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1.
Langenbecks Arch Surg ; 409(1): 300, 2024 Oct 08.
Article in English | MEDLINE | ID: mdl-39377972

ABSTRACT

BACKGROUND AND HYPOTHESIS: Parathyroid carcinoma (PTTC) is a rare malignant endocrine tumor seen in up to 1-2% of all cases of primary hyperparathyroidism. However, incidence of parathyroid carcinoma in renal hyperparathyroidism is a rare phenomenon. We aimed to evaluate the outcomes of PTTC in renal hyperparathyroidism published in the literature. METHODS: Cohort review of parathyroid cancer cases reported in Medline (via PubMed), COCHRANE and EMBASE between the period 1985 - 2023 in patients with renal hyperparathyroidism. RESULTS: A total of 48 patients (20 M: 28F), with a mean age of 49.8 (± 11.7 SD: range 20-75) years. Dialysis vintage was for a period of 8.9 (± 7.2; range 6 months to 40 years). The mean preoperative values were as follows: serum corrected calcium-2.87 IQR 2.56-3.01), PTH - 221.8 (IQR 86.6 -257.2 pmol/L) and serum phosphate - 2.07 (IQR 1.72-2.28) mmol/L. Preoperative imaging was in the form of ultrasound of the neck in 21 of 48 (44%), MIBI scan in 27/48 (56%), contrast enhanced computerized tomography in 14/48 (29%) and MRI neck in 1/48 (2%). The mean size of the cancer was 2.7 (± 1.35) cm and weight of the gland ranged between 0.9 to 4.98 g. 18/48 (37%) patients underwent a total parathyroidectomy and 30/48 (63%) had subtotal parathyroidectomy. En bloc excision of the tumour along with the thyroid along and central compartment lymph nodes was only performed in 12/48 (25%), of whom 9 (19%) had it performed at index surgery, whereas in the rest was done for persistent or recurrent disease. After a mean follow up of 34 months, 14 (29%) had local recurrence, 1 (2%) had distant metastasis to the skeletal system, and 12 (25%) to the lungs. Cohort mortality was 6 (13%) due to refractory hypercalcemia. CONCLUSIONS: Parathyroid carcinoma in renal hyperparathyroidism is rare but when encountered, en bloc excision with parathyroidectomy provides the best chance of cure. Recurrences can be difficult to treat but may be needed to treat intractable hypercalcaemia.


Subject(s)
Kidney Failure, Chronic , Parathyroid Neoplasms , Parathyroidectomy , Humans , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/complications , Parathyroid Neoplasms/pathology , Kidney Failure, Chronic/complications , Adult , Male , Female , Middle Aged , Aged , Renal Dialysis , Cohort Studies
2.
Eur J Endocrinol ; 191(4): 416-425, 2024 Sep 30.
Article in English | MEDLINE | ID: mdl-39365596

ABSTRACT

OBJECTIVE: Atypical parathyroid tumor (aPT) and parathyroid carcinoma (PC) are extremely rare parathyroid neoplasms, accounting together for <2% of all parathyroid tumors. They often present an overlapping clinical phenotype, sharing clinical, biochemical, and some histological features. They are distinguished only by the presence of local invasion, and lymph nodes or distant metastasis, which are all absent in aPTs. To date, only few studies have compared clinical presentation and features between aPTs and PCs. Our purpose was to conduct a retrospective study on a multicenter Italian database of aPT and PC patients. DESIGN AND METHODS: We comparatively analyzed main features of aPT (n = 57) and PC (n = 74) patients collected at 15 major endocrinology and endocrine surgery centers in Italy. RESULTS AND CONCLUSIONS: Atypical parathyroid tumors and PCs showed no significant differences in many clinical features and presented similar values of elevated parathyroid hormone and total serum calcium. Renal complications, namely nephrolithiasis and nephrocalcinosis, appeared to be more common in PC, with a significantly higher rate of renal colic, regardless of total serum calcium levels and 24-h calciuria. Parathyroid carcinomas showed significantly higher postoperative disease persistence and recurrence rates, presumably due to an uncomplete resection of the primary tumor in 23.5% of cases and/or presence of unremoved active metastasis, but they had similar disease-free mean time after surgery than aPT. To deepen the study of malignant parathyroid tumors, the institution of a novel Italian retro-prospective multicenter registry of aPTs and PCs is currently ongoing, and a dedicated PC European registry has been recently activated.


