scholarly journals Two cases of spontaneous remission of primary hyperparathyroidism due to auto-infarction: different management and their outcomes

2019 ◽  
Vol 2019 ◽  
Author(s):  
Peter Novodvorsky ◽  
Ziad Hussein ◽  
Muhammad Fahad Arshad ◽  
Ahmed Iqbal ◽  
Malee Fernando ◽  
...  
Keyword(s):  
Parathyroid Hormone ◽  
Primary Hyperparathyroidism ◽  
Parathyroid Adenoma ◽  
Spontaneous Remission ◽  
Calcium Excretion ◽  
List Type ◽  
Tertiary Centre ◽  
Parathyroid Adenomas ◽  
Severe Hypercalcaemia ◽  
Parathyroid Lesion

Summary Spontaneous remission of primary hyperparathyroidism (PHPT) due to necrosis and haemorrhage of parathyroid adenoma, the so-called ‘parathyroid auto-infarction’ is a very rare, but previously described phenomenon. Patients usually undergo parathyroidectomy or remain under close clinical and biochemical surveillance. We report two cases of parathyroid auto-infarction diagnosed in the same tertiary centre; one managed surgically and the other conservatively up to the present time. Case #1 was a 51-year old man with PHPT (adjusted (adj.) calcium: 3.11 mmol/L (reference range (RR): 2.20–2.60 mmol/L), parathyroid hormone (PTH) 26.9 pmol/L (RR: 1.6–6.9 pmol/L) and urine calcium excretion consistent with PHPT) referred for parathyroidectomy. Repeat biochemistry 4 weeks later at the surgical clinic showed normal adj. calcium (2.43 mmol/L) and reduced PTH. Serial ultrasound imaging demonstrated reduction in size of the parathyroid lesion from 33 to 17 mm. Twenty months later, following recurrence of hypercalcaemia, he underwent neck exploration and resection of an enlarged right inferior parathyroid gland. Histology revealed increased fibrosis and haemosiderin deposits in the parathyroid lesion in keeping with auto-infarction. Case #2 was a 54-year-old lady admitted with severe hypercalcaemia (adj. calcium: 4.58 mmol/L, PTH 51.6 pmol/L (RR: 1.6–6.9 pmol/L)) and severe vitamin D deficiency. She was treated with intravenous fluids and pamidronate and 8 days later developed symptomatic hypocalcaemia (1.88 mmol/L) with dramatic decrease of PTH (17.6 pmol/L). MRI of the neck showed a 44 mm large cystic parathyroid lesion. To date, (18 months later), she has remained normocalcaemic. Learning points: Primary hyperparathyroidism (PHPT) is characterised by excess parathyroid hormone (PTH) secretion arising mostly from one or more autonomously functioning parathyroid adenomas (up to 85%), diffuse parathyroid hyperplasia (<15%) and in 1–2% of cases from parathyroid carcinoma. PHPT and hypercalcaemia of malignancy, account for the majority of clinical presentations of hypercalcaemia. Spontaneous remission of PHPT due to necrosis, haemorrhage and infarction of parathyroid adenoma, the so-called ‘parathyroid auto-infarction’, ‘auto-parathyroidectomy’ or ‘parathyroid apoplexy’ is a very rare in clinical practice but has been previously reported in the literature. In most cases, patients with parathyroid auto-infarction undergo parathyroidectomy. Those who are managed conservatively need to remain under close clinical and biochemical surveillance long-term as in most cases PHPT recurs, sometimes several years after auto-infarction.

scholarly journals A novel CASR variant in a family with familial hypocalciuric hypercalcaemia and primary hyperparathyroidism

2020 ◽  
Vol 2020 ◽  
Author(s):  
Satyanarayana V Sagi ◽  
Hareesh Joshi ◽  
Jamie Trotman ◽  
Terence Elsey ◽  
Ashwini Swamy ◽  
...  
Keyword(s):  
Parathyroid Hormone ◽  
Primary Hyperparathyroidism ◽  
Organ Damage ◽  
Urinary Calcium ◽  
Interesting Case ◽  
List Type ◽  
Loss Of Function ◽  
Clearance Ratio ◽  
Hormone Levels ◽  
Severe Hypercalcaemia

