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BMC Endocrine Disorders May 2023Parathyroid carcinoma is a rare disease with a frequency of 0.005% of all malignancies [1, 2]. Various aspects of its pathogenesis, diagnosis, and treatment remain...
BACKGROUND
Parathyroid carcinoma is a rare disease with a frequency of 0.005% of all malignancies [1, 2]. Various aspects of its pathogenesis, diagnosis, and treatment remain poorly understood. Furthermore, cases with secondary hyperparathyroidism are fewer. In this case report, we describe a case of left parathyroid carcinoma with secondary hyperparathyroidism.
CASE PRESENTATION
The patient was a 54-year-old woman who had been on hemodialysis since the age of 40 years. At 53 years of age, her calcium levels were high, and she was diagnosed with drug-resistant secondary hyperparathyroidism and was referred to our hospital for surgical treatment. Blood tests revealed calcium levels of 11.4 mg/dL and intact parathyroid hormone (PTH) levels of 1007 pg/mL. Neck ultrasonography revealed a 22-mm large round hypoechoic mass, partially indistinct margins, and D/W ratio > 1 at the left thyroid lobe. Computed tomography scans revealed a 20-mm nodule at the left thyroid lobe. No enlarged lymph nodes or distant metastases were noted. Tc-hexakis-2-methoxyisobutylisonitrile scintigraphy revealed an accumulation at the superior pole of the left thyroid lobe. Laryngeal endoscopy revealed paralysis of the left vocal cord, signifying recurrent nerve palsy due to parathyroid carcinoma. Based on these results, a diagnosis of secondary hyperparathyroidism and suspected left parathyroid carcinoma was made, and the patient underwent surgery. Pathology results revealed hyperplasia in the right upper and lower parathyroid glands. The left upper parathyroid gland showed capsular and venous invasion, and the diagnosis was left parathyroid carcinoma. At 4 months post-surgery, calcium levels improved to 8.7 mg/dL and intact PTH levels to 20 pg/mL, with no signs of recurrence.
CONCLUSIONS
We report a case of left parathyroid carcinoma associated with secondary hyperparathyroidism. Concomitant secondary hyperparathyroidism may cause mild hypercalcemia compared to parathyroid carcinoma alone due to the added modification of dialysis. Although our patient also presented with mild hypercalcemia, a D/W ratio > 1 on preoperative echocardiography and presence of recurrent nerve palsy on laryngoscopy led to the suspicion and treatment of parathyroid carcinoma preoperatively.
Topics: Humans; Female; Adult; Middle Aged; Parathyroid Neoplasms; Hypercalcemia; Calcium; Hyperparathyroidism, Secondary; Paralysis; Parathyroid Hormone
PubMed: 37193977
DOI: 10.1186/s12902-023-01370-x -
Journal of Medical Case Reports Sep 2023There is some evidence supporting the idea that double parathyroid adenomas represent a different entity from multiglandular hyperplasia; however, the distinction among...
BACKGROUND
There is some evidence supporting the idea that double parathyroid adenomas represent a different entity from multiglandular hyperplasia; however, the distinction among them is not straightforward.
CASE PRESENTATION
We described a case of primary hyperparathyroidism (PHPT) with pronounced clinical manifestations, caused by a bilateral giant parathyroid adenoma. A 34-year-old Hispanic/Latino male was diagnosed with PHPT caused by two giant parathyroid adenomas. The preoperative tests were neck ultrasound and computed tomography scan (CT-scan), showing two masses in the territory of parathyroid glands, bilaterally distributed (right was 31 × 18 × 19 mm and the left was 38 × 15 × 14 mm); sestamibi scan was not available. Parathyroid hormone (PTH) was highly elevated. Multiple complications of PHPT were present, such as bone lytic lesions, renal and pancreatic calcifications, and cardiovascular disease, the latter of which is an overlooked complication of PHPT. Multiple endocrine neoplasia 1 and 2 (MEN 1/2) were ruled out by the absence of clinical, biochemical, and radiological findings in other endocrine glands. The patient underwent subtotal parathyroidectomy with an intraoperative histopathological study; both intraoperative and definitive histopathology results were consistent with parathyroid adenomas; afterward, adequate suppression of PTH was assured, and later on, the patient presented hungry bone syndrome (HBS).
