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Clinics and Practice Jan 2024Parathyroid adenoma is the most common cause of hypercalcemia and rarely leads to a hypercalcemic crisis, which is an unusual endocrine emergency that requires timely...
BACKGROUND
Parathyroid adenoma is the most common cause of hypercalcemia and rarely leads to a hypercalcemic crisis, which is an unusual endocrine emergency that requires timely surgical excision.
CASE PRESENTATION
A 67-year-old male was admitted to the ER of the Euroclinic Hospital, Athens, Greece, because of elevated calcium levels and a palpable right-sided neck mass, which were accompanied by symptoms of nausea, drowsiness, and weakness for six months that increased prior to our evaluation. A gradual creatinine elevation and decreasing mental state were observed as well. The initial laboratory investigation identified severely elevated serum calcium (3.6 mmol/L) levels consistent with a hypercalcemic crisis (HC) and parathyroid hormone PTH (47.6 pmol/L) due to primary hyperparathyroidism. Neck ultrasonography (USG) identified a large, well-shaped cystic mass in the right thyroid lobe. With a serum calcium concentration of 19.5 mg/dL and a PTH of 225.3 pmol/L, the patient underwent partial parathyroidectomy and total thyroidectomy, which decreased serum calcium and PTH to 2.5 mmol/L and 1.93 pmol/L, respectively. Histology revealed a giant intrathyroidal cystic parathyroid adenoma, which was responsible for the hypercalcemic crisis. Postoperatively, the patient developed severe biochemical and clinical hypocalcemia, with calcium concentrations as low as 1.65 mmol/L, consistent with hungry bone syndrome (HBS), which was treated with high doses of intravenous calcium gluconate and oral alfacalcidol, and a slow recovery of serum calcium. After discharge, parathyroid function recovered, and symptomatology resolved entirely in more than one month.
DISCUSSION/CONCLUSIONS
We present a case involving an exceptionally large intrathyroidal parathyroid adenoma that is characterized by clinical manifestations that mimic malignancy. The identification and treatment of such tumors is challenging and requires careful preoperative evaluation and postoperative care for the risk of hungry bone syndrome.
PubMed: 38391401
DOI: 10.3390/clinpract14010015 -
Experimental and Clinical... Jan 2024Deviations of calcium, phosphate, parathyroid hormone, and vitamin D levels are the basis for the diagnosis of calcium-phosphate metabolism disorders. The plasma...
Deviations of calcium, phosphate, parathyroid hormone, and vitamin D levels are the basis for the diagnosis of calcium-phosphate metabolism disorders. The plasma concentration of the biologically active form known as free calcium is regulated in a harmonious manner by its exchange in the bones and reabsorption by the kidneys. These steps take place under the control of parathyroid hormone and calcitriol. In the process of chronic kidney disease, the kidney cannot synthesize adequate calcitriol, and the resulting hypocalcemia and hyperphosphatemia cause the development of secondary hyperparathyroidism. Osteoporosis is a metabolic bone disease and is essentially the consequence of osteoclastogenesis-induced bone resorption that exceeds bone formation. Osteoporosis is common after kidney transplant. However, hypocalcemia following kidney transplant is rare. The hungry bone syndrome after parathyroidectomy is often responsible for this condition in the pretransplant period. Denosumab is a human monoclonal antibody developed against the receptor activator of nuclear factor kappa-B ligand (known as RANKL). Denosumab exerts an antiresorptive effect on bones by reducing differentiation into osteoclasts. It is an effective treatment option for osteoporosis in the general population. There is insufficient scientific data regarding the use of denosumab in kidney transplant patients. Here, we present the case of a kidney transplant recipient who developed severe hypocalcemia (serum calcium 4.7 mg/dL) after denosumab treatment for osteoporosis.
Topics: Humans; Hypocalcemia; Denosumab; Calcitriol; Calcium; Kidney Transplantation; Osteoporosis; Parathyroid Hormone; Phosphates
PubMed: 38385423
DOI: 10.6002/ect.MESOT2023.P10 -
The American Journal of Case Reports Feb 2024BACKGROUND Hyperparathyroidism is a common issue in patients on long-term hemodialysis, necessitating parathyroidectomy with deep venipuncture. These patients frequently...
