Langerhans Cell Histiocytosis of the Adult Cervical Spine: A Case Report and Literature Review

Abstract

A 36-year-old man was diagnosed with Langerhans cell histiocytosis (LCH) of the cervical spine with a unifocal expansive osteolytic lesion of C4. The surgical management with a 2-year follow-up and a review of the literature on LCH of the cervical spine are presented. Although a rare condition, LCH is an important differential diagnosis of any osteolytic lesion in the cervical spine with localized pain in a young adult patient. Review of the literature suggests a higher prevalence of LCH lesions affecting the cervical spine as compared with the thoracic or lumbar spine than historically reported.

Keywords

Langerhans cell histiocytosis - eosinophilic granuloma - cervical spine - spinal neoplasms

• References

• 1 Stockschlaeder M, Sucker C. Adult Langerhans cell histiocytosis. Eur J Haematol 2006; 76 (05) 363-368
• 2 Tomita K, Kawahara N, Baba H, Tsuchiya H, Fujita T, Toribatake Y. Total en bloc spondylectomy. A new surgical technique for primary malignant vertebral tumors. Spine 1997; 22 (03) 324-333
• 3 Brown CW, Jarvis JG, Letts M, Carpenter B. Treatment and outcome of vertebral Langerhans cell histiocytosis at the Children's Hospital of Eastern Ontario. Can J Surg 2005; 48 (03) 230-236
• 4 Bertram C, Madert J, Eggers C. Eosinophilic granuloma of the cervical spine. Spine 2002; 27 (13) 1408-1413
• 5 Islinger RB, Kuklo TR, Owens BD. , et al. Langerhans' cell histiocytosis in patients older than 21 years. Clin Orthop Relat Res 2000; (379) 231-235
• 6 Götz G, Fichter J. Langerhans'-cell histiocytosis in 58 adults. Eur J Med Res 2004; 9 (11) 510-514
• 7 Aricò M, Girschikofsky M, Généreau T. , et al. Langerhans cell histiocytosis in adults. Report from the International Registry of the Histiocyte Society. Eur J Cancer 2003; 39 (16) 2341-2348
• 8 Zheng W, Wu J, Wu Z, Xiao J. Atlantoaxial instability secondary to eosinophilic granuloma of the axis in adults: long-term follow-up in six cases. Spine J 2014; 14 (11) 2701-2709
• 9 Cantu MA, Lupo PJ, Bilgi M, Hicks MJ, Allen CE, McClain KL. Optimal therapy for adults with Langerhans cell histiocytosis bone lesions. PLoS One 2012; 7 (08) e43257
• 10 Plasschaert F, Craig C, Bell R, Cole WG, Wunder JS, Alman BA. Eosinophilic granuloma. A different behaviour in children than in adults. J Bone Joint Surg Br 2002; 84 (06) 870-872
• 11 Huang W, Yang X, Cao D. , et al. Eosinophilic granuloma of spine in adults: a report of 30 cases and outcome. Acta Neurochir (Wien) 2010; 152 (07) 1129-1137
• 12 Garg S, Mehta S, Dormans JP. Langerhans cell histiocytosis of the spine in children. Long-term follow-up. J Bone Joint Surg Am 2004; 86-A [phone]
• 13 Chaudhary V, Bano S, Aggarwal R. , et al. Neuroimaging of Langerhans cell histiocytosis: a radiological review. Jpn J Radiol 2013; 31 (12) 786-796
• 14 Chacha PB, Khong BT. Eosinophilic granuloma of bone. A diagnostic problem. Clin Orthop Relat Res 1971; 80 (80) 79-88
• 15 Bunch WH. Orthopedic and rehabilitation aspects of eosinophilic granuloma. Am J Pediatr Hematol Oncol 1981; 3 (02) 151-156
• 16 Ferris RA, Pettrone FA, McKelvie AM, Twigg HL, Chun BK. Eosinophilic granuloma of the spine: an unusual radiographic presentation. Clin Orthop Relat Res 1974; (99) 57-63
• 17 Nesbit ME, Kieffer S, D'Angio GJ. Reconstitution of vertebral height in histiocytosis X: a long-term follow-up. J Bone Joint Surg Am 1969; 51 (07) 1360-1368
• 18 Ochsner SF. Eosinophilic granuloma of bone; experience with 20 cases. Am J Roentgenol Radium Ther Nucl Med 1966; 97 (03) 719-726
• 19 Huang WD, Yang XH, Wu ZP. , et al. Langerhans cell histiocytosis of spine: a comparative study of clinical, imaging features, and diagnosis in children, adolescents, and adults. Spine J 2013; 13 (09) 1108-1117
• 20 Jiang L, Liu ZJ, Liu XG. , et al. Langerhans cell histiocytosis of the cervical spine: a single Chinese institution experience with thirty cases. Spine 2010; 35 (01) E8-E15
• 21 Chen SY, Chao SC, Kao TH, Shen CC, Tsou HK. Surgical treatment using mesh cage and plate fixation in a 9-year-old child with solitary histiocytosis lesion in the cervical spine. Eur J Pediatr Surg 2011; 21 (03) 189-191
• 22 Simanski C, Bouillon B, Brockmann M, Tiling T. The Langerhans' cell histiocytosis (eosinophilic granuloma) of the cervical spine: a rare diagnosis of cervical pain. Magn Reson Imaging 2004; 22 (04) 589-594
• 23 Abu-Bonsrah N, Goodwin CR, Crane GM, Abiola G, Sciubba DM. Langerhans cell histiocytosis of the cervical spine. Spine J 2016; 16 (01) e11-e12
• 24 Kotecha R, Venkatramani R, Jubran RF, Arkader A, Olch AJ, Wong K. Clinical outcomes of radiation therapy in the management of Langerhans cell histiocytosis. Am J Clin Oncol 2014; 37 (06) 592-596
• 25 Kriz J, Eich HT, Bruns F. , et al. Radiotherapy in Langerhans cell histiocytosis—a rare indication in a rare disease. Radiat Oncol 2013; 8: 233
• 26 Hyman DM, Puzanov I, Subbiah V. , et al. Vemurafenib in multiple nonmelanoma cancers with BRAF V600 mutations. N Engl J Med 2015; 373 (08) 726-736
• 27 Badalian-Very G, Vergilio JA, Degar BA. , et al. Recurrent BRAF mutations in Langerhans cell histiocytosis. Blood 2010; 116 (11) 1919-1923