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BACKGROUND Benign parathyroid adenoma is a cause of hypercalcemia, which can lead to acute pancreatitis. Patients with acute pancreatitis are at risk for venous thrombosis. This report describes a 34-year-old woman with hypercalcemia due to parathyroid adenoma and acute pancreatitis associated with splenic vein and superior mesenteric vein thrombosis. CASE REPORT A previously healthy 34-year-old woman presented with severe epigastric pain that radiated to the back, associated with vomiting. Her abdominal examination was soft and lax, with epigastric and left upper quadrant tenderness. Pancreatitis with splenic and superior mesenteric veins thrombosis was diagnosed. The diagnosis was confirmed by an elevated serum lipase level and contrast-enhanced computed tomography (CT) of abdomen. Her serum calcium level was elevated. However, further workup revealed elevated parathyroid hormone (PTH) levels and radiological imaging showed parathyroid adenoma. She was diagnosed with hypercalcemia-induced pancreatitis secondary to hyperparathyroidism with intraabdominal venous thrombosis. The patient was initially treated conservatively, and later underwent parathyroidectomy after her condition was stabilized. The patient is currently in good condition, after a 2-year follow-up period. CONCLUSIONS Acute pancreatitis and thrombosis secondary to primary hyperparathyroidism (PHPT) are rare, but can lead to potentially fatal complications, especially in patients without symptoms of PHPT. This report highlights the importance of recognizing that hypercalcemia associated with parathyroid adenoma can result in acute pancreatitis, leading to hypercoagulable states and inflammation of adjacent vessels, including the splenic and mesenteric veins. To the best of our knowledge, this is second case report of acute pancreatitis with intraabdominal venous thrombosis secondary to PHPT.

Background: Multiple endocrine neoplasias (MENs) are a group of hereditary diseases involving multiple endocrine glands, and their prevalence is low. MEN type 1 (MEN1) has diverse clinical manifestations, mainly involving the parathyroid glands, gastrointestinal tract, pancreas and pituitary gland, making it easy to miss the clinical diagnosis., Case Summary: We present the case of a patient in whom MEN1 was detected early. A middle-aged male with recurrent abdominal pain and diarrhea was admitted to the hospital. Blood tests at admission revealed hypercalcemia and hypophosphatemia, and emission computed tomography of the parathyroid glands revealed a hyperfunctioning parathyroid lesion. Gastroscopy findings suggested a duodenal bulge and ulceration. Ultrasound endoscopy revealed a hypoechoic lesion in the duodenal bulb. Further blood tests revealed elevated levels of serum gastrin. Surgery was performed, and pathological analysis of the surgical specimens revealed a parathyroid adenoma after parathyroidectomy and a neuroendocrine tumor after duodenal bulbectomy. The time from onset to the definitive diagnosis of MEN1 was only approximately 1 year., Conclusion: For patients who present with gastrointestinal symptoms accompanied by hypercalcemia and hypophosphatemia, clinicians need to be alert to the possibility of MEN1., Competing Interests: Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article., (©The Author(s) 2024. Published by Baishideng Publishing Group Inc. All rights reserved.)

Parathyroid carcinoma (PC) is usually associated with severe symptomatic primary hyperparathyroidism (PHPT) and accounts for less than 1% of all cases of PHPT and approximately 0.005% of all cancers. PC most commonly occurs as a sporadic disease and somatic CDC73 mutations can be detected in up to 80% of cases. Approximately 30% of patients harbor a germline mutation of the CDC73 gene. Preoperative diagnosis of PC is difficult because no disease-specific markers are available, and PC should be suspected in patients with severe hypercalcemia and end-organ complications. The diagnosis is based on the evidence of invasive tumor growth at histology and/or metastases. En bloc resection of the tumor, together with the ipsilateral thyroid lobe and adjacent structures, should be performed by an experienced surgeon when PC is suspected. This surgical approach reduces the risk of recurrence and metastasis and offers the highest chance of cure. Nonetheless, PC has a recurrence rate of 40% to 60% and, if feasible, multiple surgical procedures should be performed. When surgery is no longer an option, medical treatment is aimed to reduce hypercalcemia and target organ complications. Targeted agents have been effectively used in a few cases. We describe herein a patient with severe PHPT due to PC and provide a systematic diagnostic and treatment approach. A thorough review of the medical history, a typical clinical and biochemical phenotype and, in some cases, the revision of the histological examination provide the clues for the diagnosis of PC., (© The Author(s) 2023. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.)

BACKGROUND Parathyroid carcinoma represents about 0.005% of all malignancies and accounts for less than 1% of primary hyperparathyroidism cases. Precise preoperative diagnosis of parathyroid carcinoma is challenging, and it is usually diagnosed postoperatively by histological examination. Early suspicion of parathyroid carcinoma can lead to a more extensive surgical approach to reduce the risk of carcinoma recurrence. CASE REPORT The first case involves a 58-year-old woman who presented with severe back pain. An incidental finding on cervical magnetic resonance imaging of a soft-tissue-density mass at the right para-tracheal zone. The large size and the noticeable mass effect pushing the trachea and esophagus to the left side suggested the need for further investigations to rule out malignancy. Initially, it was thought to be a thyroid nodule investigated by fine-needle aspiration that revealed follicular thyroid cancer. After a histopathological examination, it was determined to be a parathyroid carcinoma. The second case involved a 30-year-old woman with a lower-limb tingling sensation. The significantly enlarged mass seen during thyroid ultrasound warranted surgical excision and histopathological analysis to rule out malignancy. Excision of what was considered a parathyroid adenoma revealed a histopathological finding of carcinoma, prompting a hemithyroidectomy. Both patients had high calcium and parathyroid hormone levels preoperatively. CONCLUSIONS Preoperative high calcium, intact parathyroid hormone, creatinine, and alkaline phosphatase, in addition to the lymphocyte-to-monocyte ratio and tumor diameter, are suggested to be predictive of parathyroid carcinoma diagnosis and should be carefully analyzed in all patients presenting with primary hyperparathyroidism.

