- Email: [email protected]
Intradural Extramedullary Capillary Hemangioma in the Upper Cervical Spine: First Report
Case Report
Intradural Extramedullary Capillary Hemangioma in the Upper Cervical Spine: First Report Sofiene Bouali, Nidhal Maatar, Asma Bouhoula, Khansa Abderrahmen, Jalel Kallel, Hafedh Jemel
Key words Capillary hemangioma - Cervical spine - Extramedullary - Intradural -
Abbreviations and Acronyms MRI: Magnetic resonance imaging Department of Neurosurgery, National Institute of Neurology, Tunis, Tunisia To whom correspondence should be addressed: Sofiene Bouali, M.D. [E-mail: [email protected]] Citation: World Neurosurg. (2016). http://dx.doi.org/10.1016/j.wneu.2016.04.131 Journal homepage: www.WORLDNEUROSURGERY.org
- BACKGROUND:
The occurrence of intradural extramedullary capillary hemangiomas is exceedingly rare. To date, only 39 cases of intradural extramedullary capillary hemangiomas have been reported in the English literature, and all of these cases have been described at the lumbar and thoracic spinal levels. To our knowledge, this report is the first case of capillary hemangiomas of the cervical spine in the literature. In general, this entity is misdiagnosed preoperatively as a neoplasm.
- CASE
DESCRIPTION: A 29-year-old man presented with neck pain and progressive gait disturbance, and was diagnosed with an intradural extramedullary capillary hemangioma in the cervical region.
- CONCLUSIONS:
Although rare, our case demonstrates that capillary hemangioma should be considered in the differential diagnosis of intradural extramedullary tumor of the cervical spine.
Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2016 Elsevier Inc. All rights reserved.
findings, and treatment and outcomes associated with such lesions.
INTRODUCTION Capillary hemangiomas are benign endothelial cell neoplasms, characterized by nodules of capillary-sized vessels lined by flattened endothelium.1 They are most commonly found in the skin or the mucosa of the head and neck.2 Intradural extramedullary capillary hemangioma is extremely rare.1,3,4 To date, only 39 cases of capillary hemangioma affecting the intradural extramedullary spine have been documented in the literature. The lesions were observed in the lumbar spine in 60% of the cases and in the thoracic region in the remaining cases. No previous reports have described capillary hemangiomas located in the cervical spine. Here we report a patient with an intradural extramedullary capillary hemangioma of the upper cervical spine, the first such reported case in the literature. We also review the literature related to intradural extramedullary capillary hemangiomas, and discuss the clinical manifestations, imaging and histopathological
CASE REPORT History and Presentation A 29-year-old right-handed male patient complaining of posterior neck pain for 2 years was admitted to the hospital. He had a 1-year history of numbness in the left distal upper extremity, a 6-month history of right sided paresthesia, and a 3-month history of gradually progressive gait disturbance. He reported feeling slight weakness in his right leg when walking more than a couple of blocks.
Physical Examination Findings Neurologic examination revealed diffuse hyperreflexia, more pronounced on the right side. Strength was symmetric and full bilaterally. Decreased sensation of the right hemibody to light touch and pinprick below the C5 dermatome level was noted. Other general and systemic assessments revealed no abnormalities.
WORLD NEUROSURGERY -: ---, MONTH 2016
Radiologic Findings An unenhanced computed tomography scan at the C1 level revealed a poorly circumscribed mass of low density within the spinal canal, with no bony erosion (Figure 1). Magnetic resonance imaging (MRI) revealed an intradural extramedullary dumbbell-shaped, lobulecontoured mass at the C1 level. The lesion was isointense relative to the spinal cord on T1-weighted MRI and slightly hyperintense on T2-weighted MRI. The tumor showed strong homogeneous enhancement on contrast-enhanced T1-weighted MRI. The tumor exerted a mass effect on the cervical spinal cord, displacing it to the right with compression (Figure 2A). Initial examination revealed a dilated serpentine venous structure coursing toward the left anterior surface of the lesion (Figure 2B). Surgical Findings Based on the foregoing investigations, our preoperative diagnosis was a neurogenic tumor or meningioma. The patient underwent C1 hemilaminectomy. Under general anesthesia, he was placed prone in pin fixation. A midline skin incision was made from the inion to the spinous
www.WORLDNEUROSURGERY.org
1.E1
CASE REPORT SOFIENE BOUALI ET AL.
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
Figure 1. Unenhanced cervical scan revealing a poorly circumscribed mass of low density within the spinal canal at the C1 level, with no bony erosions. (A) Coronal view. (B) Sagittal view. (C and D) Axial view.
process of C4. The unilateral occipital bone and neural arches of C1 and C2 were exposed on the left side only. Under microscopic magnification, a unilateral limited laminectomy was performed on the left-side lamina of C1 with a Kerrison punch. The dura was then opened paramedially, revealing an intradural wellcircumscribed, reddish mass on the ventrolateral side of the cervical spine. The tumor was adherent to the arachnoid and C1 nerve root, especially on the left side. Careful separation of the C1 ventral root and sectioning of the fibers entering the mass revealed a dilated serpentine vein on the left anterior surface of the lesion. This vein was coagulated, the tumor was completely resected without significant bleeding, and tThe dura was closed. The
1.E2
www.SCIENCEDIRECT.com
patient’s neck pain resolved over the course of the first postoperative week, and hypoesthesia resolved in the first postoperative month. Histopathological Findings The histopathological examination revealed typical features of capillary hemangioma, including innumerable thinwalled and irregular capillary vessels lined with endothelium (Figure 3). DISCUSSION Capillary hemangiomas are congenital hamartomatous malformations that result from proliferations of vascular endothelial cells.5,6 They are most commonly found in the skin or mucosa of the head and neck.6
Intradural extramedullary capillary hemangiomas were first described by Hanakita in 1991.7 To date, 39 cases of capillary hemangioma affecting the intradural extramedullary spine have been reported in the literature (Table 1). In the spinal intradural extramedullary space, capillary hemangiomas may arise from the blood vessels of the nerve roots in the cauda equina, the pial surface of the spinal cord, or the inner surface of the dura. Two hypotheses have been proposed regarding the pathogenesis of the disease. The lesion may evolve during early somite differentiation, at the time of angioblastic differentiation (days 21e24 of embryogenesis), owing to impaired movement and differentiation of primitive mesoderm from the embryonic mesodermal plate. Another school of thought identifies the origin as vascular structures within the epineurium of the nerve roots affected during individual ontogenesis.3,16 In the 39 cases reported in the literature, the mean patient age was 50 years (range, 20e74 years), with a male predominance (28 males and 11 females, a ratio of almost 3:1) (Table 1). Pure intradural extramedullary capillary hemangiomas frequently occurred distantly, in the lumbar region (at the conus medullaris or attached to nerve roots of the cauda equine) in 23 cases, and in the thoracic spine in 16 cases (Table 1). There have been no previous reports describing a capillary hemangioma located in the cervical region. Pain was the most common complaint and the initial reported symptom in 36 of the 39 patients (95% of cases). Back pain was often accompanied by radicular pain, especially when the tumor was present in the lumbar spine. Capillary hemangiomas infrequently produce severe neurologic deficits at diagnosis owing to their discrete and noninfiltrative nature.15 Moderate motor deficits were described in 19 cases,9,10 and sphincter dysfunction was present in only 2 cases.16,25 In our patient, preoperative symptoms included neck pain, progressive gait disturbance, and sensory symptoms, including hypoesthesia in the right hemibody. In addition, sensory disturbance and increased deep tendon reflexes were noted in the contralateral extremities. With this
WORLD NEUROSURGERY, http://dx.doi.org/10.1016/j.wneu.2016.04.131
CASE REPORT SOFIENE BOUALI ET AL.