CONCLUSIONS
The diagnosis of double parathyroid adenomas is difficult. Regarding the similarities between multiglandular hyperplasia and parathyroid adenomas, this case report contributes to the further distinction between these two clinical entities. This case report also represents, in particular, the challenge of difficult diagnosis in places with limited resources, such as developing countries.
Topics: Humans; Adult; Parathyroid Neoplasms; Hyperplasia; Hypocalcemia; Bone Diseases; Parathyroid Hormone
PubMed: 37653552
DOI: 10.1186/s13256-023-04102-w -
Veterinary Surgery : VS Oct 2021To investigate (1a) agreement of ultrasonographic, surgical, and histopathologic findings in the diagnosis of a neoplastic etiology underlying primary...
Ambidirectional cohort study on the agreement of ultrasonography and surgery in the identification of parathyroid pathology, and predictors of postoperative hypocalcemia in 47 dogs undergoing parathyroidectomy due to primary hyperparathyroidism.
OBJECTIVE
To investigate (1a) agreement of ultrasonographic, surgical, and histopathologic findings in the diagnosis of a neoplastic etiology underlying primary hyperparathyroidism (PHPT), (1b) the ability of ultrasonographically determined parathyroid gland size to distinguish between malignant (carcinoma) and non-malignant (hyperplasia, adenoma) pathology, and (2) variables associated with postoperative hypocalcemia in dogs undergoing surgical treatment of PHPT.
STUDY DESIGN
Ambidirectional cohort study.
ANIMALS
Forty-seven client owned dogs with PHPT (34 retrospective; 13 prospective).
METHODS
Data were extracted from medical records. Method agreements were explored using Cohen's Kappa statistic. A receiver operating characteristic curve (ROC) was used to determine a cut-off separating parathyroid pathologies. Univariable and multivariable models assessed associations between postoperative hypocalcemia and potential risk factors.
RESULTS
Agreement of ultrasound and surgery for number and side of affected glands was 31/47 (65.9%) and 34/47 (72.3%), respectively. In 37/47 (78.7%) cases, parathyroid tissue was correctly assessed as pathologic by the surgeon. An ultrasonographic cut-off of ≥8.0 mm (ROC AUC = 0.82) best distinguished malignant from benign pathologies. Dogs with a preoperative serum ionized calcium (iCa) concentration ≥1.75 mEq/L had 7.5 times greater odds of becoming hypocalcemic postoperatively.
CONCLUSION
A fair agreement existed between ultrasonographic and surgical findings in dogs with PHPT. A parathyroid mass ≥8.0 mm on ultrasonographic examination was suggestive of malignancy, while dogs with a preoperative serum iCa concentration ≥1.75 mEq/L were at increased risk for postoperative hypocalcemia in this study.
CLINICAL SIGNIFICANCE
This study supports the use of bilateral cervical surgical exploration to identify abnormal parathyroid glands for the treatment of PHPT.
Topics: Animals; Cohort Studies; Dog Diseases; Dogs; Hyperparathyroidism, Primary; Hypocalcemia; Parathyroid Glands; Parathyroidectomy; Prospective Studies; Retrospective Studies; Ultrasonography
PubMed: 34365655
DOI: 10.1111/vsu.13707 -
Diagnostic and Interventional Radiology... Dec 2011To compare the accuracy of planar scintigraphy, single photon emission computed tomography (SPECT), SPECT-CT, and positron emission tomography (PET) with C-11 methionine... (Comparative Study)
Comparative Study
Accuracy of parathyroid imaging: a comparison of planar scintigraphy, SPECT, SPECT-CT, and C-11 methionine PET for the detection of parathyroid adenomas and glandular hyperplasia.
PURPOSE
To compare the accuracy of planar scintigraphy, single photon emission computed tomography (SPECT), SPECT-CT, and positron emission tomography (PET) with C-11 methionine for the pre-operative detection of parathyroid adenomas.
MATERIALS AND METHODS
We retrospectively evaluated the pre-operative studies of 60 patients with primary (n=56) and secondary (n=4) hyperparathyroidism. In 25/60 patients (Group 1), only planar scans were obtained, and additional SPECT and SPECT-CT were carried out in 35/60 patients (Group 2). PET or PET-CT with C-11 methionine was conducted in 8/60 patients (Group 3).
