Original Article
Volume 4, No.2
October 2000
 Rabi Khazim
 Youssef Fares
 G Muthukumar
 Douglas Hedden
 Centre for Spinal Studies
   & Surgery
 Queen’s Medical Centre  Nottingham, NG7 2UH
 United Kingdom
 Correspondence:
 Dr. Rabi Khazim
 Consultant Orthopaedic and Spinal    Surgeon
 Southend Hospital
 Prittlewell Chase
 Westcliff-On-Sea
 Essex SSO ORY
 England
 Fax: (44) 1702 221088
 E-mail:rabi@khazim.freeserve.co.uk
 
Surgical Treatment of Aggressive Vertebral Haemangioma - 8 Cases

   ABSTACT
Vertebral angiomas are often asymptomatic lesions but a few behave as aggressive tumours and compress the spinal cord or nerve roots. These compressive lesions must be treated aggressively. The treatment modality is not unanimously accepted. We report here our experience with 8 cases of aggressive vertebral haemangioma (AVH) treated surgically. There were 4 male and 4 female, two of whom were pregnant. The average age was 30 years. Seven patients had spinal symptoms, 1 had severe intercostal neuralgia. Computed tomography (CT) and x-ray confirmed the diagnosis. Four patients were evaluated with magnetic resonance imaging (MRI) and 2 with angiography. The lesions involved the thoracic spine in 6 cases and the cervical in 2 cases. Only 1 vertebra was involved in each case. Treatment consisted of anterolateral approach with corporectomy in 4 cases, laminectomy in 4 cases. Vertebroplasty procedure with transpedicular injection of methyl-methacrylate into the body was done in 2 cases. The follow-up period ranged between 6 months and 4 years and showed total improvement in 6 cases; 4 of them had undergone anterolateral approach. We conclude that vertebrectomy is the treatment of choice for AVH. Laminectomy should be associated to a vertebroplasty or osteosynthesis of the spine.

Keywords: Vertebral haemangioma, spinal cord, and intercostal neuralgia
INTRODUCTION
Vertebral haemangioms (VH) are frequent benign tumours affecting 12% of the population.(2) These angiomas are very often asymptomatic.(2,3) They rarely become aggressive and compress the spinal cord or the roots in which case they require surgical treatment, but up to now no therapeutic method is unanimously accepted.(2) We report on our experience with 8 cases of aggressive vertebral haemangioma (AVH) and discuss their pathological, clinical, radiological and therapeutic aspects.
MATERIALS AND METHODS
Between 1985-1998, 8 patients suffering from AVH were managed at the Hopital des specialites in Rabat. There were 4 males and 4 females. The mean age was 33 years and ranged between 20 and 42 years (Table 1).

The clinical signs developed gradually in 5 cases and rapidly in 3 cases. VH was associated with pregnancy in 2 cases and spinal trauma in 2 cases. All the patients had been suffering for months from spinal pain associated with neurological disorders such as spastic tetraplegia in 2 cases (Cases 1 and 3) and paraplegia of Grade B on the Frankel scale in 4 cases (Cases 2,4,7 and 8) and Grade C in Case 5. In Case 6, the clinical signs were localised to T9 intercostal neuralgia. Plain spine films and computed tomography (CT) scan focused on the lesion were obtained in all patients. Four were explored with magnetic resonance imaging (MRI). Spinal angiography was undertaken in selective cases when the above studies suggested large feeding or draining vessels. It was performed in only 2 cases. The radiological check-up showed a “honeycomb” appearance of the involved vertebra in all cases, typical of vertebral haemangioma (Fig. 1). The thoracic spine was involved in 6 cases and the cervical spine in 2 cases. One vertebra was involved in all cases. In addition to this, radiological examination showed an intracanalicular extension with spinal cord compression and paraspinal extension. MRI also revealed high intramedullary signal indicating spinal lesion on T2-weighted images (Fig. 2 and 3). Spinal angiography used in 2 cases revealed moderate hypervascularisation (Fig. 4). All the patients were surgically treated. We used an anterior approach in 4 cases (Cases 1, 3, 4 and 8). Resection of the vertebral body with an iliac crest graft. We also used the posterior approach: laminectomy in 4 cases, Cases 2, 5, 6 and 7).