Subject(s)
Databases, Factual , Parathyroid Neoplasms , Humans , Parathyroid Neoplasms/pathology , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/epidemiology , Female , Male , Italy/epidemiology , Middle Aged , Retrospective Studies , Aged , Adult , Parathyroid Hormone/blood , Carcinoma/pathology , Carcinoma/surgery , Carcinoma/epidemiology , Parathyroidectomy , Calcium/blood
3.
BMC Endocr Disord ; 24(1): 193, 2024 Sep 19.
Article in English | MEDLINE | ID: mdl-39294625

ABSTRACT

BACKGROUND: Although the skeleton remains a common target of primary hyperparathyroidism, the classic bone disease "osteitis fibrosa cystica" is currently rare due to early diagnosis. This case represents severe classic bone manifestations of primary hyperparathyroidism due to delayed diagnosis and delayed medical attention. CASE PRESENTATION: A 19-year-old young female was symptomatically managed for chronic back pain and nonspecific bone pain in the small joints of both hands over 2 months by a general practitioner. The patient had delayed seeking for treatment for 3 months. Later, she was evaluated for tuberculosis, hematological malignancies and rheumatic disorders following a fractured T12 vertebra and underwent pedicle screw fixation. However, clinical examination and investigations, including biochemistry, imaging and histology, ruled out the above conditions. Unfortunately, serum calcium level was not performed at the initial presentation. Later, primary hyperparathyroidism was diagnosed on the basis of moderate hypercalcaemia and elevated intact PTH levels (2064 pg/ml). She had sufficient vitamin D levels and normal kidney function. Her DXA scan revealed severe secondary osteoporosis with the lowest Z score of -8 at the total lumbar spine. Ultrasonography of the thyroid revealed a hypo echoic mass in the left lower neck, and localization studies with technetium-99 m sestamibi and 4D-CT revealed a left inferior parathyroid adenoma (1.6 × 1.5 × 1.6 cm). CT scan also revealed brown tumors in the mandible and vertebrae and diffuse bony changes in the skull, sternum, humerus and vertebrae. Her radiographs revealed subperiosteal bone resorption on the radial aspects of the middle and distal phalanges and brown tumors in both the ulna and fibula. We excluded MEN and other hereditary syndromes in our patient with a personal and family history and with a normal pituitary hormone profile because of poor resources for genetic testing. She underwent parathyroid adenoma excision, and the postoperative period was complicated with hungry bone syndrome, requiring high doses of calcium and active vitamin D supplements. These supplements were gradually weaned off over 6 months, and she recovered with normal biochemical investigations. Histology revealed parathyroid adenoma without malignant features. CONCLUSION: In developing countries where routine calcium screening is not available, clinicians should be aware of various manifestations of primary hyperparathyroidism to allow diagnosis as soon as possible without delay to prevent further progression, as it is a treatable condition.


Subject(s)
Spinal Fractures , Humans , Female , Spinal Fractures/etiology , Spinal Fractures/surgery , Spinal Fractures/pathology , Young Adult , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/complications , Hyperparathyroidism, Primary/etiology , Hyperparathyroidism, Primary/pathology , Hyperparathyroidism, Primary/diagnosis , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/complications , Parathyroid Neoplasms/pathology , Adolescent , Adenoma/complications , Adenoma/pathology , Adenoma/surgery , Osteitis Fibrosa Cystica/etiology , Osteitis Fibrosa Cystica/pathology
4.
BMJ Open ; 14(9): e082901, 2024 Sep 05.
Article in English | MEDLINE | ID: mdl-39242156

ABSTRACT

OBJECTIVES: Preoperative and intraoperative diagnostic tools influence the surgical management of primary hyperparathyroidism (PHPT), whereby their performance of classification varies considerably for the two common causes of PHPT: solitary adenomas and multiglandular disease. A consensus on the use of such diagnostic tools for optimal perioperative management of all PHPT patients has not been reached. DESIGN: A decision tree model was constructed to estimate and compare the clinical outcomes and the cost-effectiveness of preoperative imaging modalities and intraoperative parathyroid hormone (ioPTH) monitoring criteria in a 21-year time horizon with a 3% discount rate. The robustness of the model was assessed by conducting a one-way sensitivity analysis and probabilistic uncertainty analysis. SETTING: The US healthcare system. POPULATION: A hypothetical population consisting of 5000 patients with sporadic, symptomatic or asymptomatic PHPT. INTERVENTIONS: Preoperative and intraoperative diagnostic modalities for parathyroidectomy. MAIN OUTCOME MEASURES: Costs, quality-adjusted life-years (QALYs), net monetary benefits (NMBs) and clinical outcomes. RESULTS: In the base-case analysis, four-dimensional (4D) CT was the least expensive strategy with US$10 276 and 15.333 QALYs. Ultrasound and 99mTc-Sestamibi single-photon-emission CT/CT were both dominated strategies while 18F-fluorocholine positron emission tomography was cost-effective with an NMB of US$416 considering a willingness to pay a threshold of US$95 958. The application of ioPTH monitoring with the Vienna criterion decreased the rate of reoperations from 10.50 to 0.58 per 1000 patients compared to not using ioPTH monitoring. Due to an increased rate of bilateral neck explorations from 257.45 to 347.45 per 1000 patients, it was not cost-effective. CONCLUSIONS: 4D-CT is the most cost-effective modality for the preoperative localisation of solitary parathyroid adenomas and multiglandular disease. The use of ioPTH monitoring is not cost-effective, but to minimise clinical complications, the Miami criterion should be applied for suspected solitary adenomas and the Vienna criterion for multiglandular disease.