A man in his 50s was referred with profound, symptomatic hypercalcaemia. He was diagnosed with primary hyperparathyroidism, confirmed on 99m Tc-sestamibi scan. He was treated for the hypercalcaemia and referred to ear, nose and throat (ENT) surgeons for parathyroidectomy, which was delayed due to the COVID-19 pandemic. In the ensuing 18 months, he had five hospital admissions with severe hypercalcaemia requiring intravenous fluids and bisphosphonate infusions. During the last admission, hypercalcaemia was resistant to maximal medical management. Emergency parathyroidectomy was planned, but delayed due to intervening COVID-19 infection. Due to persistent severe hypercalcaemia (serum calcium: 4.23 mmol/L), he was commenced on intravenous steroids, following which serum calcium normalised. Subsequently, he underwent emergency parathyroidectomy, which normalised his serum parathyroid and calcium levels. On histopathological examination, a diagnosis of parathyroid carcinoma was made. On follow-up, patient remained well and normocalcaemic. In patients with primary hyperparathyroidism unresponsive to standard therapy, but responsive to steroids, underlying parathyroid malignancy should be considered., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2023. No commercial re-use. See rights and permissions. Published by BMJ.)

A 10 yr old female spayed domestic shorthair was referred for an 11mo history of persistent total hypercalcemia and elevated ionized calcium with intermittent episodes of lethargy, vomiting, and diarrhea with a history of recurrent urinary tract infections and intermittently elevated kidney values. An abdominal ultrasound, thoracic radiographs, cervical ultrasound, and ionized calcium level, parathyroid hormone (PTH), and PTH-related peptide (PTHrp) levels were assessed. Results were consistent with chronic kidney disease, splenomegaly, diffuse thickening of small intestines, nodular lesions noted in the left thyroid and right parathyroid, and elevated ionized calcium, PTH, and elevated PTHrp levels. A left thyroidectomy and right cranial parathyroidectomy were performed. Hypocalcemia and anemia developed postoperatively, which were managed with calcium carbonate, calcitriol, and calcium gluconate and benign neglect of anemia. Histopathology was consistent with a left thyroid carcinoma and right cranial parathyroid adenoma. Thyroid carcinoma and parathyroid adenomas have not previously been reported to occur concurrently in domestic felines and should be considered when ionized calcium is elevated with both PTH and PTHrp levels increased in addition to ultrasonographic lesions., (© 2023 by American Animal Hospital Association.)

Background: The hallmark of hyperparathyroidism is hypersecretion of parathyroid hormone (PTH) which results in hypercalcemia and hypophosphatemia. While hypercalcemia due to malignancy is often brought about by PTH-related protein in adults, PTH-producing tumors are quite rare in clinical practice. Additionally, from the point of embryology, it is very difficult to examine ectopic PTH-producing tissue such as ectopic parathyroid glands. Furthermore, clear histopathological criteria are not present., Case Presentation: A 57-year-old woman was referred to our hospital for hypercalcemia. Her parathyroid hormone (PTH) level was elevated, but there were no enlarged parathyroid glands. Although 99mTc-MIBI confirmed a localized and slightly hyperfunctioning parathyroid tissue in the anterior mediastinum, it was not typical as hyperfunctioning parathyroid. We finally diagnosed her as ectopic PTH-producing cyst-like tumor with venous sampling of PTH. She underwent anterosuperior mediastinal ectopic PTH-producing cyst-like tumor resection. It is noted that intact-PTH concentration of the fluid in the cyst was very high (19,960,000 pg/mL). Based on histopathological findings, we finally diagnosed her as ectopic PTH-producing parathyroid cyst inside the thymus. After resection of anterosuperior mediastinal thymus including ectopic PTH-producing parathyroid cyst, calcium and intact-PTH levels were decreased, and this patient was discharged without any sequelae., Conclusions: We should know the possibility of superior mediastinal ectopic PTH-producing parathyroid cyst inside the thymus among subjects with ectopic PTH-producing parathyroid glands. Particularly when the cyst is present in the superior mediastinum, it is necessary to do careful diagnosis based on not only positive but also negative findings in 99mTc-MIBI. It is noted that the patient's bloody fluid in the cyst contained 19,960,000 pg/mL of intact-PTH, and its overflow into blood stream resulted in hyperparathyroidism and hypercalcemia. Moreover, in such cases, the diagnosis is usually confirmed after through histological examination of ectopic PTH-producing parathyroid glands. We think that it is very meaningful to let clinicians know this case., (© 2022. The Author(s).)

Background: Most patients with parathyroid adenomas are asymptomatic and rarely present with chronic pancreatitis (CP). Several studies have reported a positive association between primary hyperparathyroidism (PHPT) and pancreatitis. Parathyroidectomy is the definitive treatment for PHPT. IV bisphosphonates can be considered the drug of choice for bridge to surgery., Methods: We reported a 57-year-old female patient was admitted to the emergency room with left upper quadrant abdominal pain and a diagnosis of recurrent pancreatitis. Magnetic Resonance Cholangiopancreatography confirmed the diagnosis of CP. The patient had no common etiology of pancreatitis. Persistent hypercalcemia was noted despite administering intravenous fluids, and Calcitonin. Intravenous Pamidronate, a Bisphosphonate derivative, was also administered. Although calcium levels initially decreased, they were later found to rebound to previous levels., Results: A diagnosis of parathyroid adenoma and PHPT was made based on the elevated parathyroid hormone levels and cervical ultrasonography indicated right inferior parathyroid adenoma. Technetium-99m methoxy-isobutyl-isonitrile scintigraphy revealed a focal hot spot of tracer accumulation at the right lower thyroid bed. The patient underwent right lower parathyroidectomy smoothly and successfully. After right lower parathyroidectomy, she had normal serum calcium levels (9.2 mg/dL) and parathyroid hormone (16.1 pg/mL). There was no recurrent abdominal pain after the operation., Conclusion: CP is a rare manifestation of parathyroid adenoma. When patients with a history of recurrent pancreatitis, without common causes of pancreatitis, present persistent elevated serum calcium levels, PHPT could be suspected., Competing Interests: The authors have no funding and conflicts of interest to disclose., (Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.)