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
Figure 2. (A) MRI of the cervical spine. Sagittal (a) and axial (b) T1-weighted images showing a round, well-circumscribed intradural extramedullary lesion at the level of C1 isointensity relative to the spinal cord, hyperintense T2-weighted sagittal (c) and axial (d) images. The mass enhances homogeneously on sagittal (e) and axial (f) postcontrast T1-weighted sequence and compressing the spinal cord laterally. A vascular-like structure is apparent anteriorly to the mass, indicating enlarged vessels. (B) Preoperative MRI of the cervical spine. (a and b) A dilated serpentine venous structure is seen coursing toward the left anterior surface of the lesion, shown on sagittal T1-weighted (a) and T2-weighted (b) images. (c) Signal void areas (arrowhead) on precocious sagittal postcontrast images indicate draining veins.
WORLD NEUROSURGERY -: ---, MONTH 2016
slow-growing tumor in the intradural space at the C1 level, contralateral cord involvement may be induced by counterpressure on the spinal cord in contact with the contralateral lamina, which may be generated by cord deviation and by frequent neck rotation.29,30 Most capillary hemangiomas are radiologically misdiagnosed and/or mistaken for meningiomas or schwannomas.31 Our patient’s MRI findings are consistent with those in previous reports; the lesion appeared isointense relative to the spinal cord on T1-weighted MRI and isointense or hyperintense on T2-weighted MRI with strong homogeneous gadolinium enhancement.23 In this case, the presence of dilated serpentine venous structures around the lesion shown on T2-weighted MRI, similar to that seen in arteriovenous malformation, were not appreciated before surgery. Our review of the literature describing capillary hemanagiomas of the peripheral or central nervous system found the presence of enlarged draining veins reported in only 3 previous cases.10,21,32 We believe that this subject deserves further attention because it helps identify specific radiologic signs of capillary hemangiomas. All reported cases presented with a pure intradural extramedullary capillary hemangioma, with the mass extending to a maximum of 2 vertebral segments. Histologically, capillary hemangioma are thin, irregular, capillary-sized vessels caught in low-attenuating fibroses with lobular architecture and a lining of a continuous basal lamina. These lesions exhibit low mitotic activity, positive reactions for CD31 and CD34, and negative reactions for S100 and epithelial membrane antigen on histopathological examination.33 Surgery is the treatment of choice for spinal intradural capillary hemangioma. In the literature reports, all patients were initially managed by local excision, and 97% of intradural capillary hemangiomas were completely resected with good outcomes. Only one case reported recurrence by 6 months after gross total resection.13 Preoperative embolization may aid in the surgical resection of these vascular lesions.21
www.WORLDNEUROSURGERY.org
1.E3
CASE REPORT SOFIENE BOUALI ET AL.
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
21. Abdullah DC, Raghuram K, Philips CD, Jane JA Jr, Miller B. Thoracic intradural extramedullary capillary hemangioma. AJNR Am J Neuroradiol. 2004;25:1294-1296. 22. Andaluz N, Balko MG, Stanek J, Morgan C, Schwetschenau PR. Lobular capillary hemangioma of the spinal cord: case report and review of the literature. J Neurooncol. 2002;56:261-264. 23. Choi BY, Chang KH, Choe G, Han MH, Park SW, Yu IK, et al. Spinal intradural extramedullary capillary hemangioma: MR imaging findings. AJNR Am J Neuroradiol. 2001;22:799-802.
Figure 3. Histology revealing typical features of capillary hemangioma. (A) Lobular architecture with numerous capillary-sized vessels lined by a single layer of endothelial cells and dissemination of dilated vessels (hematoxylin and eosin; original magnification 5). (B) Larger-caliber and medium-sized capillary vessels lined by flattened endothelium (hematoxylin and eosin; original magnification 20).
CONCLUSION This case demonstrates that capillary hemangioma, although rare, should be considered in the differential diagnosis of intradural extramedullary tumor of the cervical spine. REFERENCES 1. Enzinger FM, Weiss SW. Benign tumors and tumor-like lesions of blood vessels. In: Enzinger FM, Weiss SW, eds. Soft tissue tumors. 3rd ed. St. Louis, MO: Mosby; 1995:579-626. 2. Shin JH, Lee HK, Jeon SR, Park SH. Spinal intradural capillary hemangioma: MR findings. AJNR Am J Neuroradiol. 2000;21:954-956.
10. Takata Y, Sakai T, Higashino K, Goda Y, Tezuka F, Sairyo K. Intradural extramedullary capillary hemangioma in the upper thoracic spine: a review of the literature. Case Rep Orthop. 2014; 2014:604131. 11. Babu R, Owens TR, Karikari IO, Moreno J, Cummings TJ, Gottfried ON, et al. Spinal cavernous and capillary hemangiomas in adults. Spine (Phila Pa 1976). 2013;38:E423-E430. 12. Sonawane DV, Jagtap SA, Mathesul AA. Intradural extramedullary capillary hemangioma of lower thoracic spinal cord. Indian J Orthop. 2012;46:475-478. 13. Kaneko Y, Yamabe K, Abe M. Rapid regrowth of a capillary hemangioma of the thoracic spinal cord. Neurol Med Chir (Tokyo). 2012;52:665-669.
3. Nowak DA, Widenka DC. Spinal intradural capillary haemangioma: a review. Eur Spine J. 2001;10: 464-472.
14. Funayama T, Sakane M, Murai S, Ochiai N. Multiple capillary hemangiomas of the cauda equina at a level of a single vertebra. J Orthop Sci. 2010;15: 598-602.
4. Liu JJ, Lee DJ, Jin LW, Kim KD. Intradural extramedullary capillary hemangioma of the cauda equina: case report and literature review. Surg Neurol Int. 2015;6(Suppl 3):S127-S131.
