenting like ours, progressing to severe pre-eclampsia as early reduction of serum calcium may reduce morbidity and mortality. ReferencesMcCarthy, A., Howarth, S., Khoo, S., Hale, J., Oddy, S., Halsall, D., ... & Samyraju, M. (2019). Management of primary hyperparathyroidism in pregnancy: a case series. Endocrinology, diabetes & metabolism case reports, 2019(1).

scholarly journals The use of cinacalcet in pregnancy to treat a complex case of parathyroid carcinoma

2014 ◽  
Vol 2014 ◽  
Author(s):  
K Nadarasa ◽  
M Bailey ◽  
H Chahal ◽  
O Raja ◽  
R Bhat ◽  
...  
Keyword(s):  
Parathyroid Carcinoma ◽  
Thyroid Tissue ◽  
Complex Case ◽  
High Dose ◽  
List Type ◽  
Radiological Findings ◽  
Chronic Knee Pain ◽  
Healthy Infants ◽  
Learning Points ◽  
In Pregnancy

Summary We present the case of a patient with metastatic parathyroid carcinoma whose hypercalcaemia was medically managed through two pregnancies. The diagnosis was made when the patient presented with chronic knee pain and radiological findings consistent with a brown tumour, at the age of 30. Her corrected calcium and parathyroid hormone (PTH) levels were significantly elevated. Following localisation studies, a right parathyroidectomy was performed with histology revealing parathyroid carcinoma, adherent to thyroid tissue. Aged 33, following biochemical recurrence of disease, the patient underwent a second operation. A subsequent CT and FDG–PET revealed bibasal pulmonary metastases. Aged 35, the patient was referred to our unit for treatment of persistent hypercalcaemia. The focus of treatment at this time was debulking metastatic disease using radiofrequency ablation. Despite advice to the contrary, the patient conceived twice while taking cinacalcet. Even though there are limited available data regarding the use of cinacalcet in pregnancy, both pregnancies continued to term with the delivery of healthy infants, using intensive medical management for persistent hypercalcaemia. Learning points Parathyroid carcinoma is a rare cause of primary hyperparathyroidism. Hypercalcaemia during pregnancy can result in significant complications for both the mother and the foetus. The use of high-dose cinacalcet in pregnancy has been shown, in this case, to aid in the management of resistant hypercalcaemia without teratogenicity.

Primary Hyperparathyroidism in Pregnancy: A Case Series and Review

2008 ◽  
Vol 118 (11) ◽  
pp. 1966-1969 ◽  
Author(s):  
Mai Thy Truong ◽  
M Lauren Lalakea ◽  
Paul Robbins ◽  
Michael Friduss
Keyword(s):  
Primary Hyperparathyroidism ◽  
Case Series ◽  
In Pregnancy

scholarly journals Primary Hyperparathyroidism in Pregnancy: Literature Review of the Diagnosis and Management

2021 ◽  
Vol 10 (13) ◽  
pp. 2956
Author(s):  
Dalal S. Ali ◽  
Karel Dandurand ◽  
Aliya A. Khan
Keyword(s):  
Primary Hyperparathyroidism ◽  
Case Reports ◽  
Safety Data ◽  
Case Series ◽  
Close Monitoring ◽  
Management Options ◽  
Diagnosis And Management ◽  
Severe Hypercalcemia ◽  
Fetal Complications ◽  
In Pregnancy

Background: Parathyroid disease is uncommon in pregnancy. During pregnancy, multiple changes occur in the calcium regulating hormones which may make the diagnosis of primary hyperparathyroidism more challenging. Close monitoring of serum calcium during pregnancy is necessary in order to optimize maternal and fetal outcomes. In this review, we will describe the diagnosis and management of primary hyperparathyroidism during pregnancy. Methods: We searched MEDLINE, CINAHL, EMBASE and Google scholar bases from 1 January 1990 to 31 December 2020. Case reports, case series, book chapters and clinical guidelines were included in this review. Conclusions: Medical management options for primary hyperparathyroidism during pregnancy are severely limited due to inadequate safety data with the various potential therapies available, and surgery is advised during the 2nd trimester of pregnancy in the presence of severe hypercalcemia (calcium adjusted for albumin greater than 3.0 mmol/L (12.0 mg/dL)). Hypercalcemia should be avoided during pregnancy in order to minimize maternal and fetal complications.