Objective: To review the features and treatment of parathyroid cancer in our series. Explore the suitable extent of initial surgery and the effect of adjuvant radiotherapy in local recurrence., Methods: Seven cases of parathyroid cancer presented from 2014 to 2021. The presenting features, diagnosis, and treatment are presented., Results: Only two patients had multiple manifestations of hypercalcemia. Marked hypercalcemia, which was revealed to be an average of 13.9 mg/dl (range from 11.8 mg/dl to 15.8 mg/dl), was observed in four patients (57%). The others' serum calcium levels were in the normal range with an average of 9.9 mg/dl (range from 8.6 mg/dl to 10.8 mg/dl). All seven patients had hyperparathyroidism with an average of 733 pg/ml (range from 113 pg/ml to 3193 pg/ml). En bloc resection was performed in two patients with neighboring structure invasion, and four patients with complete tumor capsules underwent tumor resection with limited resection of the thyroid gland. Postoperative adjuvant radiotherapy appeared unsuccessful for local recurrence., Conclusion: High calcium, high PTH, parathyroid occupation by ultrasound, and intraoperative invasion should be considered to have the possibility of parathyroid cancer. Open surgery is recommended and protecting tumor integration is the elementary surgery principle. The initial surgical extent should be decided by the invasion of the tumor. When PC has a local recurrence, the debulking surgery and adjuvant radiotherapy are always fake., Competing Interests: We declare that there is no conflict of interest regarding the publication of this paper., (Copyright © 2022 Chen Wang et al.)

An uncommon cause of primary hyperparathyroidism is a cystic parathyroid adenoma. This paper describes two patients with hypercalcemia and right knee disease. Their serum calcium concentration was high, phosphorus concentration was low, and parathyroid hormone (PTH) concentration was high. Ultrasound and computed tomography scans of the neck indicated a cystic mass near the thyroid. Parathyroid scintigraphy showed no focal uptake in one patient and low tracer concentration in the cystic mass in the other patient. Following resection of the cystic masses, both were pathologically confirmed to be a cystic parathyroid adenoma with predominantly cystic degeneration. The calcium and PTH concentrations gradually decreased to the reference range. Both patients were stable at their last follow-up. The diagnosis of a functional cystic parathyroid adenoma is highly challenging because of the different clinical manifestations and negative result on parathyroid tracer scintigraphy. For patients with high serum calcium and PTH concentrations and a cystic mass in the neck, resection of the mass and subsequent postoperative pathological diagnosis is necessary even if the clinical diagnosis of a parathyroid adenoma cannot be confirmed preoperatively. Decreases in the PTH and serum calcium concentrations indicate successful resection of a functional parathyroid adenoma.

Background: Giant parathyroid adenoma is a type of parathyroid adenoma weighing > 3.5 g and having a size of more than 2 cm., Case Presentation: This report describes giant primary parathyroid adenoma with reference to the literature. We report the case of a 48-year-old Persian man referred to the clinic with knee and lower back pain. He had a history of mitral valve replacement and several episodes of bilateral nephrolithiasis. After a thorough assessment, a neck mass with a possible thyroid origin was detected, but further assessment showed it was of parathyroid origin. The resected mass was 9 × 6× 4 cm and weighed 122 g, and histopathology showed a giant parathyroid adenoma., Conclusion: Giant parathyroid adenomas that weigh more than 110 g and are larger than 8 cm can lead to significant hypercalcemia. Despite giant parathyroid adenomas and high parathyroid hormone levels, a calcium crisis may not always occur in these patients, and the masses may be initially misdiagnosed as a thyroid mass., (© 2022. The Author(s).)

Purpose: Giant parathyroid adenoma (GPA) can present with severe biochemical derangement similar to the clinical presentation of parathyroid carcinoma (PC). This study aims to present the current evidence on surgical management of GPAs in primary hyperparathyroidism., Methods: A systematic review of the literature on GPAs was conducted following the PRISMA guidelines. Data on clinical, biochemical, preoperative diagnostic, and surgical methods were analysed., Results: Sixty-one eligible studies were included reporting on 65 GPAs in eutopic, ectopic mediastinal, and intrathyroidal locations (61.5%, 30.8%, and 7.7%, respectively). A palpable neck mass was present in 58% of GPAs. A total of 90% of patients had symptoms including fatigue, skeletal pain, pathological fracture, nausea, and abdominal pain. Ninety percent of patients had significant hypercalcaemia (mean 3.51 mmol/L; range: 2.59-5.74 mmol/L) and hyperparathyroidism with PTH levels on average 14 times above the upper limit of the normal reference. There was no correlation between the reported GPA size and PTH nor between GPA weight and PTH (p = 0.892 and p = 0.363, respectively). Twenty-four percent had a concurrent thyroidectomy for suspicious features, intrathyroidal location of GPA, or large goitre. Immunohistochemistry such as Ki-67, parafibromin, and galectin-3 was used in 18.5% of cases with equivocal histology. Ninety-five percent of GPAs were benign with 5% reported as atypical adenomas., Conclusion: The reported data on GPAs are sparse and heterogeneous. In GPAs with suspicious features for malignancy, en bloc resection with concurrent thyroidectomy may be considered. In the presence of equivocal histological features, ancillary immunohistochemistry is advocated to differentiate GPAs from atypical adenomas and PCs., (© 2021. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

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., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2022. No commercial re-use. See rights and permissions. Published by BMJ.)