15. Chung SK, Nam TK, Park SW, Hwang SN. Capillary hemangioma of the thoracic spinal cord. J Korean Neurosurg Soc. 2010;48:272-275.
5. Murphey MD, Fairbairn KJ, Parman LM, Baxter KG, Parsa MB, Smith WS. From the archives of the AFIP. Musculoskeletal angiomatous lesions: radiologic-pathologic correlation. Radiographics. 1995;15:893-917. 6. Feider HK, Yuille DL. An epidural cavernous hemangioma of the spine. AJNR Am J Neuroradiol. 1991;12:243-244. 7. Hanakita J, Suwa H, Nagayasu S, Suzuki H. Capillary hemangioma in the cauda equina: neuroradiological findings. Neuroradiology. 1991;33:458-461. 8. Alobaid A, Bennardo MR, Cenic A, Lach B. Mixed capillary-cavernous extramedullary intradural hemangioma of the spinal cord mimicking meningioma: case report. Br J Neurosurg. 2015;29:438-439. 9. Rahmanian A, Ashraf MH, Owji SM. Report of a case: an intradural between roots capillary hemangioma of cauda equina. IrJNS. 2015;1:44-46.
1.E4
www.SCIENCEDIRECT.com
16. Miri SM, Habibi Z, Hashemi M, Meybodi AT, Tabatabai SA. Capillary hemangioma of cauda equina: a case report. Cases J. 2009;2:80. 17. Kim KJ, Lee JY, Lee SH. Spinal intradural capillary hemangioma. Surg Neurol. 2006;66:212-214. 18. Alkandy LM, Hercules S, Balamurali G, Reid H, Herwadkar A, Holland JP. Thoracic intradural extramedullary capillary hemangioma. Br J Neurosurg. 2006;20:235-238. 19. Ghazi NG, Jane JA, Lopes MB, Newman SA. Capillary hemangioma of the cauda equina presenting with radiculopathy and papilledema. J Neuroophthalmol. 2006;26:98-102.
24. Roncaroli F, Scheithauer BW, Deen HG Jr. Multiple hemangiomas (hemangiomatosis) of the cauda equina and spinal cord: case report. J Neurosurg. 2000;92(2 Suppl):229-232. 25. Roncaroli F, Scheithauer BW, Krauss WE. Hemangioma of spinal nerve root. J Neurosurg. 1999; 91(2 Suppl):175-180. 26. Holtzman RN, Brisson PM, Pearl RE, Gruber ML. Lobular capillary hemangioma of the cauda equina: case report. J Neurosurg. 1999;90(2 Suppl):239-241. 27. Zander DR, Lander P, Just N, Albrecht S, Mohr G. Magnetic resonance imaging features of a nerve root capillary hemangioma of the spinal cord: case report. Can Assoc Radiol J. 1998;49:398-400. 28. Mastronardi L, Guiducci A, Frondizi D, Carletti S, Spera C, Maira G. Intraneural capillary hemangioma of the cauda equina. Eur Spine J. 1997;6:278-280. 29. Frymoyer JW, ed. The Adult Spine: Principles and Practice. New York: Raven Press; 1991:937. 30. Sairyo K, Henmi T, Endo H. Foramen magnum schwannoma with an unusual clinical presentation: case report. Spinal Cord. 1997;35:554-556. 31. Kang JS, Lillehei KO, KleinschmidtDemasters BK. Proximal nerve root capillary hemangioma presenting as a lung mass with bandlike chest pain: case report and review of literature. Surg Neurol. 2006;65:584-589. 32. Vassal F, Péoc’h M, Nuti C. Epidural capillary hemangioma of the thoracic spine with proximal nerve root involvement and extraforaminal extension. Acta Neurochir (Wien). 2011;153:2279-2281. 33. Aoyagi N, Kojima K, Kasai H. Review of spinal epidural cavernous hemangioma. Neurol Med Chir (Tokyo). 2003;43:471-475.
Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 13 March 2016; accepted 30 April 2016 Citation: World Neurosurg. (2016). http://dx.doi.org/10.1016/j.wneu.2016.04.131 Journal homepage: www.WORLDNEUROSURGERY.org
20. Yu H, Lee LY, Hwang SN, Yoo SM, Lee HY, Song IS, et al. Intradural extramedullary capillary hemangioma with long segment of transient cord edema: a case report. J Korean Radiol Soc. 2006;54:343-347.
Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2016 Elsevier Inc. All rights reserved.
WORLD NEUROSURGERY, http://dx.doi.org/10.1016/j.wneu.2016.04.131
Sex
Present case
29
M
C1
1 case
46
F
T11eT12
1 case
53
M
L1-L2
Low back pain and bilateral Hypoesthesia below the left Hyper T1, hypo T2 with radicular pain L2 dermatome contrast enhancement
1 case
53
M
L3eL4
Back pain and subjective weakness of the right leg
1 case
60
M
T2
2F 2M
2 thoracic 2 lumbar
Case
4 cases
53.5 (range, 21e76) 35
M
T12
1 case
48
M
T10eT11
1 case
34
M
L4
1 case
47
M
T6eT7
1 case
20
M
L3
Clinical Features
MRIonance Imaging Findings
Neck pain, paresthesias of Sensory impairment below Iso T1, hyper T2 with the left hand, gait the T6 dermatome contrast enhancement disturbance Bilateral leg weakness and Paraparesis saddle anesthesia
Hyporeflexic left Achilles reflex
Treatment C1 hemilaminectomy with complete resection
Prognosis Complete recovery
Iso T1, hyper T2 with T11eT12 laminectomy with Complete recovery homogeneous enhancement total resection after contrast L1eL2 laminectomy with Pain-free complete en bloc resection of the lesion
Iso T1, iso to mildly hyper L3eL4 laminectomy with Symptom improvement complete en bloc resection T2 lesion with avid homogeneous enhancement of the lesion after contrast
Reference Bouali et al
Alobaid et al8 Rahmanian et al9 Liu et al4
Thoracic girdle pain with Loss of vibration sensation Hypo to iso T1, hyper T2 progressive gait disturbance below the knee with strong enhancement on contrast-enhanced T1
T1eT2 hemilaminectomy with complete resection
Complete recovery
Takata et al10
Back and leg pain
Motor weakness and sensory abnormalities
Not commented
Laminectomy with total resection
Neurologic recovery
Babu et al11
Mid-back pain
Paraparesis, positive Babinski’s sign
Iso T1, hyper T2 with D11eD12 laminectomy homogeneous enhancement with total resection on gadolinium T1
Complete neurologic recovery
Sonawane et al12
Low back pain, gait disturbance
Paraparesis
Iso T1, hyper T2 with contrast enhancement
T10eT11 laminectomy with Complete recovery gross total resection recurrence at 6 months
Kaneko et al13
Low back pain radiating to Motor deficit and absent Hypo T1, hypo T2 with the left leg Achilles reflex in the left leg mostly homogeneous Sensory loss in the left foot enhancement
L4 left hemi-laminectomy with complete removal
Complete recovery
Funayama et al14
Low back pain radiating to Sensory impairment below Iso T1, iso T2 with the legs the T7 dermatome, homogeneous strong enhancement increased deep tendon reflex, and ankle clonus bilaterally
T6eT7 laminectomy with complete tumor removal and T6eT7 laminoplasty
Neurologic improvement
Chung et al15
Low back pain radiating to Bilateral weakness and the legs, urinary retention, asymmetrical diminished and retrograde ejaculation deep tendon reflexes in both lower extremities
L3 laminectomy with en bloc resection
Improvement in weakness Miri et al16 and urogenital problems
Iso T1, iso-hyper T2 with significant homogeneous enhancement
Continues
1.E5
CASE REPORT
www.WORLDNEUROSURGERY.org
1 case
Presenting Symptoms
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
Age, Years
Tumor Location (Level)
SOFIENE BOUALI ET AL.