Summary Familial hypocalciuric hypercalcaemia (FHH) is a dominantly inherited, lifelong benign disorder characterised by asymptomatic hypercalcaemia, relative hypocalciuria and variable parathyroid hormone levels. It is caused by loss-of-function pathogenic variants in the calcium-sensing receptor (CASR) gene. Primary hyperparathyroidism (PHPT) is characterised by variable hypercalcaemia in the context of non-suppressed parathyroid hormone levels. Unlike patients with FHH, patients with severe hypercalcaemia due to PHPT are usually symptomatic and are at risk of end-organ damage affecting the kidneys, bone, heart, gastrointestinal system and CNS. Surgical resection of the offending parathyroid gland(s) is the treatment of choice for PHPT, while dietary adjustment and reassurance is the mainstay of management for patients with FHH. The occurrence of both FHH and primary hyperparathyroidism (PHPT) in the same patient has been described. We report an interesting case of FHH due to a novel CASR variant confirmed in a mother and her two daughters and the possible coexistence of FHH and PHPT in the mother, highlighting the challenges involved in diagnosis and management. Learning points: Familial hypocalciuric hypercalcaemia (FHH) and primary hyperparathyroidism (PHPT) can coexist in the same patient. Urinary calcium creatinine clearance ratio can play a role in distinguishing between PHPT and FHH. Genetic testing should be considered in managing patients with PHPT and FHH where the benefit may extend to the wider family. Family segregation studies can play an important role in the reclassification of variants of uncertain significance. Parathyroidectomy has no benefit in patients with FHH and therefore, it is important to exclude FHH prior to considering surgery. For patients with coexisting FHH and PHPT, parathyroidectomy will reduce the risk of complications from the severe hypercalcaemia associated with PHPT.

scholarly journals Preoperative localization of suspicious parathyroid adenomas by assay of parathyroid hormone in needle aspirates

1998 ◽  
pp. 72-77 ◽  
Author(s):  
C Marcocci ◽  
S Mazzeo ◽  
G Bruno-Bossio ◽  
A Picone ◽  
E Vignali ◽  
...  
Keyword(s):  
Parathyroid Hormone ◽  
Primary Hyperparathyroidism ◽  
Parathyroid Adenoma ◽  
Nodular Goiter ◽  
Neck Mass ◽  
Parathyroid Scintigraphy ◽  
Simple Method ◽  
Cervical Mass ◽  
Parathyroid Adenomas ◽  
Group 2

OBJECTIVE: To determine the usefulness of parathyroid hormone (PTH) measurement in needle aspirates of a suspicious neck mass to confirm its parathyroid nature in patients with primary hyperparathyroidism. METHODS: Thirty-three patients with surgically proved primary hyperparathyroidism were submitted to neck ultrasound (US), parathyroid scintigraphy, and assay of PTH in the aspirate (PTHa) of the suspicious cervical mass. RESULTS: Based on the results of neck US and parathyroid scintigraphy, patients were divided into two groups. Group 1: 16 patients (seven with nodular goiter) with concordant positive US and scintigraphic results. In all but one patient, PTHa was detectable and often markedly elevated (> 1000 pg in 12 patients, between 292 pg and 803 pg in three patients and 53 pg in one patient). The patient with undetectable PTHa had a small lower left parathyroid adenoma (8x8x10 mm). Group 2: 17 patients (12 with nodular goiter) with discordant US and scintigraphic results. PTHa established the parathyroid nature of the mass in 13 cases (> 1000 pg in 8 patients, between 501 pg and 953 pg in three patients and 90 and 79 pg in two patients): 11 of these had a suspected lesion by US examination but the scintigraphy results were negative; two had a mass that gave positive scintigraphy results but was of uncertain origin according to US: in both cases an intrathyroidal parathyroid adenoma was found. PTHa was undetectable in four cases (three with nodular goiter): all of these had equivocal US results, and three had positive scans and one a negative scan. CONCLUSIONS: Assay of PTHa is a simple method and should be useful for confirming the parathyroid nature of a cervical mass in patients with discordant or non-diagnostic US and scintigraphic results.

scholarly journals Autofluorescence pattern of parathyroid adenomas

BJS Open ◽  
2021 ◽  
Vol 5 (1) ◽  
Author(s):  
M S Demarchi ◽  
W Karenovics ◽  
B Bédat ◽  
C De Vito ◽  
F Triponez
Keyword(s):  
Parathyroid Hormone ◽  
Surgical Treatment ◽  
Primary Hyperparathyroidism ◽  
Parathyroid Adenoma ◽  
Sensitivity And Specificity ◽  
Near Infrared ◽  
Parathyroid Tissue ◽  
Endocrine Surgery ◽  
Parathyroid Adenomas ◽  
Tissue Sensitivity