BACKGROUND Hyperparathyroidism is a common issue in patients on long-term hemodialysis, necessitating parathyroidectomy with deep venipuncture. These patients frequently exhibit a heightened incidence of vascular calcification, complicating deep venipuncture and increasing the risk of associated complications. Therefore, a crucial aspect of preoperative assessment in this population involves identifying vascular calcification, with a preference for ultrasound-guided intraplane puncture. Special care is required to prevent blood vessel injuries and postoperative complications such as deep vein thrombosis. CASE REPORT We present the case of a 66-year-old woman with secondary hyperparathyroidism who encountered challenges during an internal jugular vein puncture, leading to subsequent thrombosis. Initial attempts were hindered by calcification of the internal jugular vein's vascular wall, resulting in stratified damage to the vessel's wall. However, the ultrasound-guided in-plane puncture technique successfully allowed the insertion of a central venous catheter without further damage to the vascular wall. On the postoperative fourth day, thrombus formation was observed in the damaged vascular wall from the initial puncture. CONCLUSIONS The ultrasound-guided in-plane puncture technique is advantageous in overcoming the difficulties associated with puncturing through deep vein calcification in patients with secondary hyperparathyroidism. This technique also diminishes the likelihood of complications such as venous thrombosis.
Topics: Female; Humans; Aged; Catheterization, Central Venous; Phlebotomy; Jugular Veins; Thrombosis; Ultrasonography, Interventional; Hyperparathyroidism, Secondary; Vascular Calcification
PubMed: 38379277
DOI: 10.12659/AJCR.943270 -
Indian Journal of Endocrinology and... 2023Primary hyperparathyroidism (PHPT) is rarely associated with the occurrence of acute or chronic pancreatitis, requiring complex perioperative management. This study...
BACKGROUND
Primary hyperparathyroidism (PHPT) is rarely associated with the occurrence of acute or chronic pancreatitis, requiring complex perioperative management. This study aimed to assess the prevalence and disease characteristics of pancreatitis in PHPT.
MATERIALS AND METHODS
This study is a clinicopathological analysis of the medical records of patients who were diagnosed with PHPT with pancreatitis between 1989 and 2021 in the Endocrine Surgery department, SGPGI, Lucknow.
RESULTS
Out of 548 PHPT cases, 44 (8.03%) were found to be associated with pancreatitis. The mean age was 33.57 years (15-65 years); 5 were ≤20 years, while 26 were ≤30 years of age. There were 27 males and 17 females. Twenty-one cases were of acute (11 acute, nine recurrent acute, one acute on chronic), whereas 23 were of chronic pancreatitis (six chronic calcific pancreatitis). The major clinical presentation of PHPT with pancreatitis was abdominal pain (65.91%). The mean number of attacks per patient in recurrent acute pancreatitis was two. Mean PTH levels were 68.19 pmol/L. The mean tumor size (in the largest dimension) was 2.79 ± 1.4 cm while the mean tumor weight was 4.91 g. Nephrolithiasis was associated with 25 cases. An association with multiple endocrine neoplasia type 1 syndrome was seen in one case. The final histopathological diagnosis was parathyroid carcinoma in two, hyperplasia in three, and parathyroid adenoma in 39 cases. Normocalcemia was seen in 27.2%, hypercalcemic crisis in 15.9%, and 25% of patients required semi-emergency parathyroidectomy. The outcome was favorable in all, as none had any further attacks of pancreatitis.
CONCLUSION
In our study, the prevalence of pancreatitis in PHPT cases was 8.03%. The majority of patients were young. Normocalcemia was seen in 12 patients, so even if calcium levels are normal, PHPT should be suspected in young patients with pancreatitis. Parathyroidectomy resulted in the complete resolution of symptoms of pancreatitis in all 44 patients.
PubMed: 38371176
DOI: 10.4103/ijem.ijem_169_23 -
Case Reports in Oncology 2024Parathyroid carcinoma is a rare malignant endocrine tumor that is usually associated with primary hyperparathyroidism. The coexistence of parathyroid carcinoma and renal...
INTRODUCTION
Parathyroid carcinoma is a rare malignant endocrine tumor that is usually associated with primary hyperparathyroidism. The coexistence of parathyroid carcinoma and renal hyperparathyroidism is a rare phenomenon. Hence, we present a case of parathyroid carcinoma in a patient with tertiary hyperparathyroidism.
CASE PRESENTATION
Our patient is a 31-year-old woman with a past medical history of end-stage renal failure (ESRF), on hemodialysis for the past 18 years. She was referred by her nephrologist to the endocrine surgery department for consideration of parathyroidectomy in view of long-standing tertiary hyperparathyroidism complicated by hypercalcemia. Bedside ultrasonography scan (US) of the thyroid revealed three parathyroid glands and a hypoechoic right lower pole thyroid nodule with central calcification. Fine-needle aspiration cytology was performed for the suspected thyroid nodule on the same day, which eventually yielded a follicular lesion of undetermined significance. A right hemithyroidectomy and total parathyroidectomy with deltoid implantation was performed. Intraoperative exploration revealed that the thyroid nodule noted at initial US was found to be the right superior parathyroid gland invading into the right thyroid itself. The right superior parathyroid gland was excised en bloc with the right hemithyroidectomy. Post-operatively, the patient was hypocalcemic but was discharged well on post-operative day 5. Histopathological diagnosis of the right hemithyroidectomy specimen containing the right superior parathyroid gland was consistent with that of parathyroid carcinoma.
