Abstract
BackgroundDouble adenomas (DA) represents a distinct clinical entity of primary hyperparathyroidism (PHPT). DA may follow various embryologic distribution patterns and could be supernumerary and/or ectopic.Case presentationWe describe the first case of PHPT which comes as a result of double ipsilateral adenoma, of which one was both ectopic and supernumerary. A 45 year-old Greek male patient with diagnosed PHPT due to a single lower right parathyroid adenoma was admitted to our department for surgical treatment. The preoperative tests (neck US, Sestamibi scan) were conclusive for single gland disease. The patient underwent focused parathyroidectomy. The frozen section revealed a parathyroid adenoma with a slight possibility for parathyroid carcinoma. Ten minutes after the excision, intact PTH (iPTH) dropped >50% related to preoperative values and was within normal range. Right hemithyroidectomy with additional ipsilateral central neck dissection was performed, because of the possibility for parathyroid carcinoma. The final pathology report showed that the first excised tissue proved to be a parathyroid adenoma, while a second subcapsular one and a normal right upper parathyroid gland were also found.ConclusionsPreoperative localization of DA using routine imaging tests and the utility of intraoperative parathyroid hormone assay are still unreliable in detecting multiple adenomas. Furthermore, a slight possibility of a second and simultaneously supernumerary and ectopic adenoma maybe present. Therefore, it would be advisable to establish the use of more advanced imaging tests (such as 4D-CT, 4D-MRI) or other diagnostic tools when DA are suspected.
Highlights
Double adenomas (DA) represents a distinct clinical entity of primary hyperparathyroidism (PHPT)
A lot of work has been done in order to identify the pathogenesis of adenomas and the difference between them and parathyroid hyperplasia, it is still controversial if adenomas arise only by clonal or Regarding the distribution of double parathyroid adenomas, it seems that they have a nonuniform pattern as Milas et al observed in their study, it was found that superior-bilateral DA were statistically more frequent (45%)
DA constitute a real unique clinical entity of Primary Hyperparathyroidism (PHPT) and their distribution may vary widely, as in our patient, where the first adenoma was both ectopic and supernumerary and the second one was found under the thyroid capsule of the lower thyroid pole ipsilaterally
Summary
DA constitute a real unique clinical entity of PHPT and their distribution may vary widely, as in our patient, where the first adenoma was both ectopic and supernumerary and the second one was found under the thyroid capsule of the lower thyroid pole ipsilaterally. Their preoperative localization using the routine imagining tests is still rather disappointing as well as the adoption of ioPTH.
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