Skip to content

Advertisement

  • Case report
  • Open Access

Conservative treatment for osteoid osteoma of the odontoid process of the axis: a case report

  • 1,
  • 1,
  • 1,
  • 1,
  • 1 and
  • 1Email author
World Journal of Surgical Oncology201412:305

https://doi.org/10.1186/1477-7819-12-305

  • Received: 15 February 2014
  • Accepted: 25 September 2014
  • Published:

Abstract

Background

Osteoid osteoma is a primary benign bone lesion, which constitutes about 10% of all primary benign bone tumors and 3% of all primary bone tumors. The spine is involved in 10% of the cases, and the lumbar spine is the most commonly affected whereas the tumor is rarely seen in the cervical spine. With regard to the osteoid osteoma being located at the odontoid process of the axis, limited cases have been reported in the literature.

Case presentation

An osteoid osteoma of the odontoid process of the axis was diagnosed by computed tomography in an 18-year-old male patient with a 3-month history of pain. The patient’s parents refused surgery for fear of surgical risks and high expense. Considering the benign nature of osteoid osteoma, we prescribed celecoxib 200 mg per day to the patient. With the treatment, the patient’s pain was alleviated gradually and the range of motion of the cervical spine also recovered to normal. At the two-year phone follow-up, the patient was free of symptoms.

Conclusions

For this kind of benign tumor, conservative treatment plus close follow-up is applicable whereas surgery bears significant risks and a heavy economic burden.

Keywords

  • osteoid osteoma
  • axis
  • cervical
  • spine

Background

Osteoid osteoma, a primary benign bone lesion, was first defined by Jaffe in 1935 [1]. It pathologically features a highly vascularized nidus of connective tissue surrounded by sclerotic bone [2, 3]. The nidus measures about 10 mm in diameter, and the size is the main distinguishing feature between it and osteoblastoma [4]. Osteoid osteoma constitutes about 10% of all primary benign bone tumors and 3% of all primary bone tumors [5]. Most of the cases occur in the first three decades and occurs two to three times more frequently in men than in women [6]. Predilection sites of the osteoid osteoma are long bones, especially those of the lower extremities. The spine is involved in 10% of the cases, and the lumbar spine is the most commonly affected whereas the tumor is rarely seen in cervical spine [7]. With regard to the osteoid osteoma being located at the odontoid process of the axis, limited cases have been reported in the literature [8, 9]. Herein, we report an osteoid osteoma of the dens axis confirmed by radiographic examinations.

Case presentation

An 18-year-old male patient had a history of neck pain of 3 months’ duration, which was more severe at night. A physical examination revealed a remarkably reduced rotation to the right and mild kyphosis. A lateral cervical spine radiograph showed kyphosis of the cervical spine (Figure 1). A computed tomography (CT) scan revealed a lytic area involving the odontoid process of the axis with the partial ossification of the matrix and a sclerotic margin (Figure 2). By CT three-dimensional (3-D) reconstruction, the nidus was found at the conjunction of the odontoid process of the axis and the body of the axis (Figure 3).
Figure 1
Figure 1

Lateral X-ray showing kyphosis of cervical spine.

Figure 2
Figure 2

Transversal computed tomography (CT) images showing the nidus with partial ossification of matrix and sclerotic margin, and sagittal CT images of the nidus.

Figure 3
Figure 3

Computed tomography (CT) of three-dimensional (3-D) reconstruction of the nidus.

From the patient’s medical history and radiological and physical examination, we made a diagnosis of osteoid osteoma of the odontoid process of the axis. The patient’s parents refused surgery for fear of surgical risks and high expense. Considering the benign nature of osteoid osteoma, we prescribed celecoxib 200 mg per day to the patient. With the treatment, the patient’s pain was alleviated gradually, and the range of motion of the cervical spine also recovered to normal. At the two-year phone follow-up, the patient was free of symptoms. However, he refused to go to our outpatient center for radiographic examinations.