RESULTS
The results of the planar scans (Group 1) were true positive in 19/25 patients and false negative in 6/25 patients (sensitivity per patient, 76%). Histopathology confirmed 27 adenomas and two hyperplasia. Planar imaging identified 20/29 of these pathologies, whereas 9/29 were missed (sensitivity per adenoma, 69%). SPECT (Group 2) results were true positive in 34/35 patients and false negative in only one case (sensitivity per patient, 97%). On a lesion-based analysis, 38 adenomas were identified, and two were missed (sensitivity per adenoma, 95%). The sensitivities of SPECT and SPECT-CT were equal; however, SPECT-CT provided superior topographic information. C-11 methionine PET (Group 3) results were true positive in all eight patients. In one case, surgery confirmed two ipsilateral adenomas, only one of which was identified by PET (sensitivity per patient, 100%; per adenoma, 88.9%).
CONCLUSION
SPECT is superior to planar imaging. SPECT-CT has identical sensitivity compared to SPECT alone, but it provides additional topographic information. The sensitivity of PET appears to be even higher compared to SPECT. In the case of negative scintigraphic findings and proven hyperparathyroidism, additional C-11 methionine PET or PET-CT is recommended.
Topics: Adenoma; Adult; Aged; Aged, 80 and over; False Negative Reactions; Female; Humans; Hyperplasia; Male; Methionine; Middle Aged; Multimodal Imaging; Parathyroid Glands; Parathyroid Neoplasms; Positron-Emission Tomography; Radiopharmaceuticals; Retrospective Studies; Tomography, Emission-Computed, Single-Photon; Tomography, X-Ray Computed; Young Adult
PubMed: 21305468
DOI: 10.4261/1305-3825.DIR.3486-10.1 -
Langenbeck's Archives of Surgery Feb 2018Primary hyperparathyroidism (pHPT) is caused by single- or multiglandular disease (MGD). Patients with MGD have an increased risk of complications at surgery and for...
BACKGROUND
Primary hyperparathyroidism (pHPT) is caused by single- or multiglandular disease (MGD). Patients with MGD have an increased risk of complications at surgery and for persistence and recurrence after surgery. The study evaluated whether preoperative clinical and biochemical characteristics could predict MGD in patients with pHPT.
METHODS
We retrospectively evaluated patients operated 1989-2013 for first-time, non-hereditary pHPT. MGD was defined in patients with more than one pathological gland excised at surgery or with persistent hypercalcemia after the excision of a single pathological parathyroid gland, confirmed by histopathology. Clinical and biochemical variables were compared in patients with single- and multiglandular disease. Logistic regression was used to identify variables predicting MGD, yielding odds ratios (OR) with 95% confidence intervals (CI).
RESULTS
There were 707 patients, of which 79 (11%) had MGD. Patients with MGD were more likely to have negative sestamibi scintigraphy than patients with single-gland disease, 15 of 49 (31%) vs. 70 of 402 (17%; p = 0.03), to suffer from diabetes (12 of 74, 16%) vs. 45 out of 626 patients (7.2%; p < 0.01) and had lower preoperative levels of urinary calcium (3.80 vs. 4.44 mmol/L; p = 0.04). Multivariable analysis identified negative scintigraphy (OR 2.42; 95% CI 1.18 to 4.79), diabetes (OR 2.75; 95% CI 1.31 to 4.97) and elevated levels of osteocalcin (OR 3.79, 95% CI: 1.75 to 8.21) as predictors of MGD.
CONCLUSION
Negative sestamibi scintigraphy, diabetes and elevated osteocalcin levels were predictors of MGD.
Topics: Aged; Blood Glucose; Female; Humans; Hyperparathyroidism, Primary; Logistic Models; Male; Middle Aged; Osteocalcin; Parathyroid Glands; Parathyroid Hormone; Parathyroidectomy; Predictive Value of Tests; Radionuclide Imaging; Retrospective Studies
PubMed: 29294178
DOI: 10.1007/s00423-017-1647-9 -
Pediatric Nephrology (Berlin, Germany) Apr 2013In order to minimize complications on the skeleton and to prevent extraskeletal calcifications, the specific aims of the management of chronic kidney disease mineral and... (Review)
Review
In order to minimize complications on the skeleton and to prevent extraskeletal calcifications, the specific aims of the management of chronic kidney disease mineral and bone disorder (CKD-MBD) are to maintain blood levels of serum calcium and phosphorus as close to the normal range as possible, thereby maintaining serum parathyroid hormone (PTH) at levels appropriate for CKD stage, preventing hyperplasia of the parathyroid glands, avoiding the development of extra-skeletal calcifications, and preventing or reversing the accumulation of toxic substances such as aluminum and β2-microglobulin. In order to limit cardiovascular calcification, daily intake of elemental calcium, including from dietary sources and from phosphate binders, should not exceed twice the daily recommended intake for age and should not exceed 2.5 g/day. Calcium-free phosphate binders, such as sevelamer hydrochloride and sevelamer carbonate, are safe and effective alternatives to calcium-based binders, and their use widens the margin of safety for active vitamin D sterol therapy. Vitamin D deficiency is highly prevalent across the spectrum of CKD, and replacement therapy is recommended in vitamin D-deficient and insufficient individuals. Therapy with active vitamin D sterols is recommended after correction of the vitamin D deficiency state and should be titrated based on target PTH levels across the spectrum of CKD. Although the use of calcimimetic drugs has been proven to effectively control the biochemical features of secondary hyperparathyroidism, there is very limited experience with the use of such agents in pediatric patients and especially during the first years of life. Studies are needed to further define the role of such agents in the treatment of pediatric CKD-MBD.