Subject(s)
Cost-Benefit Analysis , Decision Trees , Hyperparathyroidism, Primary , Parathyroidectomy , Quality-Adjusted Life Years , Humans , Parathyroidectomy/economics , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/diagnosis , Hyperparathyroidism, Primary/economics , Decision Support Techniques , Parathyroid Hormone/blood , Four-Dimensional Computed Tomography , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/diagnosis , Treatment Outcome
5.
BMC Endocr Disord ; 24(1): 208, 2024 Sep 30.
Article in English | MEDLINE | ID: mdl-39350103

ABSTRACT

BACKGROUND: Parathyroid adenoma is the primary cause of primary hyperparathyroidism, commonly presenting with elevated parathyroid hormone (PTH) and blood calcium levels. Chronic primary hyperparathyroidism often results in bone destruction, resulting in the formation of brown tumors. The preferred clinical treatment for parathyroid adenoma is parathyroidectomy. Postoperative pancytopenia, although rare, is a critical complication that warrants further investigation into its mechanisms and management strategies. CASE PRESENTATION: We present a case of a 59-year-old female patient who was admitted due to nausea and vomiting. Positron emission tomography-computed tomography (PET-CT) revealed a mass posterior to the left thyroid lobe and multiple areas of fibrocystic osteitis throughout the body. Hematological tests showed elevated serum calcium and parathyroid hormone (PTH) levels. The patient subsequently underwent parathyroidectomy, and pathological examination confirmed the presence of a parathyroid adenoma. Postoperatively, the patient developed pancytopenia and received symptomatic treatment such as correction of anemia and elevation of white blood. At the two-month follow-up, all indicators had returned to normal. CONCLUSIONS: Pancytopenia is commonly seen in bone marrow diseases, infections and immune-related disorders, nutritional deficiencies, and metabolic diseases. This case confirms that pancytopenia can also occur postoperatively in patients with parathyroid adenoma. Therefore, Clinicians should be aware of the potential for postoperative pancytopenia following parathyroidectomy and the need for prompt management.


Subject(s)
Adenoma , Pancytopenia , Parathyroid Neoplasms , Parathyroidectomy , Postoperative Complications , Humans , Female , Pancytopenia/etiology , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/complications , Parathyroid Neoplasms/pathology , Middle Aged , Adenoma/surgery , Adenoma/complications , Adenoma/pathology , Postoperative Complications/etiology , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/complications , Hyperparathyroidism, Primary/etiology , Osteitis Fibrosa Cystica/etiology
6.
Medicine (Baltimore) ; 103(38): e39717, 2024 Sep 20.
Article in English | MEDLINE | ID: mdl-39312325

ABSTRACT

RATIONALE: Hungry bone syndrome (HBS) is a forgotten and underdiagnosed cause. Postoperative HBS refers to patients with high bone turnover before surgery, but after surgery, the inhibition of osteoclast resorption by intact parathyroid hormone suddenly decreases, resulting in a sudden increase in the amount of calcium resorbed by the bone, and a rapid, severe and persistent hypocalcemia, which may be accompanied by hypophosphatemia and hypomagnesemia. We present a case with information about HBS and related complications after parathyroidectomy (PTX). PATIENT CONCERNS: The patient was a 57-year-old woman who presented to the hospital with "pain in both ankles for more than 3 years and in both knees for more than 2 years." DIAGNOSES: A parathyroid mass was found preoperative. Unilateral resection of the lesion was performed under general anesthesia. On gross examination, the mass was reddish brown in color, about 2.9 × 2.5 × 2.3 cm, with abundant blood supply. Postoperative pathology diagnosed parathyroid adenoma. INTERVENTIONS: The patient was diagnosed with HBS on day 3 post-PTX, which lasted for 9 days. OUTCOMES: After active calcium supplementation and other pharmacological interventions, her test parameters gradually returned to normal and she was discharged on the 13th day after surgery. LESSONS: Using the case of a patient with primary hyperparathyroidism with HBS lasting 9 days after PTX for diagnosis and management, we aimed to summarize possible predictors and perioperative management strategies to reduce the incidence, severity, and duration of postoperative HBS.


