Giant Parathyroid Adenoma-Associated Fracture, Not All Lytic Bone Lesions are Cancer: A Case-Based Review

Abstract

Introduction: Due to the early diagnosis of primary hyperparathyroidism the musculoskeletal manifestations of this disease are becoming less frequent. When this disease manifests secondary to a giant adenoma, it presents with more aggressive symptoms and can have important repercussions such as the hungry bone syndrome after parathyroidectomy. There are few reported cases of hyperparathyroidism secondary to a giant adenoma in the literature, as the presence of a brown tumor is often misinterpreted as a metastatic lesion from an unknown primary tumor.

Methods: We describe a case and performed a literature review to identify all case reports. A literature search was carried out on PubMed/MEDLINE and EMBASE bibliographic databases. All available studies from May 2009 to May 2021 were included. Data were tabulated, and outcomes were cumulatively analyzed.

Results: Twenty-four cases of primary hyperparathyroidism due to giant adenoma have been described; the majority were women, with a mean age of 52 years. They presented with heterogeneous symptoms such as palpable nodules (45%), bone pain (33%), brown tumor (12.5%), asymptomatic (12.5%), metabolic profile with a mean calcemia of 13.8 mg/dL, PTH 1109 ng/L, and mean tumor weight of 47.24 g.

Conclusion: Primary hyperparathyroidism due to giant adenoma increases the risk of developing potentially serious postoperative complications such as hungry bone syndrome. This implies the need of implementing preventive measures comprising administration of intravenous zoledronic acid and early supplementation of oral calcium to prevent complications after resection.

Figure 1

Flowchart describing the case selection process.

Figure 2

(a) Chestx-ray showing loss of cortical continuity through the surgical neck of the right proximal humerus with a lytic lesion, suggesting an impacted fracture and cortical alteration of the distalclavicle. (b) Bone scintigraphy with MDP-99 m Tc, shows the abnormal distribution of the radiotracer due to diffuse and generalized uptake increase in the bone system, with a reduction in soft tissues. (c) Proximal right humerus biopsy showing multiple giant osteoclast cells, dispersed among a fibrous stroma with hemorrhagic foci and hemosiderin deposits and scarce osteoid with prominent resorption by osteoclasts indicating compatible with Brown tumor due to hyperparathyroidism.

Figure 3

A, B, C. Scintigraphy showing a nodular goiter with uptake of right thyroid lobule, and suppressed uptake in the rest of the glandule.

Figure 4

A, B, C, D. Biopsyo fright parathyroid mass. Mass showing 5 cm diameter and 16 grams weight. A neoplastic proliferation of parathyroid cells with areas of clear cells, oxyphilic cells, and chief cells. The lesion is profusely vascular with a hemorrhagicnet, with no frank necrosis. No macroscopical findings of malignancy.

Figure 5

Cruciform ligament: “Crowned dens syndrome”.