WORLD NEUROSURGERY -: ---, MONTH 2016
Table 1. Reported Cases of Pure Intradural Extramedullary Capillary Hemangioma
www.SCIENCEDIRECT.com
Age, Years
Sex
1 case
59
M
L1eL2
1 case
60
M
T9
1 case
42
M
L3eL4
Headache, pulsatile Papilledema without tinnitus, transient visual neurologic deficit obscurations, low back pain with right lower extremity radiation
Ill-defined lesion on T1, L3eL4 laminectomy with complete mass removal hypo T2, and intense homogeneous enhancement after contrast
Complete recovery
Ghazi et al19
1 case
48
M
T6eT7
Sudden-onset back pain
Paraparesis and hypoesthesia below T8
Hypo T1, hyper T2, and strong homogeneous enhancement on the contrast-enhanced T1weighted images
T6eT7 laminectomy with complete mass removal
Complete recovery
Yu et al20
1 case
32
M
T10
Low back pain and lower extremity weakness
Left-side Babinski’s sign, T9 Iso T1, hyper T2, and sensory level intense contrast enhancement
T9-T10 laminectomy and total resection
Complete recovery
Abdullah et al21
1 case
41
M
L1eL2
T11-L1 laminectomy and complete tumor resection
Complete recovery
Andaluz et al22
Case 1
28
M
L1
Choi et al23
Case 2
52
M
Case 3
51
1 case
Presenting Symptoms
Clinical Features
MRIonance Imaging Findings
Treatment
Low back pain and left leg Paresthesias in the left L4, Iso T1, hyper T2 enhancing L1eL2 laminectomy with pain L5, and S1 dermatomes and lesion at the level of the L1 complete excision diminished left knee jerk eL2 disk space Low back pain
Paraparesis
Iso T1, hyper T2, and intense contrast enhancement
Prognosis Improved symptoms
T9eT10 laminectomy with Complete recovery complete resection of the tumor
Reference Kim et al17
Alakandy18
Iso T1, hyper T2, and enhanced uniformly after contrast injection
Low back pain
Paraparesis
Not commented
T5eT6
Claudication
Paraparesis, hypoesthesia in both lower limbs
Iso T1, hyper T2, and strong Total mass excision homogeneous enhancement on contrast-enhanced T1 images in all 3 patients
M
T4eT5
Claudication and radiating Hypoesthesia below T5 pain to both lower limbs
63
F
T12eL1
Progressive numbness of Neurologic examination the ventral surface of the was normal left thigh and intermittent low back pain
Hyper T1, iso T2, with T12 laminectomy with homogeneous enhancement complete resection
Residual paresis left tibialis Nowak et al3 anterior on 14-month follow-up; no recurrence
1 case
66
F
T8eT9
Low back pain
Grade 3 paraparesis and sensory abnormality
Iso T1, hyper T2, and T8eT9 laminectomy and homogeneous, strong total resection enhancement on contrastenhanced T1
Complete recovery in clinical findings
Shin et al2
1 case
74
M
L2eL3
Progressive bilateral leg weakness
Paraparesis
Multiple enhancing nodules L2eL3 laminectomy, with in the cauda equina region complete resection
No change in neurologic deficits
Roncaroli et al24
3 cases:
CASE REPORT
Low back pain with bilateral Bilateral patellar and radiculopathy Achilles areflexia
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
WORLD NEUROSURGERY, http://dx.doi.org/10.1016/j.wneu.2016.04.131
Case
Tumor Location (Level)
SOFIENE BOUALI ET AL.
1.E6
Table 1. Continued
40e62
6 M and 8 at the level of 7 cases with 4 F the cauda lumbosciaticasciatica equina, 1 at T5 and 1 at the T1 nerve roots
6 cases with unilateral and bilateral paraparesis; hypoesthesia in 2 cases; 1 case with bowel and bladder dysfunction; 4 cases without deficits
Iso T1, iso or slightly hyper T2 with enhancement following Gd-DTPA administration
In all cases at different levels: 8 complete resection 1 incomplete resection
All patients were free of Roncaroli pain; residual paresis in 3 et al25 patients; persistent hypoesthesia in 2 patients; new hypoesthesia in 1 patient
56
F
L4
Low back pain and rightsided lumbosciatica
Hypoesthesia in the right S1 Iso T1, hyper T2, and L3eL5 laminectomy and dermatome homogeneous enhancement total resection after contrast
Residual paresis
Holtzman et al26
1 case
51
F
L4eL5
Low back pain and rightsided lumbosciatica
Weakness of leg dorsiflexion
Hyper T1, iso T2, L4eL5 laminectomy, with homogeneous enhancement complete resection after contrast
Not mentioned
Zander et al27
1 case
41
M
L5
Intermittent Low back pain and leftsided lumbosciatica
Diminished left Achilles tendon reflex
Hyperintensity on T1, L5 laminectomy, with isointensity on T2, complete resection homogeneous enhancement following Gd-DTPA administration
No neurologic deficits
Mastronardi et al28
1 case
58
M
L1eL2
Mass lesion slightly higher T12eL2 laminectomy and than cauda equina intensity, total resection which was clearly enhanced
Not mentioned
Hanakita et al7
Severe low back pain and Cauda equina syndrome left leg pain
1.E7
CASE REPORT
www.WORLDNEUROSURGERY.org
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
1 case
SOFIENE BOUALI ET AL.
