Parathyromatosis: a rare yet problematic etiology of recurrent and persistent hyperparathyroidism

Abstract

Recurrent or persistent hyperparathyroidism is an uncommon yet challenging clinical problem, and parathyromatosis is one of its very rare causes. In this minireview, we review causes of recurrent hyperparathyroidism and all cases of parathyromatosis available in the literature. The clinical course of a case of parathyromatosis with the longest follow-up (1977-2011) is described. Similar cases reported between 1975 and the present are reviewed and analyzed to characterize the clinical presentation, course, and management of this rare condition. Parathyromatosis, which is benign parathyroid tissue seeding, has been detailed in 35 patients in the English literature. The majority were female subjects, with end-stage renal disease, in their fifth to sixth decade of life. In most cases, the diagnosis was made intraoperatively; and the condition was often refractory to surgery. A calcimimetic agent was used in 5 cases with end-stage renal disease; serum calcium and/or parathyroid hormone levels decreased in 4 subjects, but only one was reported to experience increments in bone density. Medical management combining a calcimimetic with a bisphosphonate may therefore be a preferred alternative.