A 50-year-old male patient was referred to our hospital, with a 4-month history of neck pain and gradually progressive weakness of all four limbs. Over the past 2 weeks, the patient noticed significant deterioration and became bed bound. The patient denies any previous history of trauma, malignancy, or medical history of note. Clinical examination revealed spastic quadriparesis of 3/5 with 80% decreased sensation of all modalities below C5. Sphincters were found to be intact.\nPlain X-ray cervical spine on anterior-posterior (AP) view showed a large lucent lesion replacing the C5 vertebra with loss of the left pedicle []. The magnetic resonance imaging (MRI) of the cervical spine demonstrated a T1 and T2 isointense, expansile mass lesion involving the C5 vertebral body extending into the left pedicle but sparing neural foramina. On T2, small hyperintense intralesional cavity was noted in the anterior part. The lesion extended across the C5/6 intervertebral disc and into the anteroinferior part of the C4 vertebra. Significant cord compression with absent thecal sac against C5 was noted. The lesion enhanced homogeneously with gadolinium and appeared completely extradural [Figure -]. Based on imaging, metastasis, myeloma, and tuberculous granuloma were suspected.\nThe patient underwent standard anterior cervical decompression and fixation. Peroperatively, tumor appeared as a well-encapsulated, firm, multinodular mass, yellow tan in color, 3 cm × 3 cm × 2 cm in size. There was a small necrotic part corresponding to the T2 hyperintensity. The lesion was excised in piecemeal fashion [] to completely decompress the cord. No clue of bone was found within the mass. At this point, differential was further narrowed to granulomatous lesion like tuberculosis. Histology revealed sheets of histiocytes with foamy cytoplasm and bland nuclei. Focal cholesterol clefts and multinucleated giant cells were also noticed. There was no cellular atypia. CD68 was positive and S100 was found to be negative [Figure and ].\nThe serum calcium was normal, and lipid profile showed normolipemia. Clinical examination did not reveal any soft-tissue xanthomas. The patient was able to return to his normal activities 4 weeks after the operation. Postoperative imaging as per routine following anterior decompression and fixation consisted of plain X-ray cervical spine AP and lateral views [Figure and ]. Postoperative computed tomography (CT) of the same area was planned at the 6-month follow-up both to see the integrity of fixation as well as to rule out the tumor recurrence, but the patient was not able to reach out for imaging because of pandemic restrictions. Currently, he is 1 year out of his surgery, with no reported complaint as per telephonic follow-up.
A 29-year-old male patient presented with right side weakness and left side numbness that occurred just after a car accident. Upon physical examination, there was no abnormal finding, except for tenderness in the upper cervical area. Routine laboratory tests demonstrated normal results. Motor weakness of right side extremity was disclosed as 1/5 for arm and 2/5 for leg with slightly hyperactivity of deep tendon reflex. Sensory examination showed significantly decreased pain, temperature, vibration, and discriminatory touch sensations below the left C2 dermatome. Babinski's reflex and Hoffman's sign were present on the left side. Based on the symptoms and neurological findings, the assumptive diagnosis of Brown-Sequard syndrome was made. The patient had no symptoms or past medical history related to a cervical spine lesion before the trauma. Cervical plain films revealed straightening of the cervical spine with loss of normal lordosis and widening between the posterior arch of the atlas and spinous process of the axis. A computed tomography (CT) scan of the upper cervical spine revealed a large round mass which measured 25×18×24 mm with relatively homogeneous strong enhancement and was located between the C1 and C2 interspinous space. In addition, the CT scan showed cortical osseous erosions at the lower C1 posterior arc and upper C2 spinous process. Cervical magnetic resonance images (MRI) revealed an epidural dumbbell-shaped mass traversing the C1 to C2 interspinous space, compressing the surrounding structures (). The spinal cord was compressed by the lesion which was isointense on T1-weighted image (WI) and mixed hypointense on T2WI with homogenous strong enhanced after gadolinium administration. Radiologic analysis indicated possible lymphoma, meningioma, schwannoma or other neuronal tumors.\nThe patient underwent a surgery immediately for decompression of the spinal cord. A total tumor resection was performed with only a partial laminectomy from the lower half of the C1 to upper half of C2 without any instrumentation and access to the tumor. After partial lamicectomy, an extradural and yellowish mass was exposed with well-demarcated from the surrounding structures but adhesive at the epidural area. Under microscopy, the tumor was delicately dissected, and en bloc removal was achieved (). The mass was ovoid, soft like gelatin, avascular, and well-encapsulated, measuring 25 mm at the largest diameter, and was adherented to the outer layer of the dura mater. And, there was no root invasion. On the basis of the clinical and operative features, we thought that the lesion originated from the outer aspect of the dura mater. Upon sectioning, the mass was particularly yellow and white with cystic components ().\nAfter surgery, the patient's right hemiparesis fully recovered in 2 weeks. However, a slightly tingling numbness on his left arm remained and was controlled through medication in the outpatient clinic for 4 months. Currently, the patient has no symptoms without instability of cervical spine. Cervical MRI with gadolinium enhancement at 2 years after surgery demonstrated no recurrent lesion ().\nHistological examination of the tumor uncovered focal aggregation cells with ample, clear, and foamy cytoplasm in a background of loose, fibrous stroma of xanthic appearance. Large, round cells had irregular vesicular nuclei which were regular and small with condensed chromatin (). The specimen was underwent to immunohistochemical testing, yielding strongly positive for CD 68 and lysozyme stain, which confirmed that the large foam cells were histiocytes. The test with CD-1a was negative, thus ruling out Langerhans' cell histiocytosis. GFAP stain and S-100 protein were negative. Thus, a diagnosis of xanthogranuloma was established.