WORLD NEUROSURGERY -: ---, MONTH 2016
10 cases
Intradural Extramedullary Capillary Hemangioma in the Upper Cervical Spine: First Report Sofiene Bouali, Nidhal Maatar, Asma Bouhoula, Khansa Abderrahmen, Jalel Kallel, Hafedh Jemel
Key words Capillary hemangioma - Cervical spine - Extramedullary - Intradural -
Abbreviations and Acronyms MRI: Magnetic resonance imaging Department of Neurosurgery, National Institute of Neurology, Tunis, Tunisia To whom correspondence should be addressed: Sofiene Bouali, M.D. [E-mail: [email protected]] Citation: World Neurosurg. (2016). http://dx.doi.org/10.1016/j.wneu.2016.04.131 Journal homepage: www.WORLDNEUROSURGERY.org
- BACKGROUND:
The occurrence of intradural extramedullary capillary hemangiomas is exceedingly rare. To date, only 39 cases of intradural extramedullary capillary hemangiomas have been reported in the English literature, and all of these cases have been described at the lumbar and thoracic spinal levels. To our knowledge, this report is the first case of capillary hemangiomas of the cervical spine in the literature. In general, this entity is misdiagnosed preoperatively as a neoplasm.
- CASE
DESCRIPTION: A 29-year-old man presented with neck pain and progressive gait disturbance, and was diagnosed with an intradural extramedullary capillary hemangioma in the cervical region.
- CONCLUSIONS:
Although rare, our case demonstrates that capillary hemangioma should be considered in the differential diagnosis of intradural extramedullary tumor of the cervical spine.
Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2016 Elsevier Inc. All rights reserved.
findings, and treatment and outcomes associated with such lesions.
INTRODUCTION Capillary hemangiomas are benign endothelial cell neoplasms, characterized by nodules of capillary-sized vessels lined by flattened endothelium.1 They are most commonly found in the skin or the mucosa of the head and neck.2 Intradural extramedullary capillary hemangioma is extremely rare.1,3,4 To date, only 39 cases of capillary hemangioma affecting the intradural extramedullary spine have been documented in the literature. The lesions were observed in the lumbar spine in 60% of the cases and in the thoracic region in the remaining cases. No previous reports have described capillary hemangiomas located in the cervical spine. Here we report a patient with an intradural extramedullary capillary hemangioma of the upper cervical spine, the first such reported case in the literature. We also review the literature related to intradural extramedullary capillary hemangiomas, and discuss the clinical manifestations, imaging and histopathological
CASE REPORT History and Presentation A 29-year-old right-handed male patient complaining of posterior neck pain for 2 years was admitted to the hospital. He had a 1-year history of numbness in the left distal upper extremity, a 6-month history of right sided paresthesia, and a 3-month history of gradually progressive gait disturbance. He reported feeling slight weakness in his right leg when walking more than a couple of blocks.
Physical Examination Findings Neurologic examination revealed diffuse hyperreflexia, more pronounced on the right side. Strength was symmetric and full bilaterally. Decreased sensation of the right hemibody to light touch and pinprick below the C5 dermatome level was noted. Other general and systemic assessments revealed no abnormalities.
WORLD NEUROSURGERY -: ---, MONTH 2016
Radiologic Findings An unenhanced computed tomography scan at the C1 level revealed a poorly circumscribed mass of low density within the spinal canal, with no bony erosion (Figure 1). Magnetic resonance imaging (MRI) revealed an intradural extramedullary dumbbell-shaped, lobulecontoured mass at the C1 level. The lesion was isointense relative to the spinal cord on T1-weighted MRI and slightly hyperintense on T2-weighted MRI. The tumor showed strong homogeneous enhancement on contrast-enhanced T1-weighted MRI. The tumor exerted a mass effect on the cervical spinal cord, displacing it to the right with compression (Figure 2A). Initial examination revealed a dilated serpentine venous structure coursing toward the left anterior surface of the lesion (Figure 2B). Surgical Findings Based on the foregoing investigations, our preoperative diagnosis was a neurogenic tumor or meningioma. The patient underwent C1 hemilaminectomy. Under general anesthesia, he was placed prone in pin fixation. A midline skin incision was made from the inion to the spinous
www.WORLDNEUROSURGERY.org
1.E1
CASE REPORT SOFIENE BOUALI ET AL.
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
Figure 1. Unenhanced cervical scan revealing a poorly circumscribed mass of low density within the spinal canal at the C1 level, with no bony erosions. (A) Coronal view. (B) Sagittal view. (C and D) Axial view.
process of C4. The unilateral occipital bone and neural arches of C1 and C2 were exposed on the left side only. Under microscopic magnification, a unilateral limited laminectomy was performed on the left-side lamina of C1 with a Kerrison punch. The dura was then opened paramedially, revealing an intradural wellcircumscribed, reddish mass on the ventrolateral side of the cervical spine. The tumor was adherent to the arachnoid and C1 nerve root, especially on the left side. Careful separation of the C1 ventral root and sectioning of the fibers entering the mass revealed a dilated serpentine vein on the left anterior surface of the lesion. This vein was coagulated, the tumor was completely resected without significant bleeding, and tThe dura was closed. The
1.E2
www.SCIENCEDIRECT.com
patient’s neck pain resolved over the course of the first postoperative week, and hypoesthesia resolved in the first postoperative month. Histopathological Findings The histopathological examination revealed typical features of capillary hemangioma, including innumerable thinwalled and irregular capillary vessels lined with endothelium (Figure 3). DISCUSSION Capillary hemangiomas are congenital hamartomatous malformations that result from proliferations of vascular endothelial cells.5,6 They are most commonly found in the skin or mucosa of the head and neck.6
Intradural extramedullary capillary hemangiomas were first described by Hanakita in 1991.7 To date, 39 cases of capillary hemangioma affecting the intradural extramedullary spine have been reported in the literature (Table 1). In the spinal intradural extramedullary space, capillary hemangiomas may arise from the blood vessels of the nerve roots in the cauda equina, the pial surface of the spinal cord, or the inner surface of the dura. Two hypotheses have been proposed regarding the pathogenesis of the disease. The lesion may evolve during early somite differentiation, at the time of angioblastic differentiation (days 21e24 of embryogenesis), owing to impaired movement and differentiation of primitive mesoderm from the embryonic mesodermal plate. Another school of thought identifies the origin as vascular structures within the epineurium of the nerve roots affected during individual ontogenesis.3,16 In the 39 cases reported in the literature, the mean patient age was 50 years (range, 20e74 years), with a male predominance (28 males and 11 females, a ratio of almost 3:1) (Table 1). Pure intradural extramedullary capillary hemangiomas frequently occurred distantly, in the lumbar region (at the conus medullaris or attached to nerve roots of the cauda equine) in 23 cases, and in the thoracic spine in 16 cases (Table 1). There have been no previous reports describing a capillary hemangioma located in the cervical region. Pain was the most common complaint and the initial reported symptom in 36 of the 39 patients (95% of cases). Back pain was often accompanied by radicular pain, especially when the tumor was present in the lumbar spine. Capillary hemangiomas infrequently produce severe neurologic deficits at diagnosis owing to their discrete and noninfiltrative nature.15 Moderate motor deficits were described in 19 cases,9,10 and sphincter dysfunction was present in only 2 cases.16,25 In our patient, preoperative symptoms included neck pain, progressive gait disturbance, and sensory symptoms, including hypoesthesia in the right hemibody. In addition, sensory disturbance and increased deep tendon reflexes were noted in the contralateral extremities. With this
WORLD NEUROSURGERY, http://dx.doi.org/10.1016/j.wneu.2016.04.131
CASE REPORT SOFIENE BOUALI ET AL.
