Epistaxis as an initial symptom of brown tumor in hard palate: a rare case report and review

Introduction

Brown tumors are uncommon and very aggressive benign intraosseous neoplasm caused by abnormally high parathyroid hormone levels (hyperparathyroidism; HPT). The ratio of female cases to male cases is approximately 2:1. The classical “brown tumor” is commonly observed in long bones, pelvis, and ribs [1] [2]. Only 4.5 % of brown tumors involve the facial bones [3]. The mandible is the predominantly affected site in the maxillofacial area [4]. Moreover, the palate as a site of brown tumors has only been reported in a limited number of papers, as determined through our PubMed search [5] [6] [7] [8] [9] [10]. In most cases, brown tumors represent late manifestations of primary hyperparathyroidism (PHPT) [11]. Pathological results include adenoma in approximately 80 % of cases, followed by glandular hyperplasia (15 %) and parathyroid carcinoma. Hypercalcemia and hypophosphatemia are commonly noted in PHPT [12]. Secondary hyperparathyroidism (SHPT), which is induced by defective phosphate excretion and failure of vitamin D activation, also plays a role in brown tumor formation. Elevated phosphate levels, as well as decreased calcium and serum vitamin D levels, lead to the persistent stimulation of the parathyroid glands, which increases PTH release [12] [13] [14]. Hyperparathyroidism may present as anorexia, vomiting, constipation, fatigue, weight loss, muscle weakness, psychiatric symptoms, anemia, polydipsia, urologic changes, and recurrent symptoms of kidney stones during the early stage [15] [16]. However, many of these early symptoms are often ignored [17]. Nevertheless, clinical, biochemical, and radiologic findings can easily guide the diagnosis if physicians carefully consider all the findings and their association with the subject's medical history, instead of focusing only on the bone lesion [18]. In the present case, the patient was admitted to our department for repeated epistaxis with nasal congestion on one side, which are typical manifestations of a brown tumor involving the hard palate and maxillary sinus [15] [19].

• References

• 1 Arunkumar KV, Kumar S, Deepa D. Brown tumor in mandible as a first sign of vitamin D deficiency: A rare case report and review. Indian J Endocrinol Metab 2012; 16 (02) 310-315
• 2 Zhang J, Wang H. et al. Brown tumor of the rib as a first presentation of primary hyperparathyroidism: Report of three cases and literature review. Thorac Cancer 2013; 4 (04) 474-478
• 3 Zou H, Song L. et al. Brown tumor of multiple facial bones associated with primary hyperparathyroidism: A clinical case report. Medicine (Baltimore) 2018; 97 (33) e11877
• 4 Chowdhury S, Aggarwal A. et al. Brown tumor of hyperparathyroidism involving craniomaxillofacial region: a rare case report and literature review. Minerva Stomatol 2013; 62 (09) 343-348
• 5 Perez-Guillermo M, Acosta-Ortega J. et al. Cytologic aspect of brown tumor of hyperparathyroidism. Report of a case affecting the hard palate. Diagn Cytopathol 2006; 34 (04) 291-294
• 6 Daniels JS. Primary hyperparathyroidism presenting as a palatal brown tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004; 98 (04) 409-413
• 7 Kar DK, Gupta SK. et al. Brown tumor of the palate and mandible in association with primary hyperparathyroidism. J Oral Maxillofac Surg 2001; 59 (11) 1352-1354
• 8 el Kohen A, Benjelloun A. et al. Primary hyperparathyroidism presenting as a brown tumor of the maxilla and hard palate. Apropos of a case. Rev Stomatol Chir Maxillofac 2001; 102 (02) 123-128
• 9 Scott SN, Graham SM. et al. Brown tumor of the palate in a patient with primary hyperparathyroidism. Ann Otol Rhinol Laryngol 1999; 108 (01) 91-94
• 10 Sutbeyaz Y, Yoruk O. et al. Primary hyperparathyroidism presenting as a palatal and mandibular brown tumor. J Craniofac Surg 2009; 20 (06) 2101-2104
• 11 Admassie D, Kebed T. et al. Brown tumor an uncommon and late manifestation of hyperparathyroidism: a case series done at Tikur Anbessa Specialized Hospital, Addis Ababa. Ethiop Med J 2011; 49 (02) 149-153
• 12 Selvi F, Cakarer S. et al. Brown tumour of the maxilla and mandible: a rare complication of tertiary hyperparathyroidism. Dentomaxillofac Radiol 2009; 38 (01) 53-58
• 13 Mellouli N, Belkacem Chebil R. et al. Mandibular Osteitis Fibrosa Cystica as First Sign of Vitamin D Deficiency. Case Rep Dent 2018; 2018: 6814803
• 14 Al-Sharafi BA, Al-Imad SA. et al. Brown tumours of the tibia and second metacarpal bone in a woman with severe vitamin D deficiency. BMJ Case Rep 2015; DOI: bcr2014207722.
