Hypocalcemia due to Spontaneous Infarction of Parathyroid Adenoma and Osteomalacia in a Patient with Primary Hyperparathyroidism.

Abstract

Abstract. A 49 year-old Japanese woman had subjected enlargement of a cervical tumor, and also suffered two bone fractures in 2 years. The cervical tumor had enlarged further in the month prior to admission, becoming warm and tender. Endocrinological examination revealed that the serum intact PTH concentration was remarkably high at 400 pg/mL despite the low serum calcium concentration, and that the serum vitamin Ds concentration was decreased. Bone roentgenograms revealed severe osteolytic changes compatible with osteitis fibrosa cystica and a pathologic fracture of the humerus. Under a diagnosis of primary hyperparathyroidism, parathyroidectomy was performed, followed by fixation surgery for the pathologic fracture. Histologically, the cervical tumor was a parathyroid chiefcell adenoma with massive necrosis, and the bone pathology by iliac bone biopsy revealed the existence of osteomalacia. She was treated with calcium, vitamins D and K2 and calcitonin after the surgery. This case is a rare condition manifesting hypocalcemia with catastrophic osteoporosis under the coexistence of spontaneous infarction of parathyroid adenoma with osteomalacia, suggesting that the clinical features of hyperparathyroidism are modified by both the autoparathyroidiectomy and the existence of osteomalacia due to vitamin D deficiency.

Parathyroid adenoma autoinfarction: A report of a case

“…The earliest description of this entity was by Norris in 1946 1. Since this initial description, several cases have been reported, most recently in 1998 by Kovacs and Gay12 and Otsuka et al13 The clinical presentation, laboratory values, and physical examination findings of our patient were consistent with previously described cases in the literature.…”

“…Adenoma autoinfarction has resulted in spontaneous resolution of primary hyperparathyroidism 1–13. The earliest description of this entity was by Norris in 1946 1.…”

“…Although this is an acceptable means of management, reports of postinfarction progressive elevation in PTH levels11 are a strong argument for definitive management with surgical resection. In addition, most reports cited in the literature were managed with resection of the infarcted adenoma rather than expectant management 1–9, 11, 13, 14. Our patient had persistent local symptoms that resolved after definitive surgical management.…”

“…Parathyroid adenoma autoinfarction, although uncommon, is an entity that has been previously reported in the literature 1–14. The mechanism of action has yet to be determined, and patient presentation varies from case to case.…”