Cervical Eosinophilic Granuloma in Adolescents: Case Report and Literature Review

Abstract

Introduction Eosinophilic granuloma (EG) is a rare, tumor-like lesion, infrequently affecting the cervical spine, particularly in adults. Although vastly described in literature, this pathology mainly affects children, and there is still no consensus on its treatment in older patients. With the goal of contributing to increase the knowledge regarding this infrequent pathology, we present a case of a C3 eosinophilic granuloma in a 16-year-old patient, who was treated conservatively, with good results, including complete return to his previous activities.

Clinical case a 16-year-old male, elite rugby player, presented with a history of persistent neck pain, mainly at night, with no previous trauma. Upon physical examination, he reported neck pain with axial compression of the head, without neurological impairment. Both computed tomography (CT) and magnetic resonance imaging (MRI) scan revealed an aggressive lytic lesion in the C3 vertebral body, a with monostotic presentation on positron emission tomography-computed tomography (PET-CT) compatible with a primary spine tumor. A CT-guided percutaneous biopsy was obtained to establish the diagnosis and provide the proper management. The results were compatible with Langerhans cells. As he presented no symptoms or imaging findings of evident bone instability, as well as no neurological impairment, the patient was treated conservatively, with a cervical brace, oral pain medication and close follow-up. A CT obtained after four months of treatment showed reparative changes of the C3 vertebral body; at this point, the patient reported no neck pain, so he was able to return to his previous activities.

Conclusions Although an EG is rare at this age, it should be considered in the differential diagnosis of primary vertebral aggressive lytic lesions. Imaging and a vertebral biopsy are paramount to confirm the diagnosis. The treatment modality depends on the symptoms, the involvement of adjacent structures, and the stability of the affected vertebra. Conservative management including clinical and imaging follow-up is a viable option.

Keywords

eosinophilic granuloma - bone tumor - cervical - Langerhans cell histocytosis - histocytosis X

Publication History

Received: 09 October 2020

Accepted: 18 March 2022

Article published online:
03 October 2022

• Referencias

• 1 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
• 2 Bertram C, Madert J, Eggers C. Eosinophilic granuloma of the cervical spine. Spine 2002; 27 (13) 1408-1413
• 3 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 http://www.ncbi.nlm.nih.gov/pubmed/12211681
• 4 Reddy PK, Vannemreddy PSSV, Nanda A. Eosinophilic granuloma of spine in adults: a case report and review of literature. Spinal Cord 2000; 38 (12) 766-768
• 5 Greenlee JDW, Fenoy AJ, Donovan KA, Menezes AH. Eosinophilic granuloma in the pediatric spine. Pediatr Neurosurg 2007; 43 (04) 285-292
• 6 Sekiya JK, Kuhn JE. Instability of the proximal tibiofibular joint. J Am Acad Orthop Surg 2003; 11 (02) 120-128
• 7 Dickinson LD, Farhat SM. Eosinophilic granuloma of the cervical spine. A case report and review of the literature. Surg Neurol 1991; 35 (01) 57-63
• 8 Prasad GL, Divya S. Eosinophilic Granuloma of the Cervical Spine in Adults: A Review. World Neurosurg 2019; 125: 301-311
• 9 Angelini A, Mavrogenis AF, Rimondi E, Rossi G, Ruggieri P. Current concepts for the diagnosis and management of eosinophilic granuloma of bone. J Orthop Traumatol 2017; 18 (02) 83-90
• 10 Song Y, Geng W, Guo T. et al. The outcome of eosinophilic granuloma involving unilateral atlantoaxial joint: A case report and literature review. Medicine (Baltimore) 2017; 96 (25) e7197
• 11 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
• 12 Drevelegas A, Chourmouzi D, Boulogianni G, Sofroniadis I. Imaging of primary bone tumors of the spine. Eur Radiol 2003; 13 (08) 1859-1871
• 13 Haupt R, Minkov M, Astigarraga I. et al; Euro Histio Network. Langerhans cell histiocytosis (LCH): guidelines for diagnosis, clinical work-up, and treatment for patients till the age of 18 years. Pediatr Blood Cancer 2013; 60 (02) 175-184
• 14 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
• 15 Rimondi E, Mavrogenis AF, Rossi G, Ussia G, Angelini A, Ruggieri P. CT-guided corticosteroid injection for solitary eosinophilic granuloma of the spine. Skeletal Radiol 2011; 40 (06) 757-764
• 16 Howarth DM, Gilchrist GS, Mullan BP, Wiseman GA, Edmonson JH, Schomberg PJ. Langerhans cell histiocytosis: diagnosis, natural history, management, and outcome. Cancer 1999; 85 (10) 2278-2290
• 17 Villas C, Martínez-Peric R, Barrios RH, Beguiristain JL. Eosinophilic granuloma of the spine with and without vertebra plana: long-term follow-up of six cases. J Spinal Disord 1993; 6 (03) 260-268
• 18 Leonard PS. Eosinophil Granuloma of the spine. 1981: 371-376
• 19 Yeom JS, Lee CK, Shin HY, Lee CS, Han CS, Chang H. Langerhans' cell histiocytosis of the spine. Analysis of twenty-three cases. Spine 1999; 24 (16) 1740-1749
• 20 Rodallec MH, Feydy A, Larousserie F. et al. Diagnostic imaging of solitary tumors of the spine: what to do and say. Radiographics 2008; 28 (04) 1019-1041
• 21 Ravindra VM, Eli IM, Schmidt MH, Brockmeyer DL. Primary osseous tumors of the pediatric spinal column: review of pathology and surgical decision making. Neurosurg Focus 2016; 41 (02) E3
• 22 Helms C. Fundamentos de Radiología del Esqueleto de Bolsillo. 3era.. Edición. Marban; 2006
• 23 Patnaik S, Jyotsnarani Y, Uppin SG, Susarla R. Imaging features of primary tumors of the spine: A pictorial essay. Indian J Radiol Imaging 2016; 26 (02) 279-289
• 24 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
• 25 Wiesner EL, Hillen TJ, Long J, Jennings JW. Percutaneous CT-guided biopsies of the cervical spine: Technique, histopathologic and microbiologic yield, and safety at a single academic institution. AJNR Am J Neuroradiol 2018; 39 (05) 981-985
• 26 Arenzana-Seisdedos F, Barbey S, Virelizier JL, Kornprobst M, Nezelof C. Histiocytosis X. Purified (T6+) cells from bone granuloma produce interleukin 1 and prostaglandin E2 in culture. J Clin Invest 1986; 77 (01) 326-329
• 27 Gonzalez-Crussi F, Hsueh W, Wiederhold MD. Prostaglandins in histiocytosis-X. PG synthesis by histiocytosis-X cells. Am J Clin Pathol 1981; 75 (02) 243-253
• 28 Capanna R, Springfield DS, Ruggieri P. et al. Direct cortisone injection in eosinophilic granuloma of bone: a preliminary report on 11 patients. J Pediatr Orthop 1985; 5 (03) 339-342
• 29 Egeler RM, Thompson Jr RC, Voûte PA, Nesbit Jr ME. Intralesional infiltration of corticosteroids in localized Langerhans' cell histiocytosis. J Pediatr Orthop 1992; 12 (06) 811-814
• 30 Raab P, Hohmann F, Kühl J, Krauspe R. Vertebral remodeling in eosinophilic granuloma of the spine. A long-term follow-up. Spine 1998; 23 (12) 1351-1354