• 15 Lessa MM, Sakae FA. et al. Brown tumor of the facial bones: case report and literature review. Ear Nose Throat J 2005; 84 (07) 432-434
• 16 Pahlavan PS, Severin MC. Parathyroid carcinoma: A rare case with mandibular brown tumor. Wien Klin Wochenschr 2006; 118 (05) 175-179
• 17 Hussain M, Hammam M. Management challenges with brown tumor of primary hyperparathyroidism masked by severe vitamin D deficiency: a case report. J Med Case Rep 2016; 10: 166
• 18 Diamanti-Kandarakis E, Livadas S. et al. Brown tumor of the fibula: unusual presentation of an uncommon manifestation. Report of a case and review of the literature. Endocrine 2007; 32 (03) 345-349
• 19 Erdem CZ, Erdem LO. et al. Brown tumor of the maxillary sinus: a case report. Kulak Burun Bogaz Ihtis Derg 2003; 11 (04) 121-124
• 20 Cicconetti A, Matteini C. et al. Differential diagnosis in a case of brown tumor caused by primary hyperparathyroidism. Minerva Stomatol 1999; 48 (11) 553-558
• 21 Tarello F, Ottone S. et al. Brown tumor of the jaws. Minerva Stomatol 1996; 45 (10) 465-470
• 22 Unlu RE, Abaci E. et al. Brown tumor in children with normocalcemic hyperparathyroidism: a report of two cases. J Craniofac Surg 2003; 14 (01) 69-73
• 23 Misiorowski W, Czajka-Oraniec I. et al. Osteitis fibrosa cystica – a forgotten radiological feature of primary hyperparathyroidism. Endocrine 2017; 58 (02) 380-385
• 24 Panagopoulos A, Tatani I. et al. Osteolytic lesions (brown tumors) of primary hyperparathyroidism misdiagnosed as multifocal giant cell tumor of the distal ulna and radius: a case report. J Med Case Rep 2018; 12 (01) 176
• 25 Yalcin MB, Frauenhoffer EE. et al. A case of brown tumor mimicking fibrous dysplasia in a patient with chronic renal failure. Acta Orthop Traumatol Turc 2008; 42 (04) 296-301
• 26 Emin AH, Süoğlu Y. et al. Normocalcemic hyperparathyroidism presented with mandibular brown tumor: report of a case. Auris Nasus Larynx 2004; 31 (03) 299-304
• 27 Su AW, Chen CF. et al. Primary hyperparathyroidism with brown tumor mimicking metastatic bone malignancy. J Chin Med Assoc 2010; 73 (03) 177-180
• 28 Franco M, Bendini JC. et al. Radiographic follow-up of a phalangeal brown tumor. Joint Bone Spine 2002; 69 (05) 506-510
• 29 Resendiz-Colosia JA, Rodríguez-Cuevas SA. et al. Evolution of maxillofacial brown tumors after parathyroidectomy in primary hyperparathyroidism. Head Neck 2008; 30 (11) 1497-1504
• 30 Baracaldo RM, Bao D. et al. 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. Hemodial Int 2015; 19 (04) 583-592
• 31 Amini Nezhad F, Payab M. et al. Jaw tumor in recurrent primary hyperparathyroidism: A case report. Int J Surg Case Rep 2018; 52: 54-58
• 32 Pecovnik Balon B, Kavalar R. Brown tumor in association with secondary hyperparathyroidism. A case report and review of the literature. Am J Nephrol 1998; 18 (05) 460-463
• 33 Quarles LD, Yohay DA. et al. Prospective trial of pulse oral versus intravenous calcitriol treatment of hyperparathyroidism in ESRD. Kidney Int 1994; 45 (06) 1710-1721
• 34 Leal CT, Lacativa PG. et al. Surgical approach and clinical outcome of a deforming brown tumor at the maxilla in a patient with secondary hyperparathyroidism due to chronic renal failure. Arq Bras Endocrinol Metabol 2006; 50 (05) 963-967
