Surgical approach and clinical outcome of a deforming brown tumor at the maxilla in a patient with secondary hyperparathyroidism due to chronic renal failure

Abstract

Brown tumors are relatively uncommon but they are serious complications of renal osteodystrophy. We describe a 31-year-old woman with end-stage renal disease who had undergone hemodialysis for nine years and developed severe secondary hyperparathyroidism and a maxilla brown tumor despite increasing doses of oral calcitriol and calcium carbonate. The fast increase of the right maxillary bone tumor led to indication of parathyroidectomy (PTx). Despite normalization of serum PTH there was a slow regression of the mass and the patient still complained about her appearance after two-years of follow-up. Excision of the maxillary mass followed by recontouring of the maxilla was then performed, with adequate masticator rehabilitation. Tumores marrons são relativamente incomuns mas constituem sérias complicações da osteodistrofia renal. Descrevemos o caso de uma paciente, 31 anos, com doença renal em estágio terminal, em hemodiálise há nove anos, que desenvolveu severo hiperparatireoidismo secundário com tumor marrom em maxila apesar de doses crescentes de calcitriol oral e carbonato de cálcio. O rápido aumento do tumor marrom em maxila levou à indicação de paratireoidectomia (PTx). Apesar da normalização dos níveis de PTH sérico, a regressão da massa tumoral foi lenta e a paciente questionava sobre sua aparência após dois anos de seguimento. A excisão da massa maxilar foi seguida da reconstrução com adequada função mastigatória. BROWN TUMORS ARE EROSIVE bony lesions caused by rapid osteoclastic activity and peritrabecular fibrosis due to hyperparathyroidism (HPT) resulting in a local destructive phenomenon. Actually they represent a reparative cellular process rather than a true neoplasia (1,2). They are known to occur only in the setting of HPT, and are considered the most pathognomonic skeletal changes that accompany this disease. For years, these lesions have been recognized in primary hyperparathyroidism (HPT1) (3). However, brown tumors have also been reported in patients with severe hyperparathyroidism secondary to chronic renal failure (HPT2), especially those on long-term hemodialysis (4,5).

Facial disfigurement due to osteitis fibrosa cystica or brown tumor from secondary hyperparathyroidism in patients on dialysis: A systematic review and an illustrative case report

“…[32][33][34][35] These symptoms are typically referred for treatment to specialties of maxillofacial surgery, oral surgery, reconstructive and plastic surgery, ENT, ophthalmology, and dentistry. The lesions from OFC and BT are not painful and often produce symptoms, depending on the location of facial asymmetry, oral mass, nasal obstruction, sinusitis, progressive visual disturbances, proptosis, chewing diffi-culties, hearing abnormalities, and airway obstruction in the pediatric population.…”

Brown Tumor at the Jaw in Patients With Secondary Hyperparathyroidism Due to Chronic Renal Failure

“…Radiographically, they appear as well demarcated monolocular or multilocular osteolytic lesions. In the mandible, the cortical bone is expanded and thinned (12,14). Brown tumors of the jaws occasionally result in root resorption and loss of the lamina dura and may present as a space occupying mass in the sinus (17).…”

“…Actually they represent a reparative cellular process rather than a true neoplasia (6, 12). They are known to occur only in the setting of HPT, and are considered the most pathognomonic skeletal changes that accompany this disease (12). At skeletal sites excess parathyroid hormone can lead to a condition called osteitis fibrosa cystica.…”

Brown tumors in dialyzed patients with secondary hyperparathyroidism: Report of 16 cases

“…Leal et al. reported a slow regression of a maxillary BT after 2 years of follow‐up, requiring local excision . Therefore, an early diagnosis and a timely subtotal PTX in a medically resistant sHPT are an optimal option to control the growth of the bony lesion and to avoid further weakening of bone structure and consequent increased risk of fractures, compression of contiguous structures, deformities, and functional alteration of other involved areas of the skeleton …”