Subject(s)
Hyperparathyroidism, Primary , Hypocalcemia , Parathyroidectomy , Postoperative Complications , Humans , Female , Middle Aged , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/complications , Hyperparathyroidism, Primary/diagnosis , Parathyroidectomy/adverse effects , Hypocalcemia/etiology , Postoperative Complications/etiology , Syndrome , Calcium/blood , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/complications
7.
Ugeskr Laeger ; 186(29)2024 Jul 15.
Article in Danish | MEDLINE | ID: mdl-39115216

ABSTRACT

Primary hyperparathyroidism (PHPT) is the most prevalent cause of hypercalcaemia, affecting 0.3% of the population. The only curative procedure is parathyroidectomy. Ectopic adenomas are challenging to localize and frequently result in persistent PHPT. This is a case report of a 29-year-old male patient who was diagnosed with PHPT prior to neck surgery and reoperated with bilateral neck exploration. However, the PHPT was not cured, until diagnostic CT with contrast had helped localizing a 1 cm ectopic parathyroid adenoma in the right horn of the thymus gland. The adenoma was then removed successfully.


Subject(s)
Adenoma , Hyperparathyroidism, Primary , Parathyroid Neoplasms , Tomography, X-Ray Computed , Humans , Male , Adult , Parathyroid Neoplasms/diagnostic imaging , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/pathology , Adenoma/diagnostic imaging , Adenoma/surgery , Adenoma/pathology , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/diagnostic imaging , Hyperparathyroidism, Primary/etiology , Choristoma/diagnostic imaging , Choristoma/surgery , Choristoma/pathology , Parathyroidectomy
8.
World J Gastroenterol ; 30(26): 3247-3252, 2024 Jul 14.
Article in English | MEDLINE | ID: mdl-39086634

ABSTRACT

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.


Subject(s)
Hypercalcemia , Multiple Endocrine Neoplasia Type 1 , Parathyroid Neoplasms , Parathyroidectomy , Humans , Multiple Endocrine Neoplasia Type 1/surgery , Multiple Endocrine Neoplasia Type 1/diagnosis , Multiple Endocrine Neoplasia Type 1/complications , Multiple Endocrine Neoplasia Type 1/pathology , Male , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/diagnosis , Parathyroid Neoplasms/pathology , Parathyroid Neoplasms/complications , Middle Aged , Hypercalcemia/diagnosis , Hypercalcemia/etiology , Hypercalcemia/blood , Adenoma/surgery , Adenoma/diagnosis , Adenoma/pathology , Adenoma/blood , Duodenal Neoplasms/surgery , Duodenal Neoplasms/diagnosis , Duodenal Neoplasms/pathology , Hypophosphatemia/etiology , Hypophosphatemia/diagnosis , Abdominal Pain/etiology , Abdominal Pain/diagnosis , Neuroendocrine Tumors/surgery , Neuroendocrine Tumors/diagnosis , Neuroendocrine Tumors/blood , Neuroendocrine Tumors/pathology , Diarrhea/etiology , Diarrhea/diagnosis , Early Detection of Cancer/methods , Gastroscopy , Treatment Outcome
9.
JAMA Otolaryngol Head Neck Surg ; 150(9): 756-762, 2024 Sep 01.
Article in English | MEDLINE | ID: mdl-39023906

ABSTRACT

Importance: Accurate preoperative localization is critical to success in targeted parathyroidectomy for primary hyperparathyroidism. Objective: To determine if the association between the long axis of a parathyroid adenoma (PTA) candidate and strap musculature on sagittal ultrasonography (US) can be used to predict the embryologic origin of the gland. Design, Setting, and Participants: This diagnostic study was performed using the Stanford Research Repository. Patients 18 years or older with primary hyperparathyroidism who underwent parathyroidectomy between January 2009 and October 2021 were considered. Additional inclusion criteria were having clear sagittal view of the adenoma candidate on US, confirmation of the gland of origin intraoperatively, and confirmation of hypercellular parathyroid on final pathology. Data were analyzed from October 2021 to June 2022. Exposures: B-mode US and surgical parathyroidectomy. Main Outcomes and Measures: The index test was using US to measure the angle between the long axis of an adenoma candidate and the strap musculature in the sagittal plane. This angle was used to test whether inferior and superior PTAs could be accurately assigned. The hypothesis was formulated prior to data collection. Results: A total of 426 patients (mean [range] age, 61.1 [20-96] years; 316 [74.2%] female) with 442 adenomas met inclusion criteria. Of the 442 adenomas, 314 (71.0%) had measurable angles, of which 204 (46.2%) were assigned a superior origin, 238 (53.8%) were assigned an inferior origin, and 128 (29%) were indeterminate. Of the surgically identified superior PTAs, 144 (70.6%) had a definable angle, and of the surgically identified inferior PTAs, 170 (71.4%) had a definable angle. The receiver operating characteristic analysis found 94° as the optimized angle for differentiating true negatives from true positives, with an overall sensitivity of 74% and specificity of 72%. This supported using 90° as a break point for US review. True positives were considered superior adenomas with an angle greater than 90°; true negatives were inferior adenomas with an angle less than 90°. Sensitivity, specificity, positive predictive value, negative predictive value, and accuracy of angulation analysis for determining PTA origin were 72.2% (95% CI, 64.9%-79.5%), 73.5% (95% CI, 66.9%-80.1%), 69.8% (95% CI, 62.5%-77.1%), 75.8% (95% CI, 69.3%-82.3%), and 72.9%, respectively. A subgroup analysis of 426 adenomas using the posterior carotid artery border on transverse US as a surrogate for predicting gland origin showed the following for sensitivity, specificity, positive predictive value, negative predictive value, and accuracy: 49.5% (95% CI, 42.6%-56.4%), 82.3% (95% CI, 77.3%-87.3%), 71.4% (95% CI, 63.9%-78.9%), 64.6% (95% CI, 59.1%-70.1%), and 66.9%, respectively. Conclusions and Relevance: This diagnostic study showed that PTA angulation on sagittal plane US can be used to predict gland of origin and guide surgery. The relationship between adenoma and posterior carotid artery border on transverse US can also be used to predict gland origin. These easy-to-apply US-based tests can be used in conjunction with other imaging modalities to guide targeted parathyroidectomy.