CONCLUSION
Parathyroid carcinoma is a rare entity that is difficult to diagnose. In patients with ESRF, the presence of concurrent tertiary hyperparathyroidism makes this even more challenging.
PubMed: 38371168
DOI: 10.1159/000536444 -
Journal of Surgical Case Reports Feb 2024Mediastinal ectopic parathyroid adenomas, a rare cause of primary hyperparathyroidism, has evolved significantly with the advent of robotic-assisted surgery. Traditional...
Mediastinal ectopic parathyroid adenomas, a rare cause of primary hyperparathyroidism, has evolved significantly with the advent of robotic-assisted surgery. Traditional surgical approaches, while effective, may be associated with considerable morbidity and extended recovery periods. This study aims to evaluate the effectiveness, precision, and postoperative outcomes of robotic thoracoscopy with parathyroidectomy in the management of mediastinal ectopic parathyroid adenomas. A case of a 70-year-old man with a history of primary hyperparathyroidism underwent a successful left robotic thoracoscopy with parathyroidectomy in an ectopic mediastinal parathyroid adenoma. The robotic approach demonstrated advantages such as enhanced precision and minimal invasiveness. However, the learning curve and cost implications of this technology were identified as considerations. Robotic thoracoscopy with parathyroidectomy underscores the potential of robotic surgery in revolutionizing the management of mediastinal ectopic parathyroid adenomas, offering promising precision, emphasizing the need for ongoing research, and evaluation to optimize this innovative surgical method.
PubMed: 38370589
DOI: 10.1093/jscr/rjae044 -
BJR Case Reports Jan 2024We present two rare cases of parathyroid carcinomas associated with multiple brown tumours. Plain radiographs, computed tomography, and neck ultrasonography revealed the...
We present two rare cases of parathyroid carcinomas associated with multiple brown tumours. Plain radiographs, computed tomography, and neck ultrasonography revealed the presence of bone and parathyroid tumours. Despite the use of 99m Tc-methoxy isobutyl isonitrile (99mTc-MIBI) or F-fluorodeoxyglucose-positron emission tomography (18F-FDG PET)/CT, it was difficult to differentiate bone metastases from brown tumours. Parathyroid carcinoma was confirmed by histopathological examination following parathyroidectomy, resulting in spontaneous bone lesion improvement. In patients with parathyroid carcinoma presenting with bone lesions suggestive of metastasis, understanding the potential for brown tumour accumulation through 99mTc-MIBI or 18F-FDG PET/CT is pivotal. With this understanding, it is possible to diagnose brown tumours with parathyroidectomy and follow up for improvement of bone lesion and avoid invasive biopsy or surgery.
PubMed: 38352261
DOI: 10.1093/bjrcr/uaad003 -
Frontiers in Endocrinology 2024Hereditary primary hyperparathyroidism (PHPT) accounts for 5-10% of all PHPT cases, necessitating genetic testing for diagnosis and management. Among these,...
BACKGROUND
Hereditary primary hyperparathyroidism (PHPT) accounts for 5-10% of all PHPT cases, necessitating genetic testing for diagnosis and management. Among these, hyperparathyroidism-jaw tumor syndrome (HPT-JT) is an autosomal dominant disorder caused by mutations with variable clinical presentations and incomplete symptoms.
CASE SUMMARY
The proband, diagnosed with PHPT, underwent parathyroidectomy at the age of 41 with pathological examination of parathyroid carcinoma (PC). Hereditary PHPT was initially suspected due to the early-onset PHPT and family history. Genetic testing identified a heterozygous mutation, NM_024529.4: c. 687_688delAG (p. Arg229Serfs*37). Even in the absence of jaw tumors, the diagnosis of HPT-JT was confirmed based on the discovery of renal cysts. A secondary thyroidectomy was performed to reduce the risk of recurrence.
CONCLUSION
Genetic testing is strongly recommended in cases of early-onset PHPT, family history, jaw tumors, renal and uterine involvement, atypical parathyroid tumors, and PC. This testing provides valuable information for personalized management, and counseling is available for affected families.