Purpose: Spontaneous bleeding is a rare but serious complication of parathyroid adenomas and few cases were reported in the literature. Clinical manifestations and treatment may vary but sometimes an immediate surgery is required. In other cases a conservative approach can be preferred based on clinical stability and patients' conditions. The purpose of this work is to describe our case and to carry out a review of the current literature on this topic., Methods: We reported a case of a parathyroid adenoma hemorrhage in an elderly patient describing its management. Moreover, a literature review of 57 cases was carried out, with the aim of collecting data about the most involved parathyroid gland and identifying the most correct management based on clinical manifestations and chosen treatments., Results: The patients had an age between 29 and 81 years (mean 56.9 ± 16.4 years). Forty-four patients were females (77.2%), whereas 12 were males (21.8%). Based on clinical severity, age, comorbidities and calcemic status, three possible scenarios were identified, each with a recommended management., Conclusions: In parathyroid adenoma hemorrhages a careful clinical assessment is crucial to identify emergency conditions requiring immediate intubation, tracheostomy or neck exploration. Elderlies and comorbid patients have a higher risk of perioperative complications and indication for surgery should be evaluated case by case: whenever feasible, a conservative approach should be preferred in these subjects, especially in those with a stable course and without hypercalcemia-related symptoms., (© 2021. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.)

BACKGROUND Parathyroid carcinoma (PC) is an extremely rare endocrine malignancy, with a reported increase in incidence in the past decade. PC generally presents in an indolent fashion, featuring nonspecific symptoms associated with hypercalcemia. CASE REPORT Case 1: A 30-year-old man was admitted for symptoms associated with hypercalcemia and elevated parathyroid hormone (PTH). Imaging examinations showed the presence of a cervical nodular lesion. The patient underwent surgery, and the pathological diagnosis was PC. Case 2: A 45-year-old man with a history of hypothyroidism was referred to our Endocrinology Department for a cervical nodular lesion. A fine-needle aspiration was performed, and the result was suggestive of papillary carcinoma. Blood testing showed only mild hypercalcemia and PTH elevation, with no associated symptoms. The patient underwent surgery, and the histological examination confirmed the diagnosis of PC. Case 3: A 38-year-old man presented with diffuse bone pain and muscle weakness, severe hypercalcemia, high levels of PTH, and a cervical mass. The patient underwent surgery. Diagnostic pathology confirmed the diagnosis of PC. Five years later, the patient presented with a cutaneous metastasis, followed 1 year later by pulmonary metastases. CONCLUSIONS Most PCs are slow-growing tumors. Some of these tumors are diagnosed in association with hereditary syndromes. A clear distinction between benign and malignant lesions is not always simple because there is a lack of specific clinical distinguishing features of malignant lesions. Currently, surgical resection is the preferred approach; however, owing to the rarity of this condition, there is a void of high-quality data.

The most common causes of hypercalcemia are primary hyperparathyroidism (PHPT) and malignancy. Parathyroid carcinoma (PC), causing a severe PHPT, is the rarest parathyroid tumor. A diagnosis of PC is challenging because the clinical profile overlaps with that of benign counterpart. Surgery is the mainstay treatment. CDC73 mutations have been detected in up to 80% of sporadic PCs. Ectopic production of parathyroid hormone (PTH) by malignant nonparathyroid tumors is a rare condition accounting for less than 1% of hypercalcemia of malignancy. PTH secretion can be considered an aberration in the tissue specificity of gene expression and may involve heterogeneous molecular mechanisms., (Copyright © 2021 Elsevier Inc. All rights reserved.)

Competing Interests: The authors declare no conflict of interest.

Background: Primary hyperparathyroidism (PHPT) is a common endocrine disorder and the most frequent benign cause of hypercalcemia. PHPT is characterized by autonomous hypersecretion of parathyroid hormone (PTH), regardless of serum calcium levels. Familial hypocalciuric hypercalcemia (FHH) is a rare, benign syndrome only affecting the regulation of calcium metabolism. FHH is an autosomal-dominant genetic disease with high penetrance, caused by an inactivating variant in the CASR gene encoding the calcium-sensing receptor (CaSR). We present a unique case of concomitant PHPT and FHH without clinically actionable variants in MEN1., Case Presentation: A 47-year-old Caucasian man with severe hypercalcemia, genetic FHH, and initially normal parathyroid scintigraphy was referred for endocrine evaluation due to nonspecific symptoms. Biochemical evaluation showed elevated serum ionized calcium and PTH. The calcium-creatinine clearance ratio was low. All other biochemical measures were normal, including kidney function. Genetic evaluation was redone and confirmed FHH. A new parathyroid scintigraphy showed a significant single adenoma corresponding to the lower left gland. The patient underwent parathyroidectomy, and a parathyroid adenoma was removed. A reduced level of hypercalcemia persisted due to FHH., Conclusions: The correct diagnosis of the underlying cause of hypercalcemia is important to ensure the right treatment. Patients with FHH should avoid operative treatment, and PHPT should be differentiated from MEN1 to determine whether surgery should include parathyroidectomy with removal of one adenoma or 3.5 hyperplastic parathyroid glands., (© 2021. The Author(s).)

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., Competing Interests: The authors have no funding and conflicts of interests to disclose., (Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.)