Subject(s)
Adenoma , Parathyroid Neoplasms , Parathyroidectomy , Ultrasonography , Humans , Parathyroid Neoplasms/diagnostic imaging , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/pathology , Female , Middle Aged , Male , Adenoma/diagnostic imaging , Adenoma/surgery , Adenoma/pathology , Aged , Adult , Aged, 80 and over , Hyperparathyroidism, Primary/diagnostic imaging , Hyperparathyroidism, Primary/surgery , Parathyroid Glands/diagnostic imaging , Retrospective Studies
11.
BMC Pregnancy Childbirth ; 24(1): 483, 2024 Jul 17.
Article in English | MEDLINE | ID: mdl-39020280

ABSTRACT

BACKGROUND: Hypercalcemia can be a rare contributor to acute pancreatitis (AP) in pregnancy. This is primarily due to primary hyperparathyroidism (PHPT), resulting from parathyroid carcinoma. We exhibited a case report to analyze the diagnosis and treatment during the onset of hypercalcemia-induced AP. CASE PRESENTATION: A 32-year-old primigravida presented with acute pancreatitis near full-term gestation. Following a cesarean delivery, there was a reduction in serum amylase and peripancreatic exudate, but her serum calcium concentrations persistently elevated over 4.0 mmol/L. Interventions to lower the hypercalcemia were only temporarily effective, until a high serum parathyroid hormone (PTH) concentration of 1404 pg/mL was detected. Ultrasound revealed a 31 mm × 24 mm hypoechoic oval nodule in the left lower lobe of the thyroid gland. She underwent a parathyroidectomy, resulting in a dramatic decrease in serum PTH level, from preoperative levels of 2051 pg/mL to 299 pg/mL just 20 minutes after removal. Similarly, her serum calcium declined from 3.82 mmol/L to 1.73 mmol/L within 24 hours postoperatively. The final histopathology suggested parathyroid carcinoma. CONCLUSION: When refractory hypercalcemia is present, serum PTH levels should be measured to determine PHPT. Parathyroidectomy is the optimal strategy for alleviating hypercalcemia and clarifying the underlying pathology.


Subject(s)
Hypercalcemia , Pancreatitis , Parathyroid Neoplasms , Parathyroidectomy , Pregnancy Complications, Neoplastic , Pregnancy Trimester, Third , Humans , Female , Hypercalcemia/etiology , Hypercalcemia/blood , Parathyroid Neoplasms/complications , Parathyroid Neoplasms/surgery , Pregnancy , Adult , Pancreatitis/etiology , Pancreatitis/complications , Pancreatitis/blood , Pregnancy Complications, Neoplastic/surgery , Parathyroid Hormone/blood , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/complications , Hyperparathyroidism, Primary/blood , Cesarean Section , Calcium/blood
12.
BMC Endocr Disord ; 24(1): 125, 2024 Jul 25.
Article in English | MEDLINE | ID: mdl-39054438

ABSTRACT

BACKGROUND: Parathyroid carcinoma (PC) is a rare malignancy, often diagnosed incidentally through postoperative pathological examination. The occurrence of nodular goiter, intrathyroidal parathyroid carcinoma, contralateral parathyroid adenoma (PA), and papillary thyroid microcarcinoma (PTMC) is extremely uncommon, which prompted us to report our case experience. CASE PRESENTATION: We describe a 67-year-old male who presented with a cervical mass causing tracheal compression, which prompted him to seek medical advice. Based on preoperative auxiliary examination results from color Doppler ultrasound, SPECT parathyroid imaging, and blood tests, he was initially diagnosed with a suspected parathyroid adenoma and nodular goiter. Excision of the right lobe and isthmus of the thyroid, and left superior parathyroid gland was conducted, which were sent to intraoperative frozen pathological examination. During intraoperative observation, adhesion around the right thyroid lobe was discovered. Consequently, right central area lymph node dissection was performed due to suspicion of an aggressive malignant tumor. Histology and immunohistochemistry analysis revealed incidental intrathyroidal parathyroid carcinoma, contralateral parathyroid adenoma, classical papillary thyroid microcarcinoma, and nodular goiter. CONCLUSION: Parathyroid carcinoma should be highly suspected when extremely high levels of PTH and severe hypercalcemia are present, which cannot be simply explained by a preoperatively localized parathyroid adenoma, especially when suspicious malignant adhesion is found during intraoperative exploration. In cases where multifocal thyroid nodules are associated with increased uptake of 99Tc-sestamibi, the possibility of coexisting carcinomas should be considered, not only for thyroid malignancy but also for the potential presence of intrathyroidal parathyroid carcinoma.