Table 1 - Case Summaries
Case
Clinical Signs
Further examinations
Treatment
Evolution
1 26-year-old patient suffering from spastic tetraplegia of Grade B Frankel scale (FS) Plain films: C4 haemangioma MRI: showing C4 angioma associated with epidural haemangioma Angiography: tumoural blush Corporectomy of C4 Removal of the epidural angioma and iliac graft Complete recovery 3-year follow-up
2 42-year-old woman, 7 months pregnant suffering from Grade B paraplegia FS X-ray: T3 haemangioma MRI: T3 haemangioma associated with epidural haemangioma Laminectomy of T3 and acrylic cement injections Partial recovery (Grade D) 4-year follow-up
3 34-year-old patient tetraplegia predominant in the lower limbs Standard radiology and CT scan myelography showing typical appearance of C7 haemangioma C7 corporectomy of T6 and iliac graft Complete recovery of motor deficit 6-year follow-up
4 36-year-old woman, 8 months pregnant suffering from severe paraplegia of Grade B FS Standard radiology and MRI of the spine reveal T6 haemangioma combined with epidural extension Corporectomy of T6 and iliac graft Complete recovery of motor deficit 6-year follow-up
5 40-year-old patient suffering from Grade C FS paraparesia Plain films revealing a honeycomb appearance of T5. CT scan showed a T5 haemangioma Laminectomy of T5 and iliac graft Complete recovery of the motor deficit but development of a considerable kyphosis 4-year follow-up
6 34-year-old patient suffering from T9 intercostal neuralgia CT scan: typical appearance of a T9 haematoma with epidural extension Laminectomy of T9 associated with acrylic cement injection Absence of intercostal neuralgia 6-year follow-up
7 33-year-old patient suffering from severe paraplegia Grade B FS Plain films revealing a honeycomb appearance of T3. MRI showing a T3 haemangioma T3 laminectomy and ostheosynthesis using CD material Favourable evolution to Grade E 1-year follow-up
8 20-year-old patient suffering from a rachidian trauma in addition to Grade B paraplegia FS MRI indicated an angimatous T5 associated with an epidural haemangioma Corporectomy of T5 and removal of the epidural haemangioma and iliac graft Very satisfactory Recovery to Grade E


Figure 1 - Plain films of the cervical spine, sagittal view: A honeycomb appearance of C4.   Figure 2 - Sagittal T2-weighted image showing a C4 haemangioma with epidural extension and an intramedullary high intensity signal corresponding to a spinal injury.



Figure 3 - Sagittal T2-weighted image showing a T3 haemangioma with epidural extension compressing the spinal cord.		   Figure 4 - Spinal angiography in case of C4 haemangioma showing a moderate hypervascularisation within bone and soft tissue.  


 
Figure 5 - Plain films of thoracic spine showing radiological control after vertebroplasty of a T9 haemangioma.   Figure 6 - Control x-ray after corporectomy for a C4 haemangioma.


Figure 7 - Control MRI (sagittal T1-weighted image) after corporectomy for a T5 haemangioma showing iliac graft and spinal cord decompression.

Figure 8 - Control MRI on T2-weighted image showing severe spinal deformation after a laminectomy alone for a T5 haemangioma.
This laminectomy was associated with an osteo-synthesis using Cotrel Dubousset material in Case 7. In Case 5, surgery was dual. At first we approached from the anterior side but heavy bleeding made us turn to the posterior. Two patients (Cases 2 and 6) had transpedicular injections of acrylic cement (Fig. 5). However, none of our patients underwent pre-operative embolisation to reduce vascularity.

All patients were reviewed on a regular basis: every 3 months for one year, then every 6 months for 2 years and finally every year. After a 36-month follow-up, we performed clinical and radiological assessments.

RESULTS
As far as the immediate post-operative evolution was concerned, 6 patients began to recover (Cases 1, 2, 4, 5, 7 and 8). We noted the absence of intercostal neuralgia (Case 6) and only one patient (Case 3) remained in the same neurological status (paraplegia of Grade B on the Frankel scale). However, 1 patient (Case 2) who had a verebro-plasty presented, post-operatively, with intercostal neuralgia which was resolved in 10 days following intervention. The long-term evolution was favourable. The 2 patients (Cases 1 and 3) suffering from spastic tetraplegia on C4 and C7 haemangioma totally recovered from their neurological deficit. Their neurological examination was considered normal apart from a slight hyperreflexia. Four patients improved from Grade B or C paraplegia to Grade E (Cases 4, 5, 7 and 8).

Only 1 patient (Case 2) remained with a slight neurological deficit but was able to walk (Grade D). The radiological controls were, in general, satisfactory (Fig. 6 and 7). The patients who were
operated upon from the anterior side recovered completely from their neurological deficit. The grafts were fused and no spinal deformation was noticed. In Case 5, no vertebroplasty or osteosynthesis was carried out (Fig. 8).

DISCUSSION

Vertebral haemangiomas with aggressive neurological signs are rare.11-13 Less than 330 cases have been mentioned up to 1992.(2) Both sexes are equally affected and the mean age group is 40 years.(2) The predominant site is the thoracic region between T3 and T9.(1) The cervical spine is rarely involved.(11) Only 1 vertebra is affected in 77% of cases of VH and multiple location is rare.4 Pregnancy and spinal trauma can reveal this condition.12 In our series, we noticed this in 2 cases.

Different mechanisms may explain the role of gestation in the presence of neurological signs.(1)
        a) the enlargement of a previously existing haematoma due to vascular changes,
        b) increase of venous pressure and
        c) hormonal changes occurring during pregnancy which are largely responsible for the growth of a pre-existing             haematoma because of the vascular lining.