BACKGROUND Parathyroid carcinoma (PC), accounting for 0.005% of all cancers, is responsible for less than 1% of all cases of primary hyperparathyroidism, and equally affects males and females, usually in 4th or 5th decades of life. PC can occur sporadically and can be associated with congenital genetic syndromes such as hyperparathyroidism-jaw tumor syndrome (HPT-JT), isolated familial hyperparathyroidism, or multiple endocrine neoplasia 1 and 2 syndromes. Surgery is the main treatment, with a limited role of radio- and chemotherapy, which allows 49-77% of patients to survive 10 years. In this work we report the case of a patient with parathyroid carcinoma, whose treatment required 13 surgeries over a period of 27 years, together with radiotherapy and pharmacological treatment. CASE REPORT A 51-year-old woman was first diagnosed with primary hyperparathyroidism in 1993 at the age of 23. From 1993 to present, she underwent 13 surgeries and 33 courses of radiotherapy due to recurrent lesions, which initially had a character of parathyroid adenomas, then parathyromatosis, and finally were diagnosed as parathyroid carcinoma. The patient also required and currently requires complex pharmacological treatment to control the calcemia and manage the complications of the primary disease. Supervision by the multidisciplinary professional medical team allows the patient to lead a normal life with good control of the disease. CONCLUSIONS Parathyroid carcinoma is a rare disease with a number of complications; however, obtaining satisfactory long-term survival with acceptable quality of life is achievable.

Parathyroid carcinoma is a rare malignant disease that presents as a sporadic or familial primary hyperparathyroidism (PHP). The latter is associated with some genetic syndromes. It occurs with equal frequency in both sexes, unlike PHP caused by parathyroid adenoma that is more common in women. It should be suspected in cases of severe hypercalcemia, with high parathyroid hormone levels and a palpable cervical mass. Given the difficulty in distinguishing between parathyroid carcinoma and adenoma prior to the surgery, the diagnosis is often made after parathyroidectomy. The only curative treatment is complete surgical resection with oncologic block resection of the primary tumor to ensure free margins. Adjuvant therapies with chemotherapy or radiation therapy do not modify overall or disease-free survival. Recurrences are common and re-operation of resectable recurrent disease is recommended. The palliative treatment of symptomatic hypercalcemia is crucial in persistent or recurrent disease after surgery since morbidity and mortality are more associated with hypercalcemia than with tumor burden.

Hypercalcemia is usually secondary to one etiology, although two coexisting etiologies can rarely cause hypercalcemia. Here, we report a 47-year-old woman with hypercalcemia caused by comorbid parathyroid adenoma and pulmonary tuberculosis. Primary hyperparathyroidism is the most common cause of hypercalcemia. Tuberculosis is a rare cause of hypercalcemia, but Japan continues to have an intermediate tuberculosis burden. Therefore, tuberculosis should be considered as a cause of hypercalcemia in Japan. Patients with tuberculosis are often asymptomatic, making the diagnosis difficult. In the previous cases in which these diseases coexisted, one disease was diagnosed after treatment of the other. In our case, the very high 1,25-dihydroxyvitamin D level (162 pg/mL) helped us to diagnose asymptomatic tuberculosis and both diseases were diagnosed promptly. It is necessary to consider comorbidities, including tuberculosis in a case with a very high 1,25-dihydroxyvitamin D level. We report a valuable case in which the early diagnosis and treatment of tuberculosis and primary hyperparathyroidism prevented the spread of tuberculosis.

Skeletal deformation like genu valgum is reported to be rare in Primary hyperparathyroidism (PHPT). The solitary adenoma or hyperplasia of the parathyroid glands are the cause in 80-85% of the cases. We report 2 cases of girls on 12 years and 15 years of age, complaining from pain and genu valgum deformation of the lower extremities before the planned orthopaedic surgical correction. The first patient had complaints for 3 years and lost ability to walk independently, the second case lost normal gate for a period of 5 months. The paraclinical screening discovered hypercalcemia, hypophosphatemia, elevated alkaline phosphatase, normal creatinine, raised parathormone. In the first case the X-rays depicted fibrocystic osteodistrophy from a hyperparathyroid type with bone cysts, giant cell "brown tumors" and pathological bone reorganization, in the second case - coarse fibrous structure of the left knee joint with genu valgum with bone cysts in the distal metaphysis of the left femur. The ultrasound of the thyroid gland found oval hypoechoic formations with suspicious origin from the parathyroid glands. These findings were confirmed from the SPECT/CT pointing active adenomas in the parathyroid glands. Skeletal deformation like genu valgum is the reason to search for the primary diagnosis in our 2 cases. Investigation of the serum calcium and parathormone are diagnostic in 100%. The imaging diagnosis has a critical role for indicating surgical treatment of the parathyroid gland adenoma. Key words: genu valgum, hypercalcemia, paediatric parathyroid adenoma, ultrasound, SPECT/CT.

Hyperparathyroidism leading to hypercalcaemia is a rare cause of acute pancreatitis with debatable association. The diagnosis of hyperparathyroidism is frequently overlooked or delayed as symptoms are non-specific and serum calcium is not routinely measured in acute pancreatitis. Early diagnosis and treatment of hyperparathyroidism may reduce the chances of complications. We report a 35-year-old woman, who was admitted with recurrent episodes of acute pancreatitis. She was previously diagnosed with gall-stone induced acute pancreatitis, had undergone laparoscopic cholecystectomy, but the recurrence of acute pancreatitis suggested an alternative aetiology and provoked extensive investigations. Serum calcium was found to be elevated. No additional risk factor for pancreatitis was found. Further workup revealed primary hyperparathyroidism, which was due to a functioning parathyroid adenoma. She has undergone parathyroidectomy followed by an uneventful recovery. She subsequently conceived and is now in her first trimester, without any recurrence of acute pancreatitis since her surgery., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2020. No commercial re-use. See rights and permissions. Published by BMJ.)

