Abstract
Brown tumors (BT) are benign focal bone lesions that may appear in the context of primary and secondary hyperparathyroidism (HPT). Involvement of the spine is exceedingly rare. We present a case of brown tumor involving the cervical spine, the third reported in the literature. In the literature review (until August 2010), we found nine cases of spinal BT in primary HPT and 14 cases in secondary HPT. Fifteen patients (65%) had evidence of spinal cord compression. A 34-year-old woman on long-term hemodialysis, with secondary HPT, presented with a 9-month history of persistent neck pain. Radiographs of the cervical spine revealed an expansive osteolytic lesion in the posterior arch of the second cervical vertebra. MR imaging revealed an expansive mass on C2 affecting the vertebral body, odontoid process, right pedicle, laminas, and spinous process; there were no signs of spinal edema. A CT-guided needle biopsy of the lesion showed destruction of trabecular bone, infiltration of the fibroblastic cells, and abundant osteoclast-like multinucleated giant cells with hemorrhage and hemosiderin pigment, and the diagnosis of brown tumor was made. Cervical pain disappeared within a few days of parathyroidectomy, and rapid remineralization of C2 was evident within a few months. BT must always be considered in the context of hyperparathyroidism and osteolytic lesions. Vertebral BT can be particularly devastating due to medullar compression symptoms. Regression or complete disappearance of these lesions after parathyroidectomy is common, but prompt surgical decompression is necessary in case of medullar compression symptoms.
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References
Azria A, Beaudreuil J, Juquel JP, Quillard A, Bardin T (2000) Brown tumor of the spine revealing secondary hyperparathyroidism. Report of a case. Joint Bone Spine 67:230–3
Shaw MT, Davies M (1968) Primary hyperparathyroidism presenting as spinal cord compression. Br Med J 4:230–231
Barlow IW, Archer IA (1993) Brown tumor of the cervical spine. Spine 18:936–7
Hoshi M, Takami M, Kajikawa M, Teramura K, Okamoto T, Yanagida I, Matsumura A (2008) A case of multiple skeletal lesions of brown tumors, mimicking carcinoma metastases. Arch Orthop Trauma Surg 128:149–54
Vandenbussche E, Schmider L, Mutschler C, Man M, Jacquot C, Augereau B (2004) Brown tumor of the spine and progressive paraplegia in a hemodialysis patient. Spine 29:E251–5
Marini M, Vidiri A, Guerrisi R, Campodonico F, Ponzio R (1992) Progress of brown tumors in patients with renal insufficiency undergoing dialysis. Eur J Radiol 14:67–71
Tarrass F, Ayad A, Benjelloun M, Anabi A, Ramdani B, Benghanem MG, Zaid D (2006) Cauda equina compression revealing brown tumor of the spine in a long-term hemodialysis patient. Joint Bone Spine 73:748–50
Kaya RA, Cavuşoğlu H, Tanik C, Kahyaoğlu O, Dilbaz S, Tuncer C, Aydin Y (2007) Spinal cord compression caused by a brown tumor at the cervicothoracic junction. Spine J 7:728–32
Sundaram M, Scholz C (1977) Primary hyperparathyroidism presenting with acute paraplegia. Am J Roentgenol 128:674–676
Ganesh A, Kurian S, John L (1981) Complete recovery of spinal cord compression following parathyroidectomy. Postgrad Med J 57:652–653
Yokota N, Kuribayashi T, Nagamine M, Tanaka M, Matsukura S, Wakisaka S (1989) Paraplegia caused by brown tumor in primary hyperparathyroidism. Case report. J Neurosurg 71:446–8
Kashkari S, Kelly TR, Bethem D, Pepe RG (1990) Osteitis fibrosa cystica (Brown tumor) of the spine with cord compression: report of a case with needle aspiration biopsy findings. Diagn Cytopathol 6:349–53
Mustonen AO, Kiuru MJ, Stahls A, Bohling T, Kivioja A, Koskinen SK (2004) Radicular lower extremity pain as the first symptom of primary hyperparathyroidism. Skeletal Radiol 33:467–72
Altan L, Kurtoğlu Z, Yalçınkaya U, Aydınlı U, Ertürk E (2007) Brown tumor of the sacral spine in a patient with low-back pain. Rheumatol Int 28:77–81
Haddad FH, Malkawi OM, Sharbaji AA, Jbara IF, Rihani HR (2007) Primary hyperparathyroidism. A rare cause of spinal cord compression. Saudi Med J 28:783–6
Ericsson M, Holm E, Ingemansson S, Lindholm T, Svendgaard NA (1978) Secondary hyperparathyroidism combined with uremia and giant cell containing tumor of the cervical spine. A case report. Scand J Urol Nephrol 12:185–7
Bohlman ME, Kim YC, Eagan J, Spees EK (1986) Brown tumor in secondary parathyroidism causing acute paraplegia. Am J Med 81:545–7
Pumar JM, Alvarez M, Pérez Batallón A, Vidal J, Lado J, Bollar A (1990) Brown tumor in secondary hyperparathyroidism, causing progressive paraplegia. Neuroradiology 32:343
Fineman I, Johnson JP, Di-Patre PL, Sandhu H (1999) Chronic renal failure causing brown tumors and myelopathy. Case report and review of pathophysiology and treatment. J Neurosurg 90:242–6
Masutani K, Katafuchi R, Uenoyama K, Saito S, Fujimi S, Hirakata H (2001) Brown tumor of the thoracic spine in a patient on long-term hemodialysis. Clin Nephrol 55:419–23
Jackson W, Sethi A, Carp J, Talpos G, Vaidya R (2007) Unusual spinal manifestation in secondary hyperparathyroidism: a case report. Spine 32:E557–60
Ren W, Wang X, Zhu B, Liu Z (2008) Progressive paraplegia in a long-term hemodialysis patient. Am J Kidney Dis 52:A 37–9
Zaheer SN, Byrne ST, Poonnoose SI, Vrodos NJ (2009) Brown tumor of the spine in a renal transplant patient. J Clin Neurosci 16:1230–32
Mak KC, Wong YW, Lik KDK (2009) Spinal cord compression secondary to brown tumour in a patient on long-term haemodialysis: a case report. J Orthop Surg Hong Kong 17:90–5
Arabi A, Khoury N, Zahed L, Birbari A, El-Hajj Fuleihan G (2006) Regression of skeletal manifestations of hyperparathyroidism with oral vitamin D. J Clin Endocrinol Metab 91:2480–3
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Mateo, L., Massuet, A., Solà, M. et al. Brown tumor of the cervical spine: a case report and review of the literature. Clin Rheumatol 30, 419–424 (2011). https://doi.org/10.1007/s10067-010-1608-y
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DOI: https://doi.org/10.1007/s10067-010-1608-y