| Article Access Statistics|
| Viewed||1189 |
| Printed||24 |
| Emailed||0 |
| PDF Downloaded||60 |
| Comments ||[Add] |
Click on image for details.
|LETTER TO THE EDITOR
|Year : 2014 | Volume
| Issue : 5 | Page : 572-573
Radiation-induced cavernous angioma in an adult
Damodar Rout1, KM Geetha Sharmi1, R Rajeswaran2
1 Department of Neurosurgery, Sri Ramachandra University, Chennai, Tamil Nadu, India
2 Department of Radiology, Sri Ramachandra University, Chennai, Tamil Nadu, India
|Date of Submission||09-Oct-2014|
|Date of Decision||10-Oct-2014|
|Date of Acceptance||21-Oct-2014|
|Date of Web Publication||12-Nov-2014|
Department of Radiology, Sri Ramachandra University, Chennai, Tamil Nadu
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Rout D, Geetha Sharmi K M, Rajeswaran R. Radiation-induced cavernous angioma in an adult. Neurol India 2014;62:572-3
Radiation-induced cavernoma (RIC) has been suspected since the first description by Ciricillo SF et al., in 1994.  Till January 2014 only 86 cases of RIC have been reported.  Here is yet another case report.
A 31-year-old male presented with one episode of generalized tonic-clonic seizure. He had a previous history of surgery for a suprasellar tumor followed by 60 Gy of radiation therapy at the age of 17. Magnetic resonance imaging (MRI) done at that time revealed a suprasellar mass and involving the optic chiasm [Figure 1]. MRI repeated as part of evaluation of seizure revealed a right medial temporal perisylvian heterointense lesion on T1-/T2-weighted images measuring 2 × 2.2 × 2.7 cms with blooming on gradient images suggesting cavernoma [Figure 2]. The MRI also showed post operative changes in the left temporal lobe and suprasellar region done 14 years back. On cerebral angiography the lesion was occult. Total excision of the cavernoma was done by fronto-temporal craniotomy. Histopathological examination showed features consistent with cavernous angioma [Figure 3]. Repeat MRI done 14 years back showed no corresponding cavernoma. The temporal relation between the radiotherapy and appearance of cavernoma 14 years later suggests possible causal relation between the two.
|Figure 1: Non contrast coronal (a), sagittal (c) and contrast enhanced coronal (b), sagittal (d) T1 weighted images show a homogenously enhancing mass lesion in the suprachiasmatic region and involving the optic chiasm (arrow)|
Click here to view
|Figure 2: Axial T2 (a) weighted image shows heterogenous hyperintense lesion in the right temporal region (arrow). Gradient axial image (b) shows blooming suggesting cavernoma (arrow). Contrast enhanced Coronal T1 weighted images (c,d) show negligible enhancement within the lesion (arrow). Previous post operative changes are seen in the suprachiasmatic region (arrow head) and left temporal lobe (open arrow)|
Click here to view
|Figure 3: Histopathological examination showing vascular spaces of varying sizes filled with RBCs suggesting cavernous angioma. No evidence of atypia, mitosis or necrosis (H and E, x40)|
Click here to view
The reported prevalence of cavernomas in general population is 0.5-0.6%.  Cavernomas can be sporadic and familial. The familial form accounts for 30% to 50% of cases harboring a cavernous malformation.  These lesions are considered to be congenital, but the lesions can rarely be acquired. Rarely these lesions have been observed in areas of normal brain (on prior MRI) after long years of radiation therapy for other brain lesions. Various hypotheses have been proposed to explain the pathogenesis of RIC: (1) radiation therapy induced capillary wall necrosis leads to endothelial swelling, dilatation of the vessel lumen, hyalinization and fibroses predisposing to cavernoma formation; (2) radiographically occult cavernomas present prior to radiotherapy subsequently grow to radiographically demonstrable cavernomas; (3) radiation triggered release of vascular endothelial growth factor may induce neoangiogenesis;  and (4) possible radiation induced somatic mutagenesis of Krev interaction trapped protein1 (KR1T1) encoded by the CCM1 gene may result in cavernoma formation. 
RIC were predominantly reported in children suffering from medulloblastoma who had received craniospinal radiation and chemotherapy following surgery. Vinchon M et al., retrospectively studied 552 patients irradiated for brain tumor under 18 years of age since 1970 and identified 60 cavernomas.  In an another study, Lew SM et al., identified 26 cavernomas in 59 patients aged 3-21 years irradiated for medulloblastoma.  These studies suggest that the reported worldwide prevalence of RIC is a gross under estimate. The reported prevalence in the study by Burn S et al., was more than 6 times the prevalence rate cited in the literature.  Cerebral cavernomas have been the most frequent MRI abnormality (57%) in 56 childhood leukemia survivors treated with cranial radiotherapy. 
It is important to obtain clinical history pertaining to radiation therapy in radiographically diagnosed patients of cerebral cavernomas. And patients undergoing radiotherapy, regardless of their age, should have regular follow-up with MRI for any RIC. We support the suggestion by Burn S et al., for mentioning RIC as a possible complication when consent for radiation therapy is obtained so that the patients understand and their compliance for long term follow-up is better.  Once the diagnosis of RIC has been confirmed, it should be treated as any other cavernoma since the natural history remains the same for both categories of cavernomas.
| » References|| |
Circillo SF, Cogen PH, Edwards MS. Pediatric cryptic vascular malformations: Presentation, diagnosis and treatment. Pediatr Neurosurg 1994;20:137-47.
Ruggeri AG, Donnarumma P, Pichierri A, Delfini R. Two cystic cavernous angiomas after radiotherapy for atypical meningioma in adult woman: Case report and literature review. J Korean Neurosurg Soc 2014;55:40-2.
Walch J, Tettenborn B, Weber J, Hundsberger T. Radiation-induced cavernoma after total body irradiation and haematopoietic stem cell transplantation in an adult patient suffering from acute myeloid leukemia. Case Rep Neurol 2013;5:91-7.
Quinones-Hinjosa A. Schmidek and sweet operative neurosurgical techniques: indications, methods and results. In: Patel AP, Amin-Hinjani S, Ogilvy CS, editors. Surgical Management of Cavernous Malformations of the Nervous System. 6 th
ed. Vol: 11. Philadelphia: Elsevier Saunders; 2012. p. 977-93.
Vinchon M, Leblond P, Carson S, Delestret I, Baroncini M, Coche B. Radiation-induced tumors in children irradiated for brain tumor: A longitudinal study. Childs Nerv Syst 2011;27:445-53.
Lew SM, Morgan JN, Psaty E, Lefton DR, Allen JC, Abbott R. Cumulative incidence of radiation-induced cavernomas in long-term survivors of medulloblastoma. J Neurosurg 2006;104:103-7.
Burn S, Gunny R, Phipps K, Gaze M, Hayward R. Incidence of cavernoma development in children after radiotherapy for brain tumors J Neurosurg 2007;106:379-83.
Faraci M, Morana G, Bagnasco F, Barra S, Polo P, Hanau G, et al
. Magnetic resonance imaging in childhood leukemia survivors treated with cranial radiotherapy: A cross sectional, single center study. Pediatr Blood Cancer 2011;57:240-6.
[Figure 1], [Figure 2], [Figure 3]