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. 2020 Aug;27(3):217-226.
doi: 10.11005/jbm.2020.27.3.217. Epub 2020 Aug 31.

Three Rare Concurrent Complications of Tertiary Hyperparathyroidism: Maxillary Brown Tumor, Uremic Leontiasis Ossea, and Hungry Bone Syndrome

Natalie Bransky  1 , Neena Ramesh Iyer  1 , Sophie Mestman Cannon  1 , Alexander Hanlin Tyan  1 , Praneet Mylavarapu  1 , Ryan Orosco  1 , David Brain Hom  1 , Alan Ali Moazzam  1
Affiliations

Three Rare Concurrent Complications of Tertiary Hyperparathyroidism: Maxillary Brown Tumor, Uremic Leontiasis Ossea, and Hungry Bone Syndrome

Natalie Bransky et al. J Bone Metab. 2020 Aug.

Abstract

A 48-year-old woman in her 40's with end-stage renal disease and tertiary hyperparathyroidism (HPT) presented for a rapidly progressive maxillary tumor. Initial workup was notable for elevated intact parathyroid hormone (PTH) and diffuse thickening of skull and facial bones on computed tomography, and maxillary tumor biopsy with multinucleated giant cells. She underwent subtotal parathyroidectomy (with removal of a parathyroid adenoma and 2 hyperplastic glands) and partial resection of maxillary brown tumor. The patient's post-operative course was complicated by hungry bone syndrome, with hypocalcemia refractory to aggressive calcium repletion. Teriparatide (recombinant PTH) was utilized with rapid resolution of hypocalcemia. To our knowledge, this is the first case of maxillary brown tumor in tertiary HPT to be reported in the USA. This case also supports teriparatide as a novel therapeutic for hungry bone syndrome refractory to aggressive calcium repletion.

Keywords: Brown tumor; Hungry bone syndrome; Maxilla; Teriparatide; Tertiary hyperparathyroidism; Uremic leontiasis ossea.

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Conflict of interest statement

Conflicts of interest

No potential conflict of interest relevant to this article was reported.

Figures

Fig. 1.
Fig. 1.
Gross appearance of maxillary brown tumor. Color photograph of the maxillary tumor. The mass was sub-mucosal with visible ulceration of the mucosa due to trauma from the mandibular dentition.
Fig. 2.
Fig. 2.
Computed tomography (CT) appearance of oral tumor. Three panels of axial CT images, without contrast, demonstrating skull bone changes resulting from renal osteodystrophy. Thickening of the maxilla and zygoma (A), mandibular rami (B), and mandibular bodies (C). The maxillary tumor is visible in the center image as a round mass on the right anterior palate (star). The maxillary teeth are splayed (arrows).
Fig. 3.
Fig. 3.
Tc-99m MDP whole body bone scintigraphy. Nuclear bone scan images showing diffusely increased uptake in the skull, mandible, and spine consistent with renal osteodystrophy and uremic leontiasis ossea. Areas of mild focal uptake also seen in the manubrium and right 6th rib corresponding to lytic lesion on computed tomography.
Fig. 4.
Fig. 4.
Histopathology of tumor biopsy. Photomicrographs of tumor biopsy, showing scattered and clustered multinucleated giant cells in a fibrous background.
See this image and copyright information in PMC

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