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Journal of Cancer Research and Therapeutics, Vol. 6, No. 3, July-September, 2010, pp. 333-335 Case Report Meningioma and cavernous angioma following childhood radiotherapy Akshay D Baheti1, Amit S Mahore2, Bhooshan P Zade3, Rakesh Jalali3 1 Department of Radiology, King Edward Memorial Hospital & Seth G S Medical College, Mumbai, India Correspondence Address:Akshay D Baheti, Department of Radiology, King Edward Memorial Hospital & Seth G S Medical College