Subject(s)
Carcinoma, Papillary , Hyperparathyroidism, Primary , Neoplasms, Multiple Primary , Parathyroid Neoplasms , Thyroid Neoplasms , Humans , Male , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/pathology , Parathyroid Neoplasms/diagnostic imaging , Parathyroid Neoplasms/diagnosis , Thyroid Neoplasms/pathology , Thyroid Neoplasms/surgery , Thyroid Neoplasms/diagnosis , Aged , Carcinoma, Papillary/pathology , Carcinoma, Papillary/surgery , Carcinoma, Papillary/diagnosis , Carcinoma, Papillary/diagnostic imaging , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/etiology , Hyperparathyroidism, Primary/pathology , Hyperparathyroidism, Primary/diagnosis , Neoplasms, Multiple Primary/pathology , Neoplasms, Multiple Primary/surgery , Neoplasms, Multiple Primary/diagnosis , Incidental Findings
13.
BMJ Case Rep ; 17(7)2024 Jul 04.
Article in English | MEDLINE | ID: mdl-38964875

ABSTRACT

Variations in parathyroid gland positions often cause failure in initial parathyroid adenoma surgery, especially when imaging fails to localise the adenoma. This report describes a female patient with primary hyperparathyroidism for which preoperative localisation studies did not determine the position of the hyperfunctioning gland. The initial approach with bilateral cervical exploration and intraoperative parathyroid hormone monitoring was performed unsuccessfully. A mediastinal adenoma was suspected due to meticulous negative neck exploration and repeated negative images for a neck adenoma. Subsequently, a second approach involving mediastinal exploration was performed. After the removal of remnant thymic tissue in the mediastinal space, a significant drop in intraoperative parathyroid hormone levels was achieved. The pathological result confirmed the presence of a tiny pathological parathyroid adenoma within the thymus. At 6 months follow-up, postoperative biochemical assessment was consistent with normal calcium and parathyroid hormone levels.


Subject(s)
Adenoma , Hyperparathyroidism, Primary , Parathyroid Neoplasms , Humans , Parathyroid Neoplasms/complications , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/diagnostic imaging , Female , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/etiology , Hyperparathyroidism, Primary/complications , Hyperparathyroidism, Primary/diagnosis , Hyperparathyroidism, Primary/diagnostic imaging , Adenoma/complications , Adenoma/surgery , Adenoma/diagnostic imaging , Mediastinal Neoplasms/complications , Mediastinal Neoplasms/diagnostic imaging , Mediastinal Neoplasms/surgery , Parathyroid Hormone/blood , Middle Aged , Parathyroid Glands/diagnostic imaging , Parathyroidectomy
14.
Khirurgiia (Mosk) ; (6): 81-87, 2024.
Article in Russian | MEDLINE | ID: mdl-38888023

ABSTRACT

We present successful surgical treatment of a patient with chronic kidney disease (CKD) and hyperparathyroidism undergoing renal replacement therapy. At baseline, parathyroidectomy via cervical access was performed for parathyroid adenomas. After 6 years, clinical and laboratory relapse of disease required thoracoscopic resection of atypically located anterior mediastinal adenoma. This case demonstrates that this disease is one of the most difficult in modern medicine requiring a special approach in diagnosis and treatment. Patients with CKD and hyperparathyroidism need for follow-up, control of total and ionized serum calcium, inorganic phosphorus and parathormone, osteodensitometry, ultrasound and scintigraphy of thyroid and parathyroid glands, and, if necessary, CT or MRI of the neck and chest organs.