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
Figure 2. (A) MRI of the cervical spine. Sagittal (a) and axial (b) T1-weighted images showing a round, well-circumscribed intradural extramedullary lesion at the level of C1 isointensity relative to the spinal cord, hyperintense T2-weighted sagittal (c) and axial (d) images. The mass enhances homogeneously on sagittal (e) and axial (f) postcontrast T1-weighted sequence and compressing the spinal cord laterally. A vascular-like structure is apparent anteriorly to the mass, indicating enlarged vessels. (B) Preoperative MRI of the cervical spine. (a and b) A dilated serpentine venous structure is seen coursing toward the left anterior surface of the lesion, shown on sagittal T1-weighted (a) and T2-weighted (b) images. (c) Signal void areas (arrowhead) on precocious sagittal postcontrast images indicate draining veins.
WORLD NEUROSURGERY -: ---, MONTH 2016
slow-growing tumor in the intradural space at the C1 level, contralateral cord involvement may be induced by counterpressure on the spinal cord in contact with the contralateral lamina, which may be generated by cord deviation and by frequent neck rotation.29,30 Most capillary hemangiomas are radiologically misdiagnosed and/or mistaken for meningiomas or schwannomas.31 Our patient’s MRI findings are consistent with those in previous reports; the lesion appeared isointense relative to the spinal cord on T1-weighted MRI and isointense or hyperintense on T2-weighted MRI with strong homogeneous gadolinium enhancement.23 In this case, the presence of dilated serpentine venous structures around the lesion shown on T2-weighted MRI, similar to that seen in arteriovenous malformation, were not appreciated before surgery. Our review of the literature describing capillary hemanagiomas of the peripheral or central nervous system found the presence of enlarged draining veins reported in only 3 previous cases.10,21,32 We believe that this subject deserves further attention because it helps identify specific radiologic signs of capillary hemangiomas. All reported cases presented with a pure intradural extramedullary capillary hemangioma, with the mass extending to a maximum of 2 vertebral segments. Histologically, capillary hemangioma are thin, irregular, capillary-sized vessels caught in low-attenuating fibroses with lobular architecture and a lining of a continuous basal lamina. These lesions exhibit low mitotic activity, positive reactions for CD31 and CD34, and negative reactions for S100 and epithelial membrane antigen on histopathological examination.33 Surgery is the treatment of choice for spinal intradural capillary hemangioma. In the literature reports, all patients were initially managed by local excision, and 97% of intradural capillary hemangiomas were completely resected with good outcomes. Only one case reported recurrence by 6 months after gross total resection.13 Preoperative embolization may aid in the surgical resection of these vascular lesions.21
www.WORLDNEUROSURGERY.org
1.E3
CASE REPORT SOFIENE BOUALI ET AL.
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
21. Abdullah DC, Raghuram K, Philips CD, Jane JA Jr, Miller B. Thoracic intradural extramedullary capillary hemangioma. AJNR Am J Neuroradiol. 2004;25:1294-1296. 22. Andaluz N, Balko MG, Stanek J, Morgan C, Schwetschenau PR. Lobular capillary hemangioma of the spinal cord: case report and review of the literature. J Neurooncol. 2002;56:261-264. 23. Choi BY, Chang KH, Choe G, Han MH, Park SW, Yu IK, et al. Spinal intradural extramedullary capillary hemangioma: MR imaging findings. AJNR Am J Neuroradiol. 2001;22:799-802.
Figure 3. Histology revealing typical features of capillary hemangioma. (A) Lobular architecture with numerous capillary-sized vessels lined by a single layer of endothelial cells and dissemination of dilated vessels (hematoxylin and eosin; original magnification 5). (B) Larger-caliber and medium-sized capillary vessels lined by flattened endothelium (hematoxylin and eosin; original magnification 20).
CONCLUSION This case demonstrates that capillary hemangioma, although rare, should be considered in the differential diagnosis of intradural extramedullary tumor of the cervical spine. REFERENCES 1. Enzinger FM, Weiss SW. Benign tumors and tumor-like lesions of blood vessels. In: Enzinger FM, Weiss SW, eds. Soft tissue tumors. 3rd ed. St. Louis, MO: Mosby; 1995:579-626. 2. Shin JH, Lee HK, Jeon SR, Park SH. Spinal intradural capillary hemangioma: MR findings. AJNR Am J Neuroradiol. 2000;21:954-956.
10. Takata Y, Sakai T, Higashino K, Goda Y, Tezuka F, Sairyo K. Intradural extramedullary capillary hemangioma in the upper thoracic spine: a review of the literature. Case Rep Orthop. 2014; 2014:604131. 11. Babu R, Owens TR, Karikari IO, Moreno J, Cummings TJ, Gottfried ON, et al. Spinal cavernous and capillary hemangiomas in adults. Spine (Phila Pa 1976). 2013;38:E423-E430. 12. Sonawane DV, Jagtap SA, Mathesul AA. Intradural extramedullary capillary hemangioma of lower thoracic spinal cord. Indian J Orthop. 2012;46:475-478. 13. Kaneko Y, Yamabe K, Abe M. Rapid regrowth of a capillary hemangioma of the thoracic spinal cord. Neurol Med Chir (Tokyo). 2012;52:665-669.
3. Nowak DA, Widenka DC. Spinal intradural capillary haemangioma: a review. Eur Spine J. 2001;10: 464-472.
14. Funayama T, Sakane M, Murai S, Ochiai N. Multiple capillary hemangiomas of the cauda equina at a level of a single vertebra. J Orthop Sci. 2010;15: 598-602.
4. Liu JJ, Lee DJ, Jin LW, Kim KD. Intradural extramedullary capillary hemangioma of the cauda equina: case report and literature review. Surg Neurol Int. 2015;6(Suppl 3):S127-S131.