Topics: Biomarkers; Bone Diseases, Metabolic; Calcimimetic Agents; Calcium; Chelating Agents; Child; Humans; Phosphates; Renal Insufficiency, Chronic; Treatment Outcome; Vitamin D; Vitamin D Deficiency; Vitamins
PubMed: 23381010
DOI: 10.1007/s00467-012-2381-8 -
Chirurgia (Bucharest, Romania : 1990) 2019The secondary hyperparathyroidism (sHPT) affects all patients with chronic renal failure in different degrees. The chronic kidney disease is often associated with...
The secondary hyperparathyroidism (sHPT) affects all patients with chronic renal failure in different degrees. The chronic kidney disease is often associated with multiple severe comorbidities, therefore the figures for mortality are higher than in the general population. The failure of medical treatment is an indication for surgical treatment. The recurrence of the disease in secondary hyperpara-thyroidism after surgical treatment using total parathyroid with autotransplantation or subtotal parathyroidectomy may be a challenge due to hyperplasia of the remaining tissue. The purpose of this retrospective study was to highlight the risk factors for the occurrence of glandular hyperplasia in patients with secondary hyperparathyroidism and to determine optimal surgery approach for secondary hyperparathyroidism in order to minimize relapse rates. Parathyroid size evaluation may suggest the presence of nodular hyperplasia contributing to an early parathyroidectomy and at the same time selecting the best surgical treatment for sHPT patients. As resistance to medical therapy is due to the presence of nodular hyperplasia, some authors recomends subtotal parathyroidectomy with the excision of these glands, with the remaining parathyroids tissue and function controlled by medical therapy (20).
Topics: Humans; Hyperparathyroidism, Secondary; Hyperplasia; Kidney Failure, Chronic; Parathyroid Glands; Parathyroidectomy; Retrospective Studies; Risk Factors
PubMed: 31670635
DOI: 10.21614/chirurgia.114.5.594 -
JCEM Case Reports Jan 2024Parathyroid adenoma (PA) and parathyroid hyperplasia (PH) are common causes of primary hyperparathyroidism (PHPT), for which the only definitive treatment is surgery....
Parathyroid adenoma (PA) and parathyroid hyperplasia (PH) are common causes of primary hyperparathyroidism (PHPT), for which the only definitive treatment is surgery. Abnormalities in the parathyroid glands can be identified with various imaging modalities including ultrasound (US), sestamibi scan (MIBI), 4-dimensional computed tomography (4D-CT), and positron emission tomography/computed tomography (PET/CT). While it is not uncommon for parathyroid pathology to be undetected on imaging, this is more typical of low-volume hyperplasia and smaller-sized adenomas. We present the case of a 65-year-old man with PHPT who initially had a solitary parathyroid mass detected by US, but who was ultimately discovered to have massive PH with hyperplastic glands not visualized on US or MIBI. This atypical presentation may help guide providers in decisions on ordering and interpreting various imaging modalities for patients with PHPT. In this case, 4D-CT was the only modality in which large hyperplastic glands were identified, suggesting superior sensitivity. This case also highlights the importance of intraoperative parathyroid hormone testing to aid in diagnostic prediction.
PubMed: 38188905
DOI: 10.1210/jcemcr/luad173 -
BMC Endocrine Disorders Jan 2021Familial hypocalciuric hypercalcemia (FHH) is a heterogeneous autosomal-dominant disorder of calcium hemostasis that may be difficult to distinguish clinically from mild...