Subject(s)
Adenoma , Parathyroid Neoplasms , Parathyroidectomy , Humans , Middle Aged , Adenoma/surgery , Adenoma/complications , Adenoma/diagnosis , Hyperparathyroidism, Secondary/surgery , Hyperparathyroidism, Secondary/etiology , Hyperparathyroidism, Secondary/diagnosis , Mediastinal Neoplasms/surgery , Mediastinal Neoplasms/complications , Mediastinal Neoplasms/diagnosis , Mediastinum/surgery , Neoplasm Recurrence, Local/surgery , Parathyroid Glands/surgery , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/complications , Parathyroid Neoplasms/diagnosis , Parathyroidectomy/methods , Renal Insufficiency, Chronic/complications , Renal Insufficiency, Chronic/diagnosis , Thoracoscopy/methods , Treatment Outcome
15.
Ann Surg Oncol ; 31(10): 6865-6874, 2024 Oct.
Article in English | MEDLINE | ID: mdl-38879674

ABSTRACT

BACKGROUND: Distant metastatic parathyroid carcinoma (DM-PC) is a rare but often lethal entity with limited data about prognostic indicators. We sought to investigate the risk factors, patterns, and outcomes of DM-PC. METHODS: In this observational cohort study, 126 patients who underwent surgery for PC at a tertiary referral center from 2010 to 2023 were enrolled, among whom 38 had DMs. Univariate and multivariate Cox regression analyses were used to assess the effects of prognostic factors on DM. RESULTS: The cumulative incidence of DM was 14.1%, 33.8%, and 66.9% at 5, 10, and 20 years in the duration of disease course, respectively. DM-PC patients suffered a worse 5-year overall survival of 37.1% compared with 89.8% in the non-DM patients (p < 0.001). DM-PC patients also suffered more previous operations (p < 0.001), higher preoperative serum calcium (p<0.001) and parathyroid hormone (PTH) levels (p < 0.001), lower frequencies of R0 resection (p < 0.001), higher rates of pathological vascular invasion (p = 0.020), thyroid infiltration (p = 0.027), extraglandular extension (p = 0.001), upper aerodigestive tract (UAT) invasion (p < 0.001), and lymph node metastasis (p < 0.001). Multivariate Cox regression revealed that non-R0 resection (HR 6.144, 95% CI 2.881-13.106, p < 0.001), UAT invasion (HR 3.718, 95% CI 1.782-7.756, p < 0.001), and higher preoperative PTH levels (HR 1.001, 95% CI 1.000-1.001, p = 0.012) were independent risk factors of DM. CONCLUSIONS: Upper aerodigestive tract invasion and higher preoperative PTH levels might be risk factors for possible metastatic involvement of PC. R0 resection and closer surveillance should be considered in such cases to minimize the risk of DM and to optimize patient care.


Subject(s)
Parathyroid Neoplasms , Humans , Male , Female , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/pathology , Parathyroid Neoplasms/blood , Risk Factors , Middle Aged , Survival Rate , Prognosis , Follow-Up Studies , Aged , Lymphatic Metastasis , Adult , Parathyroidectomy , Neoplasm Invasiveness , Retrospective Studies , Incidence
17.
JAMA Otolaryngol Head Neck Surg ; 150(8): 658-665, 2024 Aug 01.
Article in English | MEDLINE | ID: mdl-38900416

ABSTRACT

Importance: Whether F18-choline (FCH) positron emission tomographic (PET)/computed tomographic (CT) scan can replace Tc99m-sestaMIBI (MIBI) single-photon emission (SPE)CT/CT as a first-line imaging technique for preoperative localization of parathyroid adenomas (PTA) in patients with primary hyperparathyroidism (PHPT) is unclear. Objective: To compare first-line FCH PET/CT vs MIBI SPECT/CT for optimal care in patients with PHPT needing parathyroidectomy and to compare the proportions of patients in whom the first-line imaging method resulted in successful minimally invasive parathyroidectomy (MIP) and normalization of calcemia 1 month after surgery. Design, Setting, and Participants: A French multicenter randomized open diagnostic intervention phase 3 trial was conducted. Patients were enrolled from November 2019 to May 2022 and participated up to 6 months after surgery. The study included adults with PHPT and an indication for surgical treatment. Patients with previous parathyroid surgery or multiple endocrine neoplasia type 1 (MEN1) were ineligible. Interventions: Patients were assigned in a 1:1 ratio to receive first-line FCH PET/CT (FCH1) or MIBI SPECT/CT (MIBI1). In the event of negative or inconclusive first-line imaging, they received second-line FCH PET/CT (FCH2) after MIBI1 or MIBI SPECT/CT (MIBI2) after FCH1. All patients underwent surgery under general anesthesia within 12 weeks following the last imaging. Clinical and biologic (serum calcemia and parathyroid hormone levels) assessments were performed 1 and 6 months after surgery. Main Outcomes and Measures: The primary outcome was a true-positive first-line imaging-guided MIP combined with uncorrected serum calcium levels of 2.55 mmol/l or less 1 month after surgery, corresponding to the local upper limit of normality. Results: Overall, 57 patients received FCH1 (n = 29) or MIBI1 (n = 28). The mean (SD) age of patients was 62.8 (12.5) years with 15 male (26%) and 42 female (74%) patients. Baseline patient characteristics were similar between groups. Normocalcemia at 1 month after positive first-line imaging-guided MIP was observed in 23 of 27 patients (85%) in the FCH1 group and 14 of 25 patients (56%) in the MIBI1 group. Sensitivity was 82% (95% CI, 62%-93%) and 63% (95% CI, 42%-80%) for FCH1 and MIBI1, respectively. Follow-up at 6 months with biochemical measures was available in 43 patients, confirming that all patients with normocalcemia at 1 month after surgery still had it at 6 months. No adverse events related to imaging and 4 adverse events related to surgery were reported. Conclusions: This randomized clinical trial found that first-line FCH PET/CT is a suitable and safe replacement for MIBI SPECT/CT. FCH PET/CT leads more patients with PHPT to correct imaging-guided MIP and normocalcemia than MIBI SPECT/CT thanks to its superior sensitivity. Trial Registration: ClinicalTrials.gov Identifier: NCT04040946.