Abstract Background Primary hyperparathyroidism (pHPT) is a common endocrine pathology, and it is due to a single parathyroid adenoma in 80–85 per cent of patients. Near-infrared autofluorescence (NIRAF) has recently been used in endocrine surgery to help in the identification of parathyroid tissue, although there is currently no consensus on whether this technique can differentiate between normal and abnormal parathyroid glands. The aim of this study was to describe the autofluorescence pattern of parathyroid adenoma in pHPT. Methods Between January and June 2019, patients with pHPT who underwent surgical treatment for parathyroid adenoma were enrolled. Parathyroid autofluorescence was measured. Results Twenty-three patients with histologically confirmed parathyroid adenomas were included. Parathyroid adenomas showed a heterogeneous fluorescence pattern, and a well defined autofluorescent ‘cap’ region was observed in 17 of 23 specimens. This region was on average 28 per cent more fluorescent than the rest of the adenoma, and corresponded to a rim of normal histological parathyroid tissue (sensitivity and specificity 88 and 67 per cent respectively). After resection, all patients were treated successfully, with normal postoperative values of calcium and parathyroid hormone documented. Conclusion Parathyroid adenomas show a heterogeneous autofluorescence pattern. Using NIRAF imaging, the majority of specimens showed a well defined autofluorescent portion corresponding to a rim of normal parathyroid tissue. Further studies should be conducted to validate these findings.

PARATHYROID HORMONE SECRETION IN DISORDERS OF CALCIUM METABOLISM STUDIED BY MEANS OF EDTA

1974 ◽  
Vol 75 (2) ◽  
pp. 286-296 ◽  
Author(s):  
J. H. Lockefeer ◽  
W. H. L. Hackeng ◽  
J. C. Birkenhäger
Keyword(s):  
Parathyroid Hormone ◽  
Primary Hyperparathyroidism ◽  
Calcium Metabolism ◽  
Hormone Secretion ◽  
Parathyroid Tissue ◽  
Small Mass ◽  
Idiopathic Hypercalciuria ◽  
List Type ◽  
Immunoreactive Parathyroid Hormone ◽  
Pth Level

ABSTRACT In 22 of 28 cases of primary hyperparathyroidism (PHP) the rise in the serum immunoreactive parathyroid hormone (IRPTH or PTH) level observed in response to lowering of the serum calcium by EDTA, exceeded that obtained in 8 control subjects. In 5 of these 22 patients who were studied again after parathyroidectomy the supranormal response was abolished. Fifteen of these 22 hyper-responsive PHP patients had basal IRPTH levels not exceeding the highest level in the controls and that of other groups of patients investigated (idiopathic hypercalciuria, non-parathyroid hypercalcaemia, operated PHP). Fourteen of the 22 hyper-reactive patients with PHP did not show hypocalcaemia during the infusion of EDTA. The extent of the release of PTH elicited by EDTA in cases of PHP does not as yet allow a prediction of the amount of pathological parathyroid tissue present, although all the PHP patients showing a normal release of PTH had a relatively small mass of parathyroid tissue (up to about 1 g) subsequently removed. In 9 cases of nephrolithiasis (8 of whom had idiopathic hypercalciuria) and in 7 cases of non-parathyroid hypercalcaemia, a normal PTH release was found.

scholarly journals Giant parathyroid adenoma: a case report and review of the literature

2019 ◽  
Vol 13 (1) ◽  
Author(s):  
Mohamed S. Al-Hassan ◽  
Menatalla Mekhaimar ◽  
Walid El Ansari ◽  
Adham Darweesh ◽  
Abdelrahman Abdelaal
Keyword(s):  
Parathyroid Hormone ◽  
Primary Hyperparathyroidism ◽  
Parathyroid Adenoma ◽  
Neck Mass ◽  
Rare Type ◽  
Tissue Mass ◽  
Laboratory Findings ◽  
Clinical Presentations ◽  
High Calcium