Background: Primary hyperparathyroidism may present in a myriad of manners, varying from an incidental asymptomatic biochemical finding to gastrointestinal, psychiatric, renal, and bone manifestations. While hyperparathyroidism remains a rare diagnosis in the pediatric population, the initial approach to diagnosis and management of hypercalcemia in children is imperative for the general pediatrician. Herein, we describe an adolescent who presented with a lytic bone lesion and severe, symptomatic hypercalcemia due to primary hyperparathyroidism., Case Presentation: A 14-year-old male presented with vomiting, constipation, abdominal pain, and lethargy. He had an elevated total corrected calcium of 4.3 mmol/L. He was found to have a large pelvic lytic tumor consistent with a brown tumor due to primary hyperparathyroidism. He received pharmacologic therapy for stabilization of his hypercalcemia, including intravenous saline, intravenous bisphosphonates, and calcitonin. He subsequently received definitive therapy via parathyroidectomy and his post-operative course was complicated by hungry bone syndrome. Long-term follow-up has found full resolution of the lytic lesion and restored calcium homeostasis., Conclusions: We present this case to highlight the possible presentations of hypercalcemia and hyperparathyroidism that are essential for a general pediatrician to recognize to ensure prompt diagnosis and management. Evaluation for hypercalcemia should be considered in patients with suggestive symptoms and physical exam findings. To our knowledge, this patient represents the first reported pediatric case of a pelvic brown tumor in an adolescent. While the multi-systemic complications of hyperparathyroidism may be quite severe, swift and appropriate management may mitigate these clinical outcomes.

Introduction: A 26-year-old female presented with severe hypercalcemia. She was asymptomatic. Blood analysis revealed high serum calcium (13 mg/dL = 3.25 mmol/L) and elevated intact parathyroid hormone (iPTH) levels (267.5 ng/L)., Results: Ultrasonography showed a mixed solid-cystic nodule of 30 mm in the left thyroid lower lobe and a nodular lesion of 14 mm posterior of the thyroid upper pole suggestive for parathyroid adenoma. Sestamibi scan and magnetic resonance imaging (MRI) revealed hyperfunctioning parathyroid tissue posterior of the left thyroid upper lobe. During surgery, a 15 mm nodule in the usual location of the left superior parathyroid was resected. Intraoperative frozen section revealed normal parathyroid tissue and intraoperative PTH levels did not decrease. Left thyroidectomy was performed due to the large solid-cystic nodule. Intraoperative PTH levels normalized 10 min later. Pathologic examination revealed a 28 mm nodule in the lower thyroid pole compatible with a parathyroid carcinoma (PC). Due to positive margins, a completion thyroidectomy was performed. 5-year follow-up showed no recurrence., Conclusions: PC is a rare entity comprising 0.5-2% of patients with primary hyperparathyroidism. Even more unusual is an intrathyroidal parathyroid gland (0.2%). Only a dozen cases of intrathyroidal PC have been reported. Our case is the second patient reported to be asymptomatic.

Serum intact parathyroid hormone (iPTH) levels are high or high normal in patients with parathyroid adenoma. Rarely these patients can have normal or low serum iPTH values. With sandwich immunometric assays, an exceptionally high serum iPTH level can lead to falsely low measurement due to the 'hook effect'. Here, we describe the case of a 66-year-old female patient with PTH-independent hypercalcaemia which mimicked parathyroid adenoma. A multidisciplinary team approach helped in the diagnosis and management leading to complete recovery., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2020. No commercial re-use. See rights and permissions. Published by BMJ.)

Mediastinal parathyroid carcinoma (PC) is a rare entity in primary hyperparathyroidism. The aim of this report is to demonstrate a case of mediastinal PC, and to provide a systemic literature review of this rare condition. A 34-year-old woman who had already undergone two cervical operations for hyperparathyroidism suffered from another recurrence, presenting with recurrent acute pancreatitis and persistent hypercalcemic crisis. Technetium-99 methoxyisobutylisonitrile imaging (MIBI) and computed tomography scanning (CT) identified three possible parathyroid tumors, one of which was the recurrence of residual tumor locating in the thyroid region, while the other two were ectopic tumors locating in the suprasternal fossa and thymus region, respectively. Pathological examination confirmed the diagnosis of PC. We conducted a systemic literature review by searching the PubMed MEDLINE from 1951 to 2019 for studies of all types in the English language only, using terms "mediastinal, mediastinum, parathyroid, carcinoma." Including our reported case, a total of 21 cases with ectopic mediastinal PCs were assessed for demographic data, tumor location and size, biochemical findings, and symptomatology, etc. Two thirds of the patients were men, with a mean age of 44 years old, a mean serum calcium of 14.2 mg/dl, and a mean serum intact parathyroid hormone of 1,216 pg/ml. We identified 89.5% of carcinomas in the anterosuperior mediastinum, and 10.5% in the middle mediastinum, with a mean diameter of 54 mm, and a mean weight of 216 g. MIBI and CT were the most commonly used methods to localize these mediastinal tumors, with 69.2 and 100% sensitivity, respectively. Half of the patients underwent more than one operation. Diagnosis and treatment of mediastinal PCs represent a challenge. Early suspicion, appropriate preoperative localization studies, and the cooperation of endocrinologists and surgeons are crucial in the effective management., (Copyright © 2020 Jiajue, Song, Wang and Li.)