Subject(s)
Choline , Hyperparathyroidism, Primary , Parathyroidectomy , Positron Emission Tomography Computed Tomography , Radiopharmaceuticals , Technetium Tc 99m Sestamibi , Humans , Female , Male , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/diagnostic imaging , Hyperparathyroidism, Primary/blood , Middle Aged , Positron Emission Tomography Computed Tomography/methods , Parathyroidectomy/methods , Aged , Fluorine Radioisotopes , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/diagnostic imaging , Parathyroid Neoplasms/complications , Adenoma/surgery , Adenoma/diagnostic imaging
18.
J ASEAN Fed Endocr Soc ; 39(1): 115-119, 2024.
Article in English | MEDLINE | ID: mdl-38863924

ABSTRACT

Primary hyperparathyroidism (PHPT) is rare in pregnancy. This condition is challenging to diagnose and manage due to the limited diagnostic and therapeutic options that are safe during pregnancy. If not diagnosed and managed in a timely manner, serious maternal and foetal complications may occur. We report two cases, one with surgical intervention and one without, to show the importance of timely surgical intervention and discuss the challenges in the management of PHPT in pregnancy.


Subject(s)
Hyperparathyroidism, Primary , Humans , Female , Pregnancy , Hyperparathyroidism, Primary/diagnosis , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/complications , Adult , Pregnancy Complications/diagnosis , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/complications , Parathyroid Neoplasms/diagnosis , Parathyroidectomy , Pregnancy Complications, Neoplastic/surgery , Adenoma/surgery , Adenoma/complications , Adenoma/diagnosis , Treatment Outcome
19.
Surg Clin North Am ; 104(4): 811-823, 2024 Aug.
Article in English | MEDLINE | ID: mdl-38944501

ABSTRACT

Primary hyperparathyroidism can be sporadic or part of a genetic syndrome, such as MEN1 or HPT-JT. Diagnosis of hereditary HPT requires a thorough history and physical. Parathyroidectomy is curative with greater than 95% success. However, some patients have persistent or recurrent disease requiring reoperation. Reoperative parathyroidectomy is technically challenging, and localizing the pathologic gland can difficult. Patients needing reoperation should undergo evaluation by a high-volume surgeon. Care should be taken to obtain all of the preoperative workup and operative note from the initial surgery. Radioguided parathyroidectomy can be safely and effectively performed in patients with hereditary HPT or undergoing reoperative surgery.


Subject(s)
Hyperparathyroidism, Primary , Parathyroidectomy , Recurrence , Humans , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/diagnosis , Hyperparathyroidism, Primary/genetics , Parathyroidectomy/methods , Reoperation , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/genetics , Parathyroid Neoplasms/diagnosis , Parathyroid Neoplasms/complications
20.
Surg Clin North Am ; 104(4): 791-798, 2024 Aug.
Article in English | MEDLINE | ID: mdl-38944499

ABSTRACT

Primary hyperparathyroidism (PHPT) is a disorder characterized by the autonomous overproduction of parathyroid hormone (PTH) that leads to hypercalcemia, multiple clinical sequelae, and heterogenous presentation. Whether PHPT is caused by a single benign adenoma (85%), multiglandular disease (15%), or parathyroid carcinoma (1%), surgery is the definitive treatment.


Subject(s)
Hyperparathyroidism, Primary , Parathyroid Neoplasms , Parathyroidectomy , Humans , Hyperparathyroidism, Primary/diagnosis , Hyperparathyroidism, Primary/surgery , Hyperparathyroidism, Primary/complications , Hyperparathyroidism, Primary/therapy , Parathyroidectomy/methods , Parathyroid Neoplasms/complications , Parathyroid Neoplasms/surgery , Parathyroid Neoplasms/diagnosis , Parathyroid Hormone/blood , Parathyroid Hormone/metabolism , Hypercalcemia/etiology , Hypercalcemia/diagnosis , Hypercalcemia/therapy , Adenoma/complications , Adenoma/surgery , Adenoma/diagnosis
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