Discussion

Osteoid osteoma is a benign tumor, for which the chief symptom is pain whereas other clinical symptoms are related to its location [10]. The pain is localized and may be aggravated with motion. At the same time, pain can also be alleviated with activity [11]. Localized pain is caused by the nerve fibers in the nidus. The production of prostaglandin may lead to an increase in vascular pressure, which may produce pain by stimulating afferent nerves around the nidus [12]. The pain is more severe at night and is relieved by non-steroidal anti-inflammatory drugs (NSAIDs) [13]. This feature could also be used as a diagnostic clue. Scoliosis is reported in 70% of the cases when the lesion involves the spine, and it is the most common cause of painful scoliosis in adolescents, especially when the lesion is located in lumbar or thoracic spine [1416].

CT is recommended as the best diagnostic tool to define and localize the nidus, particularly when the nidus is located at the spine [13]. With CT scanning, we can optimally visualize the nidus with perifocal marginal sclerosis [17]. Conventional radiography is insufficient to visualize the lesion because of the complicated anatomy of the spine [17]. Magnetic resonance imaging (MRI) is not as accurate as CT in demonstrating the nidus because the nidus presents different signal intensity in different patients [1820]. The increased signal intensity of the lesion on T2-weighted images or on enhanced T1- weighted images was pathologically correlated with the degree of vascularity of the fibrovascular nidal stroma and the amount of osteoid substance within the nidus [21]. On the other hand, for detecting changes in soft tissue and bone marrow around the nidus, MRI is more sensitive than CT. These changes are due to bone marrow inflammation and edema [18, 21]. In addition to CT and MRI, bone scintigraphy is also a sensitive diagnostic test.

Tumors that affect the axis vertebra are numerous and include osteoblastoma, eosinophilic granuloma, chondroma, paraganglioma, plasmacytoma, multiple myeloma and so on [22]. Osteoblastoma is the major differential diagnosis of osteoid osteoma because they have the same pathological features but distinct natures. In contrast to osteoid osteoma, osteoblastoma is more aggressive, often extending to extraskeletal soft tissues. Moreover, it often recurs and even metastasizes after surgery [2325]. In pathology, the two tumors are both lesions of osteoblastic origin [7, 26]. The most significant difference between them is the size of the nidus. A lesion is diagnosed as osteoid osteoma when its diameter is less than 15 mm and as osteoblastoma when larger [27].

Surgeons often hold progressive attitudes toward the treatment of osteoid osteoma. For example, en block excision is frequently recommended [28, 29]. In addition, minimally invasive methods, such as CT-guided thermocoagulation and percutaneous radiofrequecy ablation have also obtained satisfactory outcomes [3032]. When the tumor is combined with scoliosis, surgical excision with or without correction surgery is widely adopted. However, surgical treatment is by no means the only choice, and surgeons should weigh costs against benefits before a decision is made. In the present case, conservative treatment was administered in consideration of economic issues and surgical risks. In developing countries, the coverage of medical insurance is inadequate, and many patients cannot afford surgical expenses. For this kind of benign tumor, conservative treatment plus close follow-up is applicable. Moreover, the risky anatomic location of the lesion and the high morbidity further prompted us to choose medication, with a satisfactory outcome [13, 33].

Conclusions

For this kind of benign tumor, conservative treatment plus close follow-up is applicable whereas surgery bears significant risks and a heavy economic burden.

Consent

Acknowledgements

This work was supported by National Key Clinical Department Project and Talents Programme of Jiangsu (Grant No. WSW-005). We thank MS Zhang Linlin for her contribution to this article.

Abbreviations

CT: 

computed tomography

MRI: 

magnetic resonance imaging

NSAIDs: 

non-steroidal anti-inflammatory drugs.