• 35 Triantafillidou K, Zouloumis L. et al. Brown tumors of the jaws associated with primary or secondary hyperparathyroidism. A clinical study and review of the literature. Am J Otolaryngol 2006; 27 (04) 281-286
• 36 Mantar F, Gunduz S, Gunduz UR. A reference finding rarely seen in primary hyperparathyroidism: brown tumor. Case Rep Med 2012; 265 (05) 565
• 37 Kartini D. et al. Craniofacial Brown Tumor in Patients with Secondary Hyperparathyroidism to Chronic Renal Failure: Report of Two Cases in Cipto Mangunkusumo Hospital. Case Rep Oncol Med 2018 Volume 2018, Article ID 1801652, 7 pages
• 38 Khalil PN, Heining SM. et al. Natural history and surgical treatment of brown tumor lesions at various sites in refractory primary hyperparathyroidism. Eur J Med Res 2007; 12 (05) 222-230
• 39 Keyser JS, Postma GN. Brown tumor of the mandible. Am J Otolaryngol 1996; 17 (06) 407-410
• 40 Gheith O, Ammar H. et al. Spinal compression by brown tumor in two patients with chronic kidney allograft failure on maintenance hemodialysis. Iran J Kidney Dis 2010; 4 (03) 256-259
• 41 Mateo L, Massuet A. et al. Brown tumor of the cervical spine: a case report and review of the literature. Clin Rheumatol 2011; 30 (03) 419-424
• 42 Tarrass F, Ayad A. et al. Cauda equina compression revealing brown tumor of the spine in a long-term hemodialysis patient. Joint Bone Spine 2006; 73 (06) 748-750
• 43 Vandenbussche E, Schmider L. et al. Brown tumor of the spine and progressive paraplegia in a hemodialysis patient. Spine (Phila Pa 1976) 2004; 29 (12) E251-E255
• 44 Menard P, Pauzie F. et al. A brown tumor of the mandible and hyperparathyroidism secondary to chronic renal insufficiency. Review of the literature apropos of a case. Rev Stomatol Chir Maxillofac 1993; 94 (05) 276-280
• 45 Bandeira F, Griz L. et al. From mild to severe primary hyperparathyroidism: The Brazilian experience. Arq Bras Endocrinol Metabol 2006; 50 (04) 657-663
• 46 Raef H, Ingemansson S. et al. The effect of vitamin D status on the severity of bone disease and on the other features of primary hyperparathyroidism (pHPT) in a vitamin D deficient region. J Endocrinol Invest 2004; 27 (09) 807-812
• 47 Silverberg SJ. Vitamin D deficiency and primary hyperparathyroidism. J Bone Miner Res 2007; 22 (Suppl. 02) V100-V104
• 48 Franca TC, Griz LC. et al. Bisphosphonates can reduce bone hunger after parathyroidectomy in patients with primary hyperparathyroidism and osteitis fibrosa cystica. Rev Bras Reumatol 2011; 51 (02) 131-137
• 49 Azria A, Beaudreuil J. et al. Brown tumor of the spine revealing secondary hyperparathyroidism. Report of a case. Joint Bone Spine 2000; 67 (03) 230-233
• 50 Pinto LP, Cherubinim K. et al. Highly aggressive brown tumor in the jaw associated with tertiary hyperparathyroidism. Pediatr Dent 2006; 28 (06) 543-546
• 51 Sezer B, Koyuncu B. et al. Intralesional corticosteroid injection for central giant cell granuloma: a case report and review of the literature. Turk J Pediatr 2005; 47 (01) 75-81
• 52 Mittendorf EA, McHenry CR. Persistent parathyroid hormone elevation following curative parathyroidectomy for primary hyperparathyroidism. Arch Otolaryngol Head Neck Surg 2002; 128 (03) 275-279
• 53 Philip George AJ, Banerji JS. Brown tumor and staghorn calculi in primary hyperparathyroidism. Urology 2013; 82 (02) e13-e14