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Treatment of osteoblastoma at C3-4 in a child: a case report
© Feng et al.; licensee BioMed Central Ltd. 2014
Received: 23 March 2014
Accepted: 24 September 2014
Published: 26 September 2014
Osteoblastoma is a rare and benign osteoid-producing primary bone tumor that affects mainly the long bones. 36% of these tumors are observed around the spine and the vast majority arises around the posterior.
This report describes a case of C3-4 osteoblastoma occurring in a 5-year-and-8-month-old Han Chinese child. The pathophysiology of symptom development, evaluations, and management are presented. Because of the close proximity of the osteoblastoma to the vertebral artery canal, the artery suffered a minor laceration intraoperatively. Hemostatic gelatin sponges were used to compress the bleeding site instantly and a tricortical iliac crest fixed with a screw was also used to add pressure to the gelatin sponges. Fusion on the other side was also used to stabilize the spine. To the best of our knowledge, this is the first report of a case of osteoblastoma at C3-4 with artery injury intraoperatively.
This case delineates the difficulties in diagnosing this tumor, the challenges and problems encountered during its surgical management, and the favorable prognosis after adequate treatment.
Osteoblastoma is a rare and benign osteoid-producing primary bone tumor that affects mainly the long bones. Thirty-six percent of these tumors are observed around the spine and the vast majority arises around the posterior [1, 2]. The mean age at presentation was 20.4 years, with a range of 6 months to 75 years . Several cases of osteoblastoma of the cervical vertebrae have been reported and different aspects of their management have been discussed [4–7]. However, cases occurring at C3 or C4 are rare. This report describes a case of C3-4 osteoblastoma occurring in a 5-year-and-8-month-old child. The pathophysiology of symptom development, evaluations, and management are also presented.
A Han Chinese boy aged 5 years and 8 months was admitted to the hospital for evaluation of neck and right shoulder and arm pain persisting for more than 6 months. The intensity of the pain had gradually increased during the course of the illness. However, there was no pain during sleep.
A diagnostic delay is common in cases of cervical osteoblastoma. Symptoms of patients afflicted with osteoblastoma of the spine usually include dull, localized, and gradually increasing pain. However, these symptoms were non-specific and usually neglected by physicians, which may have been the cause of the 6-month diagnosis delay in the present case. The patient was eventually diagnosed correctly because of his severe neurological deficit and was managed surgically. Neurologic deficits are occasionally reported in the literature. In our case, the patient presented with muscle weakness, sensory disturbance, and hyperreflexia.
In some cases, the vertebral artery canal was eroded by the osteoblastoma, and the use of preoperative angiography and embolization was useful to reduce intraoperative blood loss . However, preoperative angiography and embolization were not performed in our case because of economic limitations of the patient’s family. However, vascular complications during the operation in our case indicate the necessity of preoperative embolization.
There should be always an attempt to achieve diagnostics by biopsy before surgery, which has become a necessary prelude to successful management of suspected spinal tumors. In this case, the differential diagnosis with osteosarcoma and osteoma osteoid is quite unlikely before operation. In addition, biopsy is generally preferred for a variety of reasons, including lower morbidity, cost effectiveness and shorter procedure time and patient length of stay. However, we didn’t perform pre-operative biopsy in this case for the following reasons: (1) According to the literature, sclerotic lesions showed a low diagnostic accuracy rate by biopsy . Specimens from sclerotic lesions are composed chiefly of eburnated bone containing only tiny areas of bone marrow with a few cell clusters; (2) Hemorrhage may occur during biopsy because of rich tumor vascularization; (3) The use of good quality radiological exams can fulfill a criterion for that particular histopathology. As the CT and MRI image was so suggestive of osteoblatoma, the biopsy was avoided.
This tumor was classified as benign lesion, locally aggressive, the treatment of which is ideally performed by a marginal resection. However, the surgical resection was planned to be intra-lesional resection because of the proximity of the noble anatomic structures.
On the 12-month follow-up CT, a considerable narrowing of the vertebral canal was observed, which was probably caused by insufficient tumor removal. Although a complete excision of the lesion was not achieved because of vertebral artery injury, the gross and microscopic tumor, as well as a circumferential margin of normal bony tissue was removed. At the 12-month follow-up, the patient presented without neurologic deficit, and disease progression or recurrence was not observed because of the insufficient removal.
Blindness after spinal surgery has been described as a rare but serious, irreversible, and incurable complication. Previously reports have described the delayed onset of cortical blindness secondary to vertebral artery injury following cervical spine trauma . The visual dysfunction was probably caused by embolic cortical infarction superimposed on the vertebral artery dissection. However, the patient in our case is asymptomatic, and the eye examination was documented as normal. The possible explanation for the different outcome in our case may be the presence of collateral blood supply from the contralateral vertebral artery and circle of Willis.
Whether spinal fusion is necessary after tumor resection remains controversial. Fusion for cervical stabilization substantially decreases the mobility of the cervical spine and predisposes the discs above and below the fusion to degeneration. However, stabilization of the cervical spine is critical to prevent progressive kyphotic deformity. Several previous reports have described kyphotic deformity following intralesional excision of osteoblastoma [11–13]. Children and young adults (25 years or younger) are considered at higher risk of postlaminectomy kyphosis than older adults because of the skeletal immaturity and continued growth characteristic of those stages. A long-term follow-up is needed for our case to determine whether tumor recurrence or spinal deformity will occur after the surgical reconstruction.
In conclusion, this case delineates the difficulties in diagnosing this tumor, the challenges and issues encountered during the surgical management, and also the favorable prognosis after adequate treatment.
Written informed consent was obtained from the parents of the young patient for the publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal. The human subjects Institutional Review Board of Sichuan University approved this study prospectively.
The authors would like to acknowledge the financial support from the National Natural Science Foundation of China (Grant No: 81201430). The authors also acknowledge Melanie Gupte for her assistance with manuscript preparation.
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