Declarations

Authors’ Affiliations

(1)
Spine Surgery, Drum Tower Hospital, Nanjing University Medical School, 321 Zhongshan Road, Nanjing, 210008, China

References

  1. Jaffe HL: Osteoid osteoma: a benign osteoblastic tumor composed of osteoid and atypical bone. Arch Surg. 1935, 31: 709-715. 10.1001/archsurg.1935.01180170034003.View ArticleGoogle Scholar
  2. Kirman EO, Hutton PA, Pozo JL, Ransford AO: Osteoid-osteoma and benign osteoblastoma of the spine. J Bone Joint Surg (Br). 1984, 66: 21-26.Google Scholar
  3. Pettine KA, Klassen RA: Osteoid-osteoma and osteoblastoma of the spine. J Bone Joint Surg Am. 1986, 68: 354-360.PubMedGoogle Scholar
  4. Jaffe HL: Benign osteoblastoma. Bull Hosp Joint Dis. 1956, 17: 141-151.PubMedGoogle Scholar
  5. Hurtgen KL, Buehler M, Santolin SM: Osteoid osteoma of the vertebral body with extension across the intervertebral disc. J Manipulative Physiol Ther. 1996, 19: 118-122.PubMedGoogle Scholar
  6. Resnick D, Kyriakos M, Greenway G: Tumors and tumor-like lesions of bone: Imaging and pathology of specific lesions. Diagosis of Bone and Joint Disorders. Edited by: Resnick D. 1995, Philadelphia, PA: Saunders, 3628-3657. 3Google Scholar
  7. Zilieli M, Cagli S, Basdemir G, Ersahin Y: Osteoid osteomas and osteoblastomas of the spine. Neurosurg Focus. 2003, 15: E5-Google Scholar
  8. Al-Balas H, Omaeri H, Mustafa Z, Matalka I: Osteoid osteoma of the odontiod process of the axis associated with atlanto-axial fusion. Br J Radiol. 2009, 82: e126-e128. 10.1259/bjr/55010187.View ArticlePubMedGoogle Scholar
  9. Neumann D, Dorn U: Osteoid osteoma of the dens axis. Eur Spine J. 2007, 16: 271-274. 10.1007/s00586-007-0332-z.PubMed CentralView ArticlePubMedGoogle Scholar
  10. Hermann G, Abdelwahab IF, Casden A, Mosesson R, Klein MJ: Osteoid osteoma of a cervical vertebral body. Br J Radiol. 1999, 72: 1120-1123. 10.1259/bjr.72.863.10700833.View ArticlePubMedGoogle Scholar
  11. Greenspan A: Benign bone forming lesions: osteoma, osteoid osteoma, osteoblastoma. Clinical imaging, pathologic and differential considerations. Skel Radiol. 1993, 22: 485-500.Google Scholar
  12. Hasegaga T, Hirose T, Sakamoto R, Seki K, Ikata T, Hizawa K: Mechannism of pain in osteoid ostomas: an immunohistochemical study. Histopathology. 1993, 22: 487-491. 10.1111/j.1365-2559.1993.tb00163.x.View ArticleGoogle Scholar
  13. Kneisl JS, Simon MA: Medical management compared with operative treatment for osteoid osteoma. J Bone Joint Surg Am. 1992, 74: 179-185.PubMedGoogle Scholar
  14. Maiuri F, Signoreli C, Lavano A, Gambardella A, Simari R, D'Andrea F: Osteoid osteoma of the spine. Surg Neurol. 1986, 25: 375-380. 10.1016/0090-3019(86)90214-4.View ArticlePubMedGoogle Scholar
  15. Metha MH: Pain provoked scoliosis: observations on the evolution of the deformity. Clin Orthop. 1978, 135: 58-65.Google Scholar
  16. Saifuddin A, White J, Sherazi Z, Shaikh MI, Natali C, Ransford AO: Osteoid osteoma and osteoblastoma of the spine: Factors associated with the presence of scoliosis. Spine. 1998, 23: 47-53. 10.1097/00007632-199801010-00010.View ArticlePubMedGoogle Scholar
  17. Gamba JL, Martinez S, Apple J, Harrelson JM, Nunley JA: Computed tomography of axial skeletal osteiod osteoma. AJR. 1984, 142: 769-772. 10.2214/ajr.142.4.769.View ArticlePubMedGoogle Scholar