BACKGROUND
Familial hypocalciuric hypercalcemia (FHH) is a heterogeneous autosomal-dominant disorder of calcium hemostasis that may be difficult to distinguish clinically from mild primary hyperparathyroidism. Loss-of-function mutations mainly involving Arg15 residue of the adaptor-related protein complex 2, sigma subunit 1 (AP2S1) cause a rarer, more recently recognized form of FHH, FFH type-3. Recently, F-fluorocholine positron emission tomography/computed tomography (FCH-PET/CT) showed superior sensitivity to conventional imaging in localizing parathyroid adenomas. We report a new FFH type-3 patient who underwent unnecessary parathyroidectomy in association with misleading FCH-PET/CT imaging.
CASE PRESENTATION
A 29-year old woman was initially evaluated for parathyroid hormone (PTH)-dependent hypercalcemia in 2013. Medical history was positive only for chronic constipation and malaise with no personal or family history of hypercalcemia, kidney stones, or neck surgery. Over seven years, serum calcium level was 2.51-2.89 mmol/L with concomitant PTH level of 58.7-94.8 mmol/L. Serum phosphate levels were in the low/low normal range. Serum creatinine and magnesium levels were normal. 25-hydroxy vitamin D level was 13 nmol/L. 24-hour urine calcium level was 1.92 mmol/day but increased to 6.99 mmol/day after treatment with cholecalciferol 1000 IU daily. Bone mineral density and renal ultrasound were normal. Parathyroid ultrasound showed two hypoechoic nodules inferior to the left and right thyroid lobes; however, technitium-sestamibi scans (2013, 2016, 2018) were negative. FCH-PET/CT (2019) showed focal uptake co-localizing with the nodule inferior to the left thyroid lobe. The patient underwent left inferior parathyroidectomy and pathology was consistent with parathyroid hyperplasia. However, postoperatively, serum calcium and PTH levels remained elevated and FCH-PET/CT and ultrasound showed persistence of the uptake/nodule. Whole exome sequencing showed Arg15Cys mutation in the AP2S1 gene characteristic of FHH type-3.
CONCLUSIONS
In this new case of FHH type-3, FCH-PET/CT failed to localize to the hyperplastic parathyroid glands and localized instead to apparently a lymph node. This, together with increased urinary calcium after vitamin D supplementation, led to unnecessary parathyroidectomy. Given the increasingly lower cost of genetic testing and the cost of follow up and unnecessary surgery, it may prudent to include genetic testing for FHH early on in patients with mild PTH-dependent hypercalcemia.
Topics: Adaptor Protein Complex 2; Adaptor Protein Complex sigma Subunits; Adult; Bone Density; Calcium; Choline; Female; Humans; Hypercalcemia; Hyperparathyroidism, Primary; Kidney; Parathyroid Glands; Parathyroid Hormone; Parathyroidectomy; Positron Emission Tomography Computed Tomography; Radiopharmaceuticals; Treatment Outcome
PubMed: 33499837
DOI: 10.1186/s12902-021-00683-z -
Problemy Endokrinologii Nov 2021Multiple lesions of the parathyroid glands (PTG) in primary hyperparathyroidism (PHPT) can be sporadic or develop as part of hereditary syndromes, manifesting at young...
Multiple lesions of the parathyroid glands (PTG) in primary hyperparathyroidism (PHPT) can be sporadic or develop as part of hereditary syndromes, manifesting at young age. There the description of a severe sporadic PHPT with big parathyroid neoplasms in the young patient is presented. Clinical data made it possible to suspect MEN-1 syndrome or parathyroid carcinomas; however, mutations CDKN, CDC73, MEN1 were excluded. The patient underwent removal of three identified tumors: benign adenomas of the left PTG and hyperplasia of the right one. Postoperative hypocalcemia and severe hungry bone syndrome required the administration of vitamin D and calcium carbonate preparations. However, a year after the operation, a «mild» recurrent disease was confirmed. Taking into account the patient's refusal to reoperation and a significant improvement of the target organs state, active observation was continued. The patient needs further careful dynamic monitoring by specialists in order to timely identify indications for repeated surgical treatment to improve the life quality and span.
Topics: Adenoma; Humans; Hyperparathyroidism, Primary; Multiple Endocrine Neoplasia Type 1; Parathyroid Glands; Parathyroid Neoplasms
PubMed: 35018759
DOI: 10.14341/probl12798