Spontaneous necrosis of parathyroid adenoma: biochemical and imaging follow-up for two years

Abstract

SummaryWe report a patient with a biochemically and radiologically confirmed parathyroid adenoma, which underwent spontaneous resolution by necrosis. The patient was followed-up over the subsequent two years during which time the tumour and hypercalcaemia recurred. Sequential radiological and biochemical changes at the time of diagnosis, spontaneous necrosis and recurrence are documented fully. Keywords: hyperparathyroidism; hypocalcaemia; apoplexy Primary hyperparathyroidism is one of the commonest endocrine disorders. However, spontaneous resolution by necrosis of a parathyroid adenoma is rare. Although there have been approximately 10 reported cases since the condition was first described in 1946, 1 in most cases the infarcted gland was removed soon after diagnosis. Therefore, the natural history, biochemical and hormonal changes following spontaneous infarction are not well known. We report a patient who had spontaneous infarction of her parathyroid adenoma, while awaiting surgery, with detailed documentation of the above-mentioned features. Case reportA 78-year-old woman was referred to our clinic for further management of Type 2 diabetes. She gave a history of renal stone, with right nephrectomy, 8 years before. She was put on oral hypoglycaemic therapy. She was found incidentally to have hypercalcaemia with a calcium concentration of 3.43 mmol/l (normal range 2.20-2.62 mmol/l) together with a reduced phosphate concentration of 0.55 mmol/l (0.82-1.40 mmol/l). Alkaline phosphatase was normal at 85 IU/l (45-145 IU/l). There was renal impairment with a plasma creatinine concentration of 160 µmol/l (44-107 µmol/l), but she had stable glycaemic control as indicated by a HbA 1c concentration of 7.4%. Physical examination revealed mild peripheral neuropathy and the nephrectomy scar, but no obvious neck mass. Further investigations confirmed primary hyperparathyroidism. Her parathyroid hormone (PTH) was 44 pmol/l (1.16-5.67 pmol/l, intact PTH assay; coefficient of variation 7%) with concomitant plasma calcium of 3.40 mmol/l and ionized calcium of 1.61 mmol/l (1.13-1.32 mmol/l).Ultrasound of the abdomen confirmed the previous right nephrectomy but showed no other abnormality. Her hypercalcaemia improved to approximately 2.8 mmol/l with adequate hydration, a low calcium diet and oral phosphate solution. She was referred for parathyroidectomy and was on the waiting list for surgery. During subsequent follow-up, her alkaline phosphatase gradually increased to 1465 IU/l, indicating development of parathyroid bone disease. Her glycaemic control was stable on oral hypoglycaemic therapy and there was no further deterioration of diabetic complications.She was put on the waiting list for surgery, which was very long in our hospital. Two years later, she underwent pre-operative imaging to localise the parathyroid adenoma. Computed tomography (CT) showed a 2.5 × 1.5 cm welldefined parathyroid adenoma posterior to the upper pole of the left lobe of the thyroid. The adenoma was of homogenous soft tissue attenuation and revea...

Atypical presentations of parathyroid gland pathology: A pictorial review

“…Rupture and haemorrhage of a parathyroid lesion (also referred to as parathyroid apoplexy) is the result of necrosis and involution. The thin capsule surrounding the gland predisposes to extracapsular dissection of haematoma into the deep neck spaces and mediastinum [20]. Patients can present acutely with airway compromise and sudden neck swelling as well as neck and/or chest bruising, dysphagia and dysphonia (the result of compression on the oesophagus, vagus or recurrent laryngeal nerve).…”

“…Serum biochemistry can be very variable with patients presenting in hypercalcaemic crises, in eucalcaemic or hypocalcaemic states; the latter two are thought to be the consequence of parathyroid gland infarction [21]. Whilst spontaneous gland infarction can lead to remission of primary hyperparathyroidism, this does not always equate to an auto-parathyroidectomy as biochemical evidence of recurrent disease have been reported, likely secondary to remnant parathyroid tissue [20].…”

Spontaneous necrosis of solid gallbladder adenocarcinoma accompanied with pancreaticobiliary maljunction

“…Moreover, GB cancer is sometimes detected in association with pancreaticobiliary maljunction (PBM), which is the union of the pancreatic and biliary ducts at a site outside the duodenal wall [3] . Some previous studies have described spontaneous necrosis of parathyroid and adrenal gland tumors [4][5][6][7] . However, spontaneous necrosis of GB tumors is quite rare.…”

Análise da expressão de genes ligados ao estresse de retículo endoplasmático no adenoma de paratireoide

“…A apoptose é, então, um mecanismo chave na preservação da qualidade de todo o processo de seleção protéica, eliminação de células mutantes e consequente inibição de proliferação tumoral (Rozpedek et al, 2016 (Chan et al, 2000;Baskar et al, 2004;Ahadizadeh et al, 2017). Chan et al (2000) observaram correlação da ocorrência de necrose com o tamanho do adenoma, principalmente com maior gravidade em nódulos acima de 2,0 cm de diâmetro, recomendando a indicação de tratamento cirúrgico para nódulos acima de 1,0 cm.…”

“…A apoptose é, então, um mecanismo chave na preservação da qualidade de todo o processo de seleção protéica, eliminação de células mutantes e consequente inibição de proliferação tumoral (Rozpedek et al, 2016 (Chan et al, 2000;Baskar et al, 2004;Ahadizadeh et al, 2017). Chan et al (2000) observaram correlação da ocorrência de necrose com o tamanho do adenoma, principalmente com maior gravidade em nódulos acima de 2,0 cm de diâmetro, recomendando a indicação de tratamento cirúrgico para nódulos acima de 1,0 cm. Há alguns estudos que mostram aumento de expressão de alguns genes relacionados à apoptose em células de adenoma de paratireoide, assim como no carcinoma e na hiperplasia, em diferentes proporçõ es, podendo servir como marcadores de diferenciação entre as apresentações e propiciar um tratamento cirúrgico mais adequando ao tipo histológico (Segiet et al, 2018).…”