15. Chung SK, Nam TK, Park SW, Hwang SN. Capillary hemangioma of the thoracic spinal cord. J Korean Neurosurg Soc. 2010;48:272-275.
5. Murphey MD, Fairbairn KJ, Parman LM, Baxter KG, Parsa MB, Smith WS. From the archives of the AFIP. Musculoskeletal angiomatous lesions: radiologic-pathologic correlation. Radiographics. 1995;15:893-917. 6. Feider HK, Yuille DL. An epidural cavernous hemangioma of the spine. AJNR Am J Neuroradiol. 1991;12:243-244. 7. Hanakita J, Suwa H, Nagayasu S, Suzuki H. Capillary hemangioma in the cauda equina: neuroradiological findings. Neuroradiology. 1991;33:458-461. 8. Alobaid A, Bennardo MR, Cenic A, Lach B. Mixed capillary-cavernous extramedullary intradural hemangioma of the spinal cord mimicking meningioma: case report. Br J Neurosurg. 2015;29:438-439. 9. Rahmanian A, Ashraf MH, Owji SM. Report of a case: an intradural between roots capillary hemangioma of cauda equina. IrJNS. 2015;1:44-46.
1.E4
www.SCIENCEDIRECT.com
16. Miri SM, Habibi Z, Hashemi M, Meybodi AT, Tabatabai SA. Capillary hemangioma of cauda equina: a case report. Cases J. 2009;2:80. 17. Kim KJ, Lee JY, Lee SH. Spinal intradural capillary hemangioma. Surg Neurol. 2006;66:212-214. 18. Alkandy LM, Hercules S, Balamurali G, Reid H, Herwadkar A, Holland JP. Thoracic intradural extramedullary capillary hemangioma. Br J Neurosurg. 2006;20:235-238. 19. Ghazi NG, Jane JA, Lopes MB, Newman SA. Capillary hemangioma of the cauda equina presenting with radiculopathy and papilledema. J Neuroophthalmol. 2006;26:98-102.
24. Roncaroli F, Scheithauer BW, Deen HG Jr. Multiple hemangiomas (hemangiomatosis) of the cauda equina and spinal cord: case report. J Neurosurg. 2000;92(2 Suppl):229-232. 25. Roncaroli F, Scheithauer BW, Krauss WE. Hemangioma of spinal nerve root. J Neurosurg. 1999; 91(2 Suppl):175-180. 26. Holtzman RN, Brisson PM, Pearl RE, Gruber ML. Lobular capillary hemangioma of the cauda equina: case report. J Neurosurg. 1999;90(2 Suppl):239-241. 27. Zander DR, Lander P, Just N, Albrecht S, Mohr G. Magnetic resonance imaging features of a nerve root capillary hemangioma of the spinal cord: case report. Can Assoc Radiol J. 1998;49:398-400. 28. Mastronardi L, Guiducci A, Frondizi D, Carletti S, Spera C, Maira G. Intraneural capillary hemangioma of the cauda equina. Eur Spine J. 1997;6:278-280. 29. Frymoyer JW, ed. The Adult Spine: Principles and Practice. New York: Raven Press; 1991:937. 30. Sairyo K, Henmi T, Endo H. Foramen magnum schwannoma with an unusual clinical presentation: case report. Spinal Cord. 1997;35:554-556. 31. Kang JS, Lillehei KO, KleinschmidtDemasters BK. Proximal nerve root capillary hemangioma presenting as a lung mass with bandlike chest pain: case report and review of literature. Surg Neurol. 2006;65:584-589. 32. Vassal F, Péoc’h M, Nuti C. Epidural capillary hemangioma of the thoracic spine with proximal nerve root involvement and extraforaminal extension. Acta Neurochir (Wien). 2011;153:2279-2281. 33. Aoyagi N, Kojima K, Kasai H. Review of spinal epidural cavernous hemangioma. Neurol Med Chir (Tokyo). 2003;43:471-475.
Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 13 March 2016; accepted 30 April 2016 Citation: World Neurosurg. (2016). http://dx.doi.org/10.1016/j.wneu.2016.04.131 Journal homepage: www.WORLDNEUROSURGERY.org
20. Yu H, Lee LY, Hwang SN, Yoo SM, Lee HY, Song IS, et al. Intradural extramedullary capillary hemangioma with long segment of transient cord edema: a case report. J Korean Radiol Soc. 2006;54:343-347.
Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2016 Elsevier Inc. All rights reserved.
WORLD NEUROSURGERY, http://dx.doi.org/10.1016/j.wneu.2016.04.131
Sex
Present case
29
M
C1
1 case
46
F
T11eT12
1 case
53
M
L1-L2
Low back pain and bilateral Hypoesthesia below the left Hyper T1, hypo T2 with radicular pain L2 dermatome contrast enhancement
1 case
53
M
L3eL4
Back pain and subjective weakness of the right leg
1 case
60
M
T2
2F 2M
2 thoracic 2 lumbar
Case
4 cases
53.5 (range, 21e76) 35
M
T12
1 case
48
M
T10eT11
1 case
34
M
L4
1 case
47
M
T6eT7
1 case
20
M
L3
Clinical Features
MRIonance Imaging Findings
Neck pain, paresthesias of Sensory impairment below Iso T1, hyper T2 with the left hand, gait the T6 dermatome contrast enhancement disturbance Bilateral leg weakness and Paraparesis saddle anesthesia
Hyporeflexic left Achilles reflex
Treatment C1 hemilaminectomy with complete resection
Prognosis Complete recovery
Iso T1, hyper T2 with T11eT12 laminectomy with Complete recovery homogeneous enhancement total resection after contrast L1eL2 laminectomy with Pain-free complete en bloc resection of the lesion
Iso T1, iso to mildly hyper L3eL4 laminectomy with Symptom improvement complete en bloc resection T2 lesion with avid homogeneous enhancement of the lesion after contrast
Reference Bouali et al
Alobaid et al8 Rahmanian et al9 Liu et al4
Thoracic girdle pain with Loss of vibration sensation Hypo to iso T1, hyper T2 progressive gait disturbance below the knee with strong enhancement on contrast-enhanced T1
T1eT2 hemilaminectomy with complete resection
Complete recovery
Takata et al10
Back and leg pain
Motor weakness and sensory abnormalities
Not commented
Laminectomy with total resection
Neurologic recovery
Babu et al11
Mid-back pain
Paraparesis, positive Babinski’s sign
Iso T1, hyper T2 with D11eD12 laminectomy homogeneous enhancement with total resection on gadolinium T1
Complete neurologic recovery
Sonawane et al12
Low back pain, gait disturbance
Paraparesis
Iso T1, hyper T2 with contrast enhancement
T10eT11 laminectomy with Complete recovery gross total resection recurrence at 6 months
Kaneko et al13
Low back pain radiating to Motor deficit and absent Hypo T1, hypo T2 with the left leg Achilles reflex in the left leg mostly homogeneous Sensory loss in the left foot enhancement
L4 left hemi-laminectomy with complete removal
Complete recovery
Funayama et al14
Low back pain radiating to Sensory impairment below Iso T1, iso T2 with the legs the T7 dermatome, homogeneous strong enhancement increased deep tendon reflex, and ankle clonus bilaterally
T6eT7 laminectomy with complete tumor removal and T6eT7 laminoplasty
Neurologic improvement
Chung et al15
Low back pain radiating to Bilateral weakness and the legs, urinary retention, asymmetrical diminished and retrograde ejaculation deep tendon reflexes in both lower extremities
L3 laminectomy with en bloc resection
Improvement in weakness Miri et al16 and urogenital problems
Iso T1, iso-hyper T2 with significant homogeneous enhancement
Continues
1.E5
CASE REPORT
www.WORLDNEUROSURGERY.org
1 case
Presenting Symptoms
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
Age, Years
Tumor Location (Level)
SOFIENE BOUALI ET AL.
