Spontaneous remission of primary hyperparathyroidism from parathyroid apoplexy.

Nylen, Eric; Shah, A N; Hall, James L.

doi:10.1210/jcem.81.4.8636326

Cited by 26 publications

(21 citation statements)

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“…Spontaneous cases of parathyroid apoplexy, were firstly described in 1946 (7) and defined pathologically by the presence of hemorrhage or infarction within the adenoma (8). Spontaneous parathyroid apoplexy occurs rarely in primary hyperparathyroidism and features heterogeneous manifestations such as cervical pain, neck mass and compression symptoms, hematoma or ecchymosis, hypercalcemic crisis due to acute PTH release, and hypocalcemia with tetany or convulsions due to auto-parathyroidectomy (8). Asymptomatic cases and potentially life-threatening manifestations are also described (9).…”

Section: Discussionmentioning

confidence: 99%

Parathyroid Apoplexy Following Cinacalcet Treatment in Primary Hyperparathyroidism

Dalmazi¹,

Giuliani²,

Napolitano³

2018

Front. Endocrinol.

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Cinacalcet, a calcimimetic drug, is considered a safe and valid option for the treatment of hypercalcemia in patients with primary hyperparathyroidism who are unable to undergo parathyroidectomy. Hypocalcemia and gastrointestinal adverse reactions are the main side effects reported in patients treated with cinacalcet. We present here the case of an 80-years-old patient with primary hyperparathyroidism treated with cinacalcet for 17 months who developed a severe and symptomatic episode of hypocalcemia requiring hospitalization 1 month after reaching a daily dose of 180 mg. Follow-up laboratory and imaging exams showed remission of primary hyperparathyroidism and disappearance of the parathyroid adenoma, suggesting a possible association between cinacalcet therapy and parathyroid infarction resulting in normalization of the elevated serum parathyroid hormone levels and severe hypocalcemia. No known cases of iatrogenic parathyroid apoplexy have thus far been described. We report here the first case of parathyroid apoplexy associated with the administration of cinacalcet in a patient with primary hyperparathyroidism. Parathyroid apoplexy features heterogeneous clinical manifestations ranging from relatively asymptomatic to potentially life-threatening cases. The occurrence of this complication should be carefully considered in patients with primary hyperparathyroidism in therapy with cinacalcet.

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Section: Discussionmentioning

confidence: 99%

Parathyroid Apoplexy Following Cinacalcet Treatment in Primary Hyperparathyroidism

Dalmazi¹,

Giuliani²,

Napolitano³

2018

Front. Endocrinol.

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“…Nylen and coworkers suggested that the pathogenesis of ‘autoparathyroidectomy’ can be divided into three broad etiologies: (i) autoinfarction (necrosis without hemorrhage), (ii) acute extracapsular hemorrhage and (iii) acute intracapsular hemorrhage (10). However, the etiology of ‘autoparathyroidectomy’ remains still not clear, especially in clinical cases lacking histological verification.…”

Section: Discussionmentioning

confidence: 99%

“…However, the etiology of ‘autoparathyroidectomy’ remains still not clear, especially in clinical cases lacking histological verification. Besides, the time of recurrence depends on the degree of cellular damage (10). In the present case, modest local symptoms and the lack of evidence of the previously documented parathyroid lesion, along with the prolonged uneventful biochemical follow-up, strongly suggest a US-FNAB-induced intracapsular hemorrhage rather than an infarction.…”

Section: Discussionmentioning

confidence: 99%

Cured primary hyperparathyroidism after fine-needle aspiration biopsy-induced parathyroid disappearance

Kara

Valle

Vincentis

et al. 2017

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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Spontaneous or fine-needle aspiration (FNAB)-induced remission of primary hyperparathyroidism (PHPT) may occur, especially for cystic lesions. However, the disease generally relapses over a short time period. We present a case of PHPT due to an enlarged hyperfunctioning parathyroid that underwent long-term (almost 9 years) clinical and ultrasonographic remission after the disappearance of the lesion following ultrasound (US)-assisted FNAB. A 67-year-old woman with PHPT underwent biochemical and US examinations that confirmed the diagnosis and showed a lesion suggestive for parathyroid adenoma or hyperplasia. US-FNAB of the lesion confirmed its parathyroid nature by means of elevated levels of parathyroid hormone within the needle washing fluid. At the second visit, the patient referred slight neck swelling that resolved spontaneously in the days after the US-FNAB. At subsequent follow-up, the enlarged parathyroid was not found; it was visible neither with US nor with magnetic resonance imaging. Biochemical remission persists after 9 years. This is the first reported case of cure of PHPT after US-FNAB performed on a hyperfunctioning parathyroid resulting in its complete disappearance over a period of 9 years of negative biochemical and ultrasonographic follow-up.Learning points:Spontaneous or fine-needle aspiration-induced remission of primary hyperparathyroidism can occur.Both circumstances may present disease relapse over a variable time period, but definite remission is also possible even though long-term periodic follow-up should be performed.Parathyroid damage should be ruled out in case of neck symptomatology after parathyroid fine-needle aspiration or spontaneous symptomatology in patients with history of primary hyperparathyroidism.

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“…Parathyroidectomy has been considered the only definitive cure, however, watchful waiting with monitoring for the presence of end-organ damage is also warranted in selected cases (4). Over last decades, several cases of spontaneous remission of PHPT have been reported and attributed to spontaneous necrosis, haemorrhage and infarction of parathyroid adenoma and this phenomenon has been referred to as ‘parathyroid auto-infarction’, ‘auto-parathyroidectomy’ or ‘parathyroid apoplexy’ (5, 6, 7, 8, 9).…”

Section: Introductionmentioning

confidence: 99%

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

Novodvorský

Hussein

Arshad

et al. 2019

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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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.

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Spontaneous remission of primary hyperparathyroidism from parathyroid apoplexy.

Cited by 26 publications

References 0 publications

Parathyroid Apoplexy Following Cinacalcet Treatment in Primary Hyperparathyroidism

Parathyroid Apoplexy Following Cinacalcet Treatment in Primary Hyperparathyroidism

Cured primary hyperparathyroidism after fine-needle aspiration biopsy-induced parathyroid disappearance

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

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