1. G. Rizzi, A. Berardi, V. Bozzini, M. Gladi, G. Merlicco
A rare case of cauda equina cavernous angioma with intratumoral and
subaracnoid hemorrhage. Case report and review of the Literature
G. Rizzi, A. Berardi, V. Bozzini, M. Gladi, G. Merlicco
Department of Neurosurgery, Azienda Ospedaliero-Universitaria, “OO-RR” Foggia, Italy
Introduction The management of patients with benign intradural extramedullary spinal tumors differs in many aspects from approaches for their intracranial counterpart. Cavernous angiomas of the spinal cord are extremely rare lesions, in fact they represent only 5% to 12% of all vascular lesions of the spine. Their location in intradural extramedullary space and their coexisting with multiple cerebral cavernous angioma is rare. A rare case of cavernous angioma of the cauda equina with intralesion and spinal subarachnoid hemorrhage in patient with multiple cerebral cavernous hemangiomas is reported.
Materials and Methods
A 51-years-old woman, with family and personal asymptomatic history of multiple cavernous cerebral angiomas located in temporal, occipital left lobe and in pontomesencefalic region [fig.1]. came under our observation for a six month history of low back pain, bilateral sciatica and paraparesis. The neurologic examination showed her to have hypoesthesia without a clear radicular distribution. Absence of rotuleus, achilles and plantal reflex was noted. The patient underwent an MRI scan of the lumbar region that showed the presence of an intradural-extramedullary lesion of 2.1 cm in correspondence with L2. The lesion appeared hyperintense on T1-weighted images and disomogeneus in long TR sequences. MR imaging with gadolinium no increment signal in T1 while an anomalous enhancement of a spinal root nerve was evident [fig 1]. Presence of hemorrhagic fluid at L5-S1.
Fig 1A T1W SPIR MDC
Fig. 1B T2W TSE
Figure 1A/B: Preoperative T1 and T2 weighted MRI scan after contrast enhancement, showing heterogeneous hyperintense intradural cavernous angioma at L2 level
The patient underwent L1-L2-L3 laminectomy. The dura was opened under microscope magnification. Among the spinal root nerve of cauda equine, a 2.1 x 2 cm dark-bluish mass was exposed closely adherent to a single root nerve [fig.2]. After cutting the root, the lesion was totally removed. Histopathologic examination showed a large pseudocystic space containing blood and numerous blood vessels of various sizes with thin walls close by, lined by flat endothelium suggestive of a cavernous hemangioma with abundant intralesional hemorrhage [fig 3-4-5]. The post-operative course was free from complications. The paraparesis and sensory deficits improved and the patient was discharged on the sixth postoperative day. MRI at six months after surgery showed the complete removal of the lesion [fig 6]. The patient is healthy and free of cerebral cavernous angiomas in follow-up.
Fig4
Fig 3
Fig.3. Low power magnification shows a large hemorragic pseudocyst and numerous dilated blood vessels (H&E, 20x).
Fig.4. Cavernous blood vessels in the nerve (H&E, 100x).
Fig.5. Immunohistochemical staining for CD31 highlights endothelial cells (IHC, 100x).
Fig 5
Fig : 6 .Postoperative MRI at six months after surgery showing the completely removal of the lesion
Fig: 2. Intraoperative photographs showing a 2.1 x 2 cm dark-bluish mass was exposed closely adherent to a single root nerve
Results
The post-operative course was free of complication. The paraparesis and sensory deficits improved and the patient was discharged on the sixth postoperative day. MRI at six months after surgery showed the completely removal of the lesion [ fig 7]. The patient is healthy and in follow-up of cerebral cavernous angiomas.
Conclusions: Cavernous angiomas of the cauda equine, in particular in association with cerebral cavernous hemangiomas, is an extremely rare condition that may present low back pain, sciatica, neurologic deficit and subarachnoid hemorrhage. It can be successfully treated by surgery in order to prevent bleeding and dangerous enlargement of the lesion.