The majority of patients with parathyroid carcinoma (PC) have significant clinical signs and simultaneous symptoms related to the unregulated hypersecretion of parathyroid hormone (PTH) by the tumor. The aim of this case was to report a patient presenting an isolated bone fracture leading to the diagnosis of PC. A 20-years-old female patient presenting a fracture of both bones of the forearm following a fall from her own height. Imageries showed diffuse bone demineralization. Biology revealed malignant hypercalcemia at 4.1 mmol/L and PTH at 1331 pg/mL. Bone densitometry showed severe osteoporosis with a femoral and lumbar T-score < - 3DS. Imageries showed a right parathyroid mass of 32 mm. An one-piece excision of the pathological gland, right thyroid lobectomy and ipsilateral central lymph node dissection were performed. Postoperatively, the patient presented a hungry bone syndrome with severe hypocalcemia and required substitutive treatment. PTH on day 1 was normal. Pathology analysis found a PC with Ki67 at 3%, lymph node removal was negative. Complete one-piece surgical excision is the only potentially curative treatment for PC. Preoperative suspicion and intraoperative recognition of malignant features is important in order to propose an appropriate compartmental surgery, which can provide the lowest possible recurrence rate.

Background: Primary hyperparathyroidism is a rare condition of disease which can seldomly present as giant retrotrhyroideal cysts, complicating the localization of the adenoma to resect., Case Presentation: A 56-year old female presented with hypercalcaemia of 3.38 mmol/L (2.2-2.65 mmol/L) and a history of breast cancer. A fast growing cystic parathyroidal adenoma was diagnosed by a multimodal approach including comprehensive diagnostic imaging (ultrasonography, scintigraphies, dynamic MRI) and cytopathological investigations after ultrasonography-guided puncture. The patient was cured by surgical extraction of the whole adenoma. In retrospect, the rapid growth was most likely induced by cinacalcet (initially 30 mg/d, later 60 mg/d) therapy which the patient received for few months only. Primary hyperparathyroidism was ascertained because surgical removal of the solitary adenoma cured the patient. Furthermore, there was no relevant renal insufficiency or history of prolonged calcium-level deregulation., Conclusions: This phenomenon of cystic degeneration of parathyroidal adenoma under therapy with cinacalcet has previously been described in secondary hyperparathyroidism, but not in primary hyperparathyroidism and should be considered in diagnostic approach.

Unexpected Postoperative Diagnosis of Parathyroid Carcinoma Abstract. Parathyroid carcinoma is a rare endocrine malignancy. It typically presents itself with similar clinical features as seen in patients with primary hyperparathyroidism caused by a parathyroid adenoma. Due to overlapping clinical manifestations, imaging findings and pathological characteristics, the differentiation between benign and malignant parathyroid disease poses a diagnostic challenge, especially prior to surgery. We report the case of a 67-year-old male who presented with symptomatic hypercalcemia and primary hyperparathyroidism. During resection of the enlarged parathyroid gland, suspicious macroscopic features were noticed and the histopathology finding confirmed the malignancy. Parathyroid carcinoma is rarely diagnosed preoperatively, which is mainly due to the lack of distinctive symptoms or morphological features suggesting a malignant disease. As parathyroid carcinoma often has a genetic background, knowledge of mutation status and family history is of high relevance. Surgery with complete resection of the parathyroid carcinoma is the mainstay for a successful treatment and a good prognosis.

Paediatric hypercalcaemia is a rare condition which can be easily overlooked or misdiagnosed. We report two paediatric patients who presented to the Department of Paediatrics, Pardubice Hospital, Pardubice, Czech Republic, in 2009 and 2010, respectively. Each patient was diagnosed with hypercalcaemia due to a different cause. The first case involved a seven-month-old infant who presented with failure to thrive, vomiting and psychomotor retardation. Fluorescent in situ hybridisation revealed Williams-Beuren syndrome. The second patient was a 16-year-old girl with abdominal pain and renal colic due to hypercalcaemia-induced urolithiasis. High parathyroid hormone serum levels suggested primary hyperparathyroidism. An adenoma of the left upper parathyroid gland was diagnosed via technetium-99m-labelled methoxyisobutyl isonitrile single photon emission computed tomography and removed surgically. Hypercalcaemia should be considered in the differential diagnosis of various disease states, particularly among infants who fail to thrive or children with abdominal pain.

Parathyroid or hypercalcaemic crisis is a rare presenting manifestation of primary hyperparathyroidism and usually seen in relation to parathyroid carcinoma and multiple myeloma. We present a middle-aged woman with extreme hypercalcaemia due to parathyroid adenoma presenting as acute kidney injury. This case highlights the need to consider primary hyperparathyroidism in differentials of a patient presenting with severe hypercalcaemia and renal dysfunction. Also this is the second reported case in literature of a patient surviving such extreme hypercalcaemia due to parathyroid adenoma., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2018. No commercial re-use. See rights and permissions. Published by BMJ.)

Background: Carcinoma of the parathyroid gland (PC) described by De Quervain since 1909, it represents the least common neoplasm, with an incidence of 1,25/10,000,000 peoples. It has been reported approximately 1,000 cases of CP in world literature. There are two series in Mexico, one of eight patients and other with four cases. Because CP is functionally active, its early clinical behavior is similar to that of parathyroid benign neoplasms., Clinical Case: A 66-year-old female with history of thighbone pain and spontaneous femoral fracture, osteolytic lesions, hypercalcemia, elevated levels of alkaline phosphatase and parathyroid hormone detected; the scintigraphy showed a functioning tumor located in upper mediastinum. By hemithyroidectomy in block, the tumor was resected. Histopathological study reported parathyroid carcinoma., Discussion: PC is the least common neoplasia, in patients with parathyroid hormone levels greater than 1,000 pg/ml and hypercalcemia upper of 14 mg/dl this disease should be suspected.