Clinical signs are dominated by medullary or radicular compression. The myelopathy secondary to AVH is caused by many factors. Most common are the epidural extension of vertebral angioma in the concentric reduction of the spinal canal.(5,10) Other factors can interfere and occur ie. a compressive pathological fracture, an haema-torachis or a medullary ischaemia due to diversion of the blood flow causing neurological symptoms.(3,4) The diagnosis of AVH can often be made by the characteristic appearance on x-ray films. The vertebra carries vertical striations and trabecul-ations. These are most commonly seen in the vertebral body but may be present elsewhere. The CT scan is mandatory for two reasons:
  1) to confirm the diagnosis, as indicated by irregular spongious appearance coexisting with the presence of lytic areas and diffused cortical swelling.(8)  
  2) to appreciate the size of the tumour to know whether the lesion of the vertebral area is partial or complete; or if the posterior arch is affected and extent of the paraspinal soft tissue involvement.  

The MRI may also provide additional information regarding the aggressiveness of the haemangioma.(13) Mottled, increased “fatty” signals on both T1- and T2- weighted MRI of intraosseous haemangiomas are characteristic of non-evolving intraosseous lesions. Conversely, osseous, or extraosseous haemangiomas that show an isointense signal on T1-weighted images are commonly found with symptomatic lesions. Laredo concluded that fat-intensity lesions are generally inactive, while haemangiomas demonstrating soft tissue intensity on CT scan and low signal intensity on T1-weighted images indicates a hypervascular lesion with the potential to compress the spinal cord.(13)

Spinal angiography gives important information. Firstly, the relationship between the vascularisation of the spine and the tumour and the intensity of this vascularisation. Secondly, it identifies feeding and draining vessels, especially the Adamkiewickz artery when the angioma is located in the low thoracic spine.(14,17)

Once VH is diagnosed, the problem is to choose the most effective therapeutic strategy leading to decompression of the spinal cord.(2)

Numerous therapeutic alternatives exist.(6) Surgery can consist of the resection of the vertebral body and the epidural haemangioma associated with an iliac crest or tibial graft; a decompressive laminectomy with transpedicular injection of acrylic cement or its percutaneous injection in order to set the vertebra. Other options include pre-operative embolisation that will reduce the risk of bleeding, and radiotherapy. All these methods may be used individually or in combination

Embolisation was first used in 1973.(16) It requires the use of a medullar angiography in order to ensure that the angioma is not vascularised by a spinal artery.(16) However, it may not be applied as such for aggressive vertebral haemangiomas. Its main interest is to allow surgery with the lowest bleeding rate.(2)

Vertebroplasty has greatly developed in recent years and has given encouraging results.(17,18) It ensures mechanical stability of the vertebra.(2) It is so effective that it may be used alone when the haemangioma has caused radicular or spinal pain, as was the case with our sixth patient. Intercostal neuralgia secondary to the epidural leak of the acrylic cement have been reported.(2,17) One case of intercostal neuralgia, 10 days post-operative, was noticed in our series.

Surgery is indicated when the haemangioma has caused neurological deficits.(2) It consists of an extensive resection of the haemangioma.(6,13) It might be a corporectomy, arthropediculectomy, or a laminectomy depending on the site of the lesion.(6,13) A pre-operative embolisation is not required in most cases, especially when the angiography has shown a poorly vascularised haemangioma as was the case with 2 of our patients.(15) The operation may be a laminectomy if the anterior approach has failed and/or the patient’s health does not allow heavy surgical intervention.(5,13) Laminectomy should be combined with transpedicular acrylic cement injections or with an osteosynthesis of the rachis in order to avoid post-operative kyphosis.(2)

Radiotherapy has been used since 1930. A dose ranging from 2,000 to 4,000 rad is indicated even though its fibrous effect on this type of tumour is not clearly proven. It is mainly used in cases of medical contraindication to surgery, in case of recurrence, or in case of multiple vertebral haemangioma.(2,11) However, there is risk of post-radiotherapy myelopathy or pulmonary radio-necrosis.

Table 2 - Frankel scale
Grade A:
 Complete motor or sensory loss
Grade B:
 Some sensory preservation but no motor functions
Grade C:
 Some motor power, but of no practical use
Grade D:
 Useful motor power
Grade E:
 Normal motor and sensory functions

CONCLUSION
Through the review of our 8 cases, we can better support the therapeutic indications in the event of AVH. A medullar compression requires a corporectomy with removal of the epidural haemangioma, yet pre-operative embolisation is seldom necessary. In case the anterior approach fails or if the neurological symptoms are slight, the neurosurgeon may alternatively resort to a laminectomy with vertebroplasty or an ostheosynthesis of the spine. The occurrence of isolated intercostal neuralgia requires only vertebroplasty.

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