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

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 biochemic...

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Autores principales: Novodvorsky, Peter, Hussein, Ziad, Arshad, Muhammad Fahad, Iqbal, Ahmed, Fernando, Malee, Munir, Alia, Balasubramanian, Sabapathy P
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Bioscientifica Ltd 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6510711/
https://www.ncbi.nlm.nih.gov/pubmed/31063971
http://dx.doi.org/10.1530/EDM-18-0136
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author Novodvorsky, Peter
Hussein, Ziad
Arshad, Muhammad Fahad
Iqbal, Ahmed
Fernando, Malee
Munir, Alia
Balasubramanian, Sabapathy P
author_facet Novodvorsky, Peter
Hussein, Ziad
Arshad, Muhammad Fahad
Iqbal, Ahmed
Fernando, Malee
Munir, Alia
Balasubramanian, Sabapathy P
author_sort Novodvorsky, Peter
collection PubMed
description 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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spelling pubmed-65107112019-05-15 Two cases of spontaneous remission of primary hyperparathyroidism due to auto-infarction: different management and their outcomes Novodvorsky, Peter Hussein, Ziad Arshad, Muhammad Fahad Iqbal, Ahmed Fernando, Malee Munir, Alia Balasubramanian, Sabapathy P Endocrinol Diabetes Metab Case Rep Unique/Unexpected Symptoms or Presentations of a Disease 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. Bioscientifica Ltd 2019-05-07 /pmc/articles/PMC6510711/ /pubmed/31063971 http://dx.doi.org/10.1530/EDM-18-0136 Text en © 2019 The authors http://creativecommons.org/licenses/by-nc-nd/4.0/ This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. (http://creativecommons.org/licenses/by-nc-nd/4.0/) .
spellingShingle Unique/Unexpected Symptoms or Presentations of a Disease
Novodvorsky, Peter
Hussein, Ziad
Arshad, Muhammad Fahad
Iqbal, Ahmed
Fernando, Malee
Munir, Alia
Balasubramanian, Sabapathy P
Two cases of spontaneous remission of primary hyperparathyroidism due to auto-infarction: different management and their outcomes
title Two cases of spontaneous remission of primary hyperparathyroidism due to auto-infarction: different management and their outcomes
title_full Two cases of spontaneous remission of primary hyperparathyroidism due to auto-infarction: different management and their outcomes
title_fullStr Two cases of spontaneous remission of primary hyperparathyroidism due to auto-infarction: different management and their outcomes
title_full_unstemmed Two cases of spontaneous remission of primary hyperparathyroidism due to auto-infarction: different management and their outcomes
title_short Two cases of spontaneous remission of primary hyperparathyroidism due to auto-infarction: different management and their outcomes
title_sort two cases of spontaneous remission of primary hyperparathyroidism due to auto-infarction: different management and their outcomes
topic Unique/Unexpected Symptoms or Presentations of a Disease
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6510711/
https://www.ncbi.nlm.nih.gov/pubmed/31063971
http://dx.doi.org/10.1530/EDM-18-0136
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