A 28-year-old Syrian refugee presented with right-sided knee pain and progressive deterioration of the general condition over the past months. Laboratory diagnostics revealed severe hypercalcemia due to primary hyperparathyroidism, and computed tomography (CT) scanning demonstrated disseminated osteolytic lesions throughout the skeleton. Histologically, these lesions were characterized by multinuclear giant cells (defining these lesions as so-called brown tumors). Finally, surgical removal of a jugular mass allowed the histopathologic diagnosis of a sporadic parathyroid carcinoma. In the patient, this condition was associated with a mutation in the HPRT2 gene locus.

Parathyroid carcinoma is a very rare malignant tumor of the parathyroid gland. Clinically, this cancer is characterized by the presence of severe primary hyperparathyroidism. Diagnosis is based on histological examination but is not always easy. Surgery is the treatment of choice. We report the case of a 59-year old woman with a personal history of arterial hypertension and of recurrent renal lithiasis, presenting with diffuse bone pain associated with asthenia. Neck examination showed hard basi-cervical swelling with nonpalpable lower edge. Laboratory tests showed hypercalcaemia 4.1 mmol/l, hyperparathyroidism with very high parathyroid hormone (PTH) value (1088 pg/ml or 13 times normal). Technetium-99m-sestamibi scintigraphy showed fixed MIBI abnormality in the projection of the left inferior parathyroid. The patient underwent left inferior parathyroidectomy, with ipsilateral mediastinal-recurrent nerve dissection. The postoperative course was marked by normalization of plasma calcium and THP. Anatomopathological examination showed parathyroid carcinoma. The diagnosis of parathyroid carcinoma is generally based on the combination of biological, radiological and histological signs. The severity of this pathology is due to severe hypercalcaemia and to the risk of recurrence and distant metastases, justifying prolonged monitoring.

A 49-year-old man presented in the emergency department with altered sensorium and renal failure and was placed on a ventilator. Evaluation with MRI of the brain showed enhancing lesion in the occipital lobe. Biochemical tests revealed elevated calcium and parathyroid hormone (PTH) levels. Further evaluation revealed multiple lesions at both lobes of the lungs and nodular lesion in the right inferior pole of the thyroid. Diagnosis of metastatic parathyroid carcinoma was suspected. Patient was stabilised with bisphosphonates and haemodialysis and planned for surgery. Intraoperatively a hard lesion was found in the right inferior parathyroid infiltrating the surrounding structures. En bloc resection was done. General condition of the patient improved and he was weaned from the ventilator. Histopathology confirmed the diagnosis of parathyroid carcinoma. Postoperatively, there was persistent elevation of PTH in spite of a fall in calcium levels indicating functioning brain and pulmonary metastases. Due to extensive metastases, the patient was given palliative cinacalcet and was kept under follow-up., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Ltd (unless otherwise stated in the text of the article) 2017. All rights reserved. No commercial use is permitted unless otherwise expressly granted.)

Parathyroid carcinoma is a very rare malignancy responsible for 0.4 to 5.2% of hyperparathyroidism. Clinical diagnosis is difficult and treatment should be codified. Surgery is the only curative treatment. We report the case of a female patient treated for malignancy-associated hypercalcemia revealing parathyroid carcinoma. The patient underwent surgery; after three months she developed lymphatic recurrence. Given the absence of other secondary involvement, bilateral lymph node dissection followed by chemotherapy was performed. Parathyroid carcinoma is often suspected on the basis of biological, radiological and especially intraoperative macroscopic criteria, but definitive diagnosis is made histopathologically. Surgery is currently the only curative treatment and the role of adjuvant therapy is to establish., Competing Interests: Les auteurs ne déclarent aucun conflit d'intérêts.

Objective: To assess the diagnostic and therapeutic options in the care of patients with primary hyperparathyreosis in outpatient practice.Cohort and methods: The study included all the patients with primary hyperparathyroidism treated at the 2nd Internal Medicine Department, Masaryk University and the University Hospital of St. Anne in Brno in the period from Jan 1, 2008 to Dec 31, 2013. The sample consisted of 218 patients, including 41 men and 177 women. Patients with secondary hyperparathyroidism, especially patients with underlying hypovitaminosis D, renal insufficiency and those taking medications with possible effects on parathyroid hormone levels, have not been included in the study. A special attention was paid to differences between the normocalcaemic and hypercalcaemic patients. Ultrasound scanning was performed in all patients, while scintigraphy was indicated in patients who are considered for possible surgical treatment., Results: In the group of 218 patients, serum calcium levels at the baseline were pathologically elevated in 31 patients (14 %) and normal in 187 patients (86 %). One fifth of patients with normocalcaemic primary hyperparathyroidism developed long-term hypercalcaemia - within two years in two thirds of the patients from the onset of the disease and sporadically also after more than four years of follow-up. Parathyroid adenoma was found and removed in 30 hypercalcemic patients (in 97 % of all 31 hypercalcemic patients operated on) and in 2 normocalcemic patients (40 % of all 5 the normocalcemic patients operated on). Pharmacological treatment was administered to 22 patients, of which 9 patients received long-term treatment and 13 patients received pharmacotherapy only during the preoperative preparation for patients with very high serum calcium levels., Conclusion: The results support the opinion that primary hyperparathyroidism is a biphasic disease. The initial normocalcemic period is often asymptomatic or associated with symptoms of little importance. Severe complications, however, may already be present also in normocalcemic patients. The decision of when patients with normocalcemic primary hyperparathyroidism should be monitored and when initiation of treatment is needed should also require more detailed information.Key words: hypercalcaemia - hyperparathyroidism asymptomatic and primary - normocalcaemia - outpatient care - parathyroid hormone - surgery and pharmacotherapy.