How to treat primary hyperparathyroidism in pregnancy? A case series

2020 ◽  
Author(s):  
Gastelum Alheli Arce ◽  
Azka Latif ◽  
Kinaan Farhan ◽  
Sangeeta Mutnuri
Keyword(s):  
Primary Hyperparathyroidism ◽  
Case Series ◽  
In Pregnancy

scholarly journals Unplanned pregnancy on a direct oral anticoagulant (Rivaroxaban): A warning

2015 ◽  
Vol 9 (1) ◽  
pp. 40-42 ◽  
Author(s):  
B Myers ◽  
R Neal ◽  
O Myers ◽  
M Ruparelia
Keyword(s):  
Inhibitory Action ◽  
Unplanned Pregnancy ◽  
Case Reports ◽  
Oral Anticoagulants ◽  
Direct Oral Anticoagulants ◽  
Factor X ◽  
Childbearing Age ◽  
Factor Ii ◽  
Pregnancy And Delivery ◽  
In Pregnancy

Direct oral anticoagulants (DOACs or NOACs -non-vitamin K oral anticoagulants), as the name suggests, are oral anticoagulants with a direct inhibitory action either against factor X or factor II (thrombin). Pregnant women were excluded from participating in all the large trials of the DOACs and they are considered contra-indicated in pregnancy and breast feeding. We present a case of inadvertent exposure to rivaroxaban in a woman who presented at 25 weeks' gestation. The management of her pregnancy and delivery is described, and the previous published case reports are reviewed with a discussion about the use of DOACs in woman of childbearing age.

scholarly journals Multidisciplinary team efforts to improve the pregnancy outcome of pregnancy complicated with primary hyperparathyroidism: case series from a single hospital

2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Hai-ning Jiao ◽  
Li-hao Sun ◽  
Yan Liu ◽  
Jian-qiao Zhou ◽  
Xi Chen ◽  
...  
Keyword(s):  
Primary Hyperparathyroidism ◽  
Multidisciplinary Team ◽  
Clinical Manifestations ◽  
Treatment Strategies ◽  
Case Series ◽  
Retrospective Case ◽  
Infant Outcomes ◽  
Management Algorithm ◽  
Preoperative Serum ◽  
In Pregnancy

Abstract Background There is no consensus or management algorithm for primary hyperparathyroidism (PHPT) in pregnancy. Methods This study comprises a retrospective case series. From August 2014 to December 2020, 9 cases of PHPT in pregnancy were diagnosed by a multidisciplinary team (MDT) consultation center of obstetrics in our hospital. Their clinical manifestations, treatment strategies, and maternal and infant outcomes were analyzed. Results The median onset age of the patients was 32 (25 ~ 38) years. PHPT was diagnosed in two cases before pregnancy, in six cases during pregnancy and in one case postpartum. The main clinical manifestations were nausea, vomiting, and other nonspecific symptoms, with anemia as the most common maternal complication. Hypercalcemia crisis was developed in one case. The median levels of preoperative serum calcium and parathyroid hormone (PTH) were 3.08 (2.77 ~ 4.21) mmol/L and 300.40 (108.80 ~ 2603.60) pg/ml, respectively. The parathyroid ultrasonography tests were positive in eight cases and negative in one patient who had an ectopic lesion localized by 99mTc-MIBI. Parathyroidectomy was conducted in 7 cases during the 2nd trimester, including 2 patients diagnosed before pregnancy who refused surgery, 1 patient during the 1st trimester, and 1 patient postpartum, with a significant reduction in serum concentrations of calcium and PTH. A management algorithm was developed. Conclusion This case series suggests that pregnant women with PHPT should be managed by MDT according to the algorithm. If PHPT is confirmed in fertile women before pregnancy, parathyroidectomy should be strongly suggested and performed. If PHPT is diagnosed during pregnancy, even in its mild form, surgical treatment, optimally during the 2nd trimester, is effective and safe for pregnancy and neonatal outcome.

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