  18. Kransdorf MJ, Stull MA, Gilkey FE, Moser RP: Osteoid osteoma. RadioGraphic. 1991, 11: 671-696. 10.1148/radiographics.11.4.1887121.View ArticleGoogle Scholar
  19. Houang B, Grenier N, Greselle JF: Osteoid osteoma of the cervical spine: misleading MR features about a case involving the uncinate process. Neuroradiology. 1990, 31: 541-551. 10.1007/BF00340138.View ArticleGoogle Scholar
  20. Woods ER, Martel W, Mandell SH, Crabbe JP: Reactive soft-tissue mass associated with osteoid osteoma : correlation of MR imaging features with pathologic findings. Radiology. 1993, 186: 221-225. 10.1148/radiology.186.1.8416568.View ArticlePubMedGoogle Scholar
  21. Assoun J, Richardi G, Railhac JJ, Crabbe JP: Osteoid osteoma: MR imaging versus CT. Radiology. 1994, 191: 217-223. 10.1148/radiology.191.1.8134575.View ArticlePubMedGoogle Scholar
  22. Piper JG, Menezes AH: Management strategies for tumors of the axis vertebra. J Neurosurg. 1996, 84: 543-551. 10.3171/jns.1996.84.4.0543.View ArticlePubMedGoogle Scholar
  23. Bruneau M, Cornelius JF, George B: Osteoid osteomas and osteoblastomas of the occipitocervical junction. Spine. 2005, 30: 567-571. 10.1097/01.brs.0000154690.04674.06.View ArticleGoogle Scholar
  24. Pieterse AS, Vernon-Roberts B, Paterson DC, Cornish BL, Lewis PR: Osteoid osteoma transforming to aggressive (low grade malignant) osteoblastoma: a case report and literature review. Histopathology. 1983, 7: 789-800. 10.1111/j.1365-2559.1983.tb02291.x.View ArticlePubMedGoogle Scholar
  25. Ozaki T, Liljenqvist U, Hillmann A, Halm H, Lindner N, Gosheger G, Winkelmann W: Osteoid osteoma and osteoblastoma of the spine: experiences with 22 patients. Clin Orthop. 2002, 397: 394-402.View ArticlePubMedGoogle Scholar
  26. De Praeter MP, Dua GF, Seynaeve PC, Vermeersch DG, Klaes RL: Occipital pain in osteoid osteoma of the atlas. A report of two cases. Spine. 1999, 24: 912-914. 10.1097/00007632-199905010-00014.View ArticlePubMedGoogle Scholar
  27. Nemoto O, Moser RP, Van Dam BE, Vermeersch DG, Klaes RL: Osteoblastoma of the spine. A review of 75 cases. Spine. 1990, 15: 1271-1280.View ArticleGoogle Scholar
  28. Azouz EM, Kozlowski K, Marton D, Sprague P, Zerhouni A, Asselah F: Osteoid osteoma and osteoblastoma of the spine in children: Report of 22 cases with brief literature review. Pediatr Radiol. 1986, 16: 25-31. 10.1007/BF02387501.View ArticlePubMedGoogle Scholar
  29. Raskas DS, Graziano GP, Heidelberger KP, Heidelberger KP, Hensinger RN: Osteoid and osteoma of the spine. J Spinal Disord. 1992, 5: 204-211. 10.1097/00002517-199206000-00010.View ArticlePubMedGoogle Scholar
  30. Cove JA, Taminiau AH, Obermann WR, Vanderschueren GM: Osteoid osteoma of the spine treanted with percutaneous computed tomography-guided thermocoagulation. Spine. 2000, 25: 1283-1286. 10.1097/00007632-200005150-00014.View ArticlePubMedGoogle Scholar
  31. Rosenthal DI, Springfiled DS, Gebhardt MC, Rosenberg AE, Mankin HJ: Osteoid osteoma: percutaneous radio-frequency ablation. Radiology. 1995, 197: 451-454. 10.1148/radiology.197.2.7480692.View ArticlePubMedGoogle Scholar
  32. Vanderschueren GM, Taminiau AH, Obermann WR, Bloem JL: Osteoid osteoma: clinical results with thermocoagulation. Radiology. 2002, 224: 82-86. 10.1148/radiol.2241011135.View ArticlePubMedGoogle Scholar
  33. Ilays I, Younge DA: Medical management of osteoid ostoma. Can J Surg. 2002, 45: 435-437.Google Scholar

Copyright

Advertisement