Topics: Humans; Adenoma; Fibroma; Hyperparathyroidism; Jaw Neoplasms; Mutation; Parathyroid Neoplasms; Tumor Suppressor Proteins; Adult
PubMed: 38348418
DOI: 10.3389/fendo.2024.1330185 -
The Journal of Clinical Endocrinology... Feb 2024Current evidence of cardiovascular (CV) risk in primary hyperparathyroidism (PHPT) is still inconsistent.
CONTEXT
Current evidence of cardiovascular (CV) risk in primary hyperparathyroidism (PHPT) is still inconsistent.
OBJECTIVE
To prospectively investigate changes of early atherosclerosis in patients with PHPT undergoing parathyroidectomy (PTx) or conservative management, according to Consensus criteria.
METHODS
Biochemical parameters of PHPT, CV risk factors (systolic and diastolic blood pressure-BP-, total-, HDL- and LDL-cholesterol, triglyceride, glycosilated hemoglobin, and HOMA-IR), and carotid intima-media thickness (IMT) and plaque were assessed in 52 consecutive postmenopausal PHPT patients both at baseline and ≥24 months after surgery (PTx: n = 22) or conservative management (no-PTx: n = 30).
RESULTS
At baseline, PTx and no-PTx showed comparable age, BMI, renal function, 25(OH)D levels, and did not differ for CV risk factors, IMT and plaques, nor for the prevalence of smoking, diabetes mellitus, antihypertensive or statin therapy, while differing for all parameters characterizing PHPT. Follow-up length in PTx was longer (p = 0.004) than in no-PTx. Parameters characterizing PHPT significantly improved ≥24 months after surgery, whereas in no-PTx serum phosphate slightly decreased and PTH increased. Systolic and diastolic BP increased at follow-up in both groups, while other CV risk factors did not significantly vary. In PTx IMT did not significantly vary after surgery (0.85 ± 0.14 to 0.89 ± 0.22 mm, p = 0.366), whereas it significantly increased in no-PHPT (0.80 ± 0.18 to 0.93 ± 0.23 mm, p = 0.008), even adjusting for BP values. Plaque prevalence and incidence did not significantly differ in the two groups.
CONCLUSION
Our results suggest that in postmenopausal PHPT patients subclinical atherosclerosis could be halted by PTx, whereas it worsens over time in not operated patients with milder disease.
PubMed: 38345411
DOI: 10.1210/clinem/dgae053 -
Case Reports in Endocrinology 2024Primary hyperparathyroidism (PHPT) and familial hypocalciuric hypercalcaemia (FHH) are common causes of hypercalcaemia. Patients are mostly asymptomatic in the case of...
Case Presentation: Functional Assessment of a Variant Identified in a Patient with Hypercalcaemia Confirms Familial Hypocalciuric Hypercalcaemia in the Patient and a Sister Previously Misdiagnosed with Primary Hyperparathyroidism.
BACKGROUND
Primary hyperparathyroidism (PHPT) and familial hypocalciuric hypercalcaemia (FHH) are common causes of hypercalcaemia. Patients are mostly asymptomatic in the case of FHH and often so in the case of PHPT. In addition, biochemical parameters show considerable overlap, making differential diagnosis difficult. Genetic screening for inactivating variants in the calcium-sensing receptor () gene that are causative of FHH assists with the diagnosis since such variants are not generally associated with PHPT. However, novel variants must undergo functional assessment before they can be definitively assigned a causative role in FHH. . We describe a 73-year-old female (patient A) who presented with mild parathyroid hormone (PTH)-dependent hypercalcaemia and a history of osteoporosis. Family history revealed that her sister (patient B) had presented a decade earlier with symptoms of PHPT including a history of mild hypercalcaemia and multiple renal calculi, prompting parathyroid surgery. However, a subtotal parathyroidectomy did not resolve her hypercalcaemia long term. On this basis, genetic screening was performed on patient A. This identified a heterozygous variant in the , NM_000388.4:c.T101C: p.Leu34Pro (L34P). Functional analysis showed that the L34P variant was unable to produce mature, dimerized receptor and did not respond to Ca ions. Adopting American College of Medical Genetics-based guidelines, the variant was classified as 'Pathogenic (II)'. Patient B was subsequently found to carry the L34P variant heterozygously, confirming a diagnosis of FHH, not PHPT.
CONCLUSION
This study shows the importance of examining patient's family history in providing clues to the diagnosis in isolated cases of hypercalcaemia. In this case, history of a sister's unsuccessful parathyroidectomy prompted genetic screening in a patient who might otherwise have undergone inappropriate parathyroid surgery. Screening detected an inactivating variant, firming up a diagnosis of FHH. These studies reaffirm the requirement for functionally assessing novel variants prior to assigning causality to FHH.
PubMed: 38343604
DOI: 10.1155/2024/6652801