Abstract Background Giant parathyroid adenoma is a rare type of parathyroid adenoma defined as weighing > 3.5 g. They present as primary hyperparathyroidism but with more elevated laboratory findings and more severe clinical presentations due to the larger tissue mass. This is the first reported case of giant parathyroid adenoma from the Middle East. Case presentation A 52-year-old Indian woman presented with a palpable right-sided neck mass and generalized fatigue. Investigations revealed hypercalcemia with elevated parathyroid hormone and an asymptomatic kidney stone. Ultrasound showed a complex nodule with solid and cystic components, and Sestamibi nuclear scan confirmed a giant parathyroid adenoma. Focused surgical neck exploration was done and a giant parathyroid adenoma weighing 7.7 gm was excised. Conclusions Giant parathyroid adenoma is a rare cause of primary hyperparathyroidism and usually presents symptomatically with high calcium and parathyroid hormone levels. Giant parathyroid adenoma is diagnosed by imaging and laboratory studies. Management is typically surgical, aiming at complete resection. Patients usually recover with no long-term complications or recurrence.

scholarly journals Nontraumatic Parapharyngeal Haematoma: A Rare Lesion

2018 ◽  
Vol 2018 ◽  
pp. 1-2 ◽  
Author(s):  
Pedro Carneiro de Sousa ◽  
Inês Gambôa ◽  
Delfim Duarte ◽  
Nuno Trigueiros-Cunha
Keyword(s):  
Case Report ◽  
Parathyroid Hormone ◽  
Conservative Treatment ◽  
Parathyroid Adenoma ◽  
Parapharyngeal Space ◽  
Neck Swelling ◽  
Parathyroid Adenomas ◽  
Rare Lesion ◽  
Symptoms And Signs ◽  
Emergent Surgery

Nontraumatic haematoma of parapharyngeal space is very rare and may cause dysphagia and dyspnea. The authors present a case report of a 74-year-old woman with sudden nontraumatic neck swelling without dyspnea and with left pharyngeal bulging and endolaryngeal displacement. Parathyroid hormone elevation and imaging exams confirmed bleeding from a parathyroid adenoma. Symptoms and signs resolved after one week of conservative treatment. There are few cases of parapharyngeal haematomas caused by parathyroid adenomas. Most patients can be managed without emergent surgery, but close airway monitoring is fundamental.

Recurrence of primary hyperparathyroidism following spontaneous remission with intracapsular hemorrhage of a parathyroid adenoma

2008 ◽  
Vol 26 (3) ◽  
pp. 295-297 ◽  
Author(s):  
Keisuke Kataoka ◽  
Manabu Taguchi ◽  
Akira Takeshita ◽  
Megumi Miyakawa ◽  
Yasuhiro Takeuchi
Keyword(s):  
Primary Hyperparathyroidism ◽  
Parathyroid Adenoma ◽  
Spontaneous Remission

scholarly journals Spontaneous Remission of Primary Hyperparathyroidism due to Hemorrhagic Infarction in the Parathyroid Adenoma.

1996 ◽  
Vol 35 (8) ◽  
pp. 646-649 ◽  
Author(s):  
Koshi NATSUI ◽  
Kiyoshi TANAKA ◽  
Michio SUDA ◽  
Akihiro YASODA ◽  
Shin YONEMITSU ◽  
...  
Keyword(s):  
Primary Hyperparathyroidism ◽  
Parathyroid Adenoma ◽  
Spontaneous Remission ◽  
Hemorrhagic Infarction

scholarly journals Rare Case of Severe Hypercalcemia Secondary to Atypical Sestamibi Negative Parathyroid Cystic Adenoma

2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A266-A267
Author(s):  
Timur Gusov ◽  
John Chen Liu ◽  
Sowjanya Naha ◽  
F N U Marium ◽  
Joseph Theressa Nehu Parimi ◽  
...  
Keyword(s):  
Primary Hyperparathyroidism ◽  
Parathyroid Adenoma ◽  
Parathyroid Gland ◽  
Severe Hypercalcemia ◽  
Parathyroid Adenomas ◽  
Sestamibi Scan ◽  
Serum Pth ◽  
The Right ◽  
99Mtc Sestamibi ◽  
Cystic Adenoma