WORLD NEUROSURGERY -: ---, MONTH 2016
Table 1. Reported Cases of Pure Intradural Extramedullary Capillary Hemangioma
www.SCIENCEDIRECT.com
Age, Years
Sex
1 case
59
M
L1eL2
1 case
60
M
T9
1 case
42
M
L3eL4
Headache, pulsatile Papilledema without tinnitus, transient visual neurologic deficit obscurations, low back pain with right lower extremity radiation
Ill-defined lesion on T1, L3eL4 laminectomy with complete mass removal hypo T2, and intense homogeneous enhancement after contrast
Complete recovery
Ghazi et al19
1 case
48
M
T6eT7
Sudden-onset back pain
Paraparesis and hypoesthesia below T8
Hypo T1, hyper T2, and strong homogeneous enhancement on the contrast-enhanced T1weighted images
T6eT7 laminectomy with complete mass removal
Complete recovery
Yu et al20
1 case
32
M
T10
Low back pain and lower extremity weakness
Left-side Babinski’s sign, T9 Iso T1, hyper T2, and sensory level intense contrast enhancement
T9-T10 laminectomy and total resection
Complete recovery
Abdullah et al21
1 case
41
M
L1eL2
T11-L1 laminectomy and complete tumor resection
Complete recovery
Andaluz et al22
Case 1
28
M
L1
Choi et al23
Case 2
52
M
Case 3
51
1 case
Presenting Symptoms
Clinical Features
MRIonance Imaging Findings
Treatment
Low back pain and left leg Paresthesias in the left L4, Iso T1, hyper T2 enhancing L1eL2 laminectomy with pain L5, and S1 dermatomes and lesion at the level of the L1 complete excision diminished left knee jerk eL2 disk space Low back pain
Paraparesis
Iso T1, hyper T2, and intense contrast enhancement
Prognosis Improved symptoms
T9eT10 laminectomy with Complete recovery complete resection of the tumor
Reference Kim et al17
Alakandy18
Iso T1, hyper T2, and enhanced uniformly after contrast injection
Low back pain
Paraparesis
Not commented
T5eT6
Claudication
Paraparesis, hypoesthesia in both lower limbs
Iso T1, hyper T2, and strong Total mass excision homogeneous enhancement on contrast-enhanced T1 images in all 3 patients
M
T4eT5
Claudication and radiating Hypoesthesia below T5 pain to both lower limbs
63
F
T12eL1
Progressive numbness of Neurologic examination the ventral surface of the was normal left thigh and intermittent low back pain
Hyper T1, iso T2, with T12 laminectomy with homogeneous enhancement complete resection
Residual paresis left tibialis Nowak et al3 anterior on 14-month follow-up; no recurrence
1 case
66
F
T8eT9
Low back pain
Grade 3 paraparesis and sensory abnormality
Iso T1, hyper T2, and T8eT9 laminectomy and homogeneous, strong total resection enhancement on contrastenhanced T1
Complete recovery in clinical findings
Shin et al2
1 case
74
M
L2eL3
Progressive bilateral leg weakness
Paraparesis
Multiple enhancing nodules L2eL3 laminectomy, with in the cauda equina region complete resection
No change in neurologic deficits
Roncaroli et al24
3 cases:
CASE REPORT
Low back pain with bilateral Bilateral patellar and radiculopathy Achilles areflexia
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
WORLD NEUROSURGERY, http://dx.doi.org/10.1016/j.wneu.2016.04.131
Case
Tumor Location (Level)
SOFIENE BOUALI ET AL.
1.E6
Table 1. Continued
40e62
6 M and 8 at the level of 7 cases with 4 F the cauda lumbosciaticasciatica equina, 1 at T5 and 1 at the T1 nerve roots
6 cases with unilateral and bilateral paraparesis; hypoesthesia in 2 cases; 1 case with bowel and bladder dysfunction; 4 cases without deficits
Iso T1, iso or slightly hyper T2 with enhancement following Gd-DTPA administration
In all cases at different levels: 8 complete resection 1 incomplete resection
All patients were free of Roncaroli pain; residual paresis in 3 et al25 patients; persistent hypoesthesia in 2 patients; new hypoesthesia in 1 patient
56
F
L4
Low back pain and rightsided lumbosciatica
Hypoesthesia in the right S1 Iso T1, hyper T2, and L3eL5 laminectomy and dermatome homogeneous enhancement total resection after contrast
Residual paresis
Holtzman et al26
1 case
51
F
L4eL5
Low back pain and rightsided lumbosciatica
Weakness of leg dorsiflexion
Hyper T1, iso T2, L4eL5 laminectomy, with homogeneous enhancement complete resection after contrast
Not mentioned
Zander et al27
1 case
41
M
L5
Intermittent Low back pain and leftsided lumbosciatica
Diminished left Achilles tendon reflex
Hyperintensity on T1, L5 laminectomy, with isointensity on T2, complete resection homogeneous enhancement following Gd-DTPA administration
No neurologic deficits
Mastronardi et al28
1 case
58
M
L1eL2
Mass lesion slightly higher T12eL2 laminectomy and than cauda equina intensity, total resection which was clearly enhanced
Not mentioned
Hanakita et al7
Severe low back pain and Cauda equina syndrome left leg pain
1.E7
CASE REPORT
www.WORLDNEUROSURGERY.org
INTRADURAL EXTRAMEDULLARY CAPILLARY HEMANGIOMA IN THE UPPER CERVICAL SPINE
1 case
SOFIENE BOUALI ET AL.
WORLD NEUROSURGERY -: ---, MONTH 2016
10 cases