Abstract Primary hyperparathyroidism (PHPT) is defined as excessive secretion of parathyroid hormone (PTH) originating from the parathyroid gland. The most common cause is a single parathyroid adenoma which is typically solid. Cystic parathyroid adenomas (CPA) are the cause of about 1–2% of cases of primary hyperparathyroidism. It is known that cystic parathyroid adenomas are a result of degeneration of an existing parathyroid adenoma. SestaMIBI is an imaging study based on uptake of radioactive technetium99 and used to localize parathyroid adenomas. We describe an unusual case of severe hypercalcemia secondary to 99mTc sestaMIBI negative atypical parathyroid cystic adenoma. A 56-year-old male presented to our facility with nausea and vomiting. His past medical history included hypertension and hepatitis C with no history of fractures or kidney disease. Physical examination was normal. Upon admission the patient was afebrile with blood pressure of 170/120 mmHg and heart rate of 62 bpm. Chemistry showed Calcium of 14.5 mg/dL (8.6–10.2mg/dL), phosphorus 2.2 (2.7–4.5) mh/dL, magnesium 1.8 (1.7–2.6)mg/dL, intact PTH of 375 (15–65) pg/mL, PTH-related peptide &lt;2.0 pmol/L(&lt;2 pmol/L), 25-OH vitamin D of 19 ng/ml (30–80), Creatinine 1.22 (0.7–1.2)mg/dL, alkaline phosphatase 95 (40–129) units/L. He was started on aggressive hydration, calcitonin 4 units/kg, 4 mg of IV Zolendroninc acid. Neck sonogram revealed a large, complex, predominantly anechoic lesion with solid vascular components and thick internal septations in the inferior and medial aspect of the right thyroid lobe measuring 3 x 2 x 5.5 cm. Findings were confirmed with CT of the neck. Since Sestamibi scan (planar and SPECT/CT) did not show uptake in parathyroid glands, the cyst was thought to be of thyroid origin. Fine needle aspiration was not able to detect cellular material, but PTH was &gt;100 pg/ml on the FNA sample. Otolaryngology service was consulted for parathyroidectomy. During the surgical treatment, the right upper parathyroid gland was removed with no changes in serum PTH. Next, the cystic lesion was removed with normalization of serum PTH (from 218 pg/ml to 35.2 pg/ml respectively). Intraoperative frozen section analysis was read as a cystic parathyroid adenoma. The final pathology report revealed cystic parathyroid tissue favoring parathyroid adenoma with focal atypia. Hypercalcemia resolved. Conclusions: Atypical cystic parathyroid adenomas are a rare cause of PHPT. 90% of parathyroid cysts are nonfunctional. Above mention is a case of a patient presenting with hypercalcemic crisis secondary to cystic parathyroid adenoma, which posed a diagnostic challenge as both neck ultrasound and 99mTc sestaMIBI scan were inconclusive. These findings should trigger suspicion for functional parathyroid lesions. Cystic components should be evaluated for PTH levels and if significantly elevated should be treated as a parathyroid adenoma.

scholarly journals Primary Hyperparathyroidism: Modern Conception and Clinical Observation

2020 ◽  
Vol 10 (2) ◽  
pp. 94-101
Author(s):  
L. M. Farkhutdinova
Keyword(s):  
Parathyroid Hormone ◽  
Primary Hyperparathyroidism ◽  
Parathyroid Adenoma ◽  
Clinical Manifestations ◽  
Premature Death ◽  
Developed Countries ◽  
Severe Form ◽  
Mineral Density ◽  
Target Organs ◽  
Increased Risk

The article is devoted to one of the actual medical and social problems — primary hyperparathyroidism, the late diagnosis of which leads to the development of severe complications and an increased risk of premature death. Unlike developed countries, where 80% of cases are represented by mild forms of the disease, in the Russian Federation this indicator does not exceed 30%, while 70% are manifest forms. Widespread awareness of doctors of various specialties in the diagnosis of parathyroid adenoma is necessary for the timely detection of the disease. The article reflects the main stages of the study of the disease, the pathogenesis of the clinical manifestations of primary hyperparathyroidism, the classic symptoms of which are changes in the target organs of the parathyroid hormone — bone tissue, urinary system and gastrointestinal tract, is considered. Bone disorders are the most common manifestation of hyperparathyroidism and are characterized by increased bone metabolism with a progressive decrease in bone mineral density. Typical changes in the kidneys include nephrolithiasis and nephrocalcinosis, causing the formation of renal failure. Gastrointestinal signs of hyperparathyroidism are erosion and ulcers of the stomach and duodenum, prone to bleeding, recurrent pancreatitis. Diagnosis of the disease is based on laboratory results, characterized by elevated levels of calcium and parathyroid hormone in the blood. Visualization of the paradenomas in most cases is provided by ultrasound and scintigraphy. Removal of parathyroid adenoma is the most effective treatment. A clinical case of a severe form of the disease is presented, indicating an urgent need to take measures to solve the problem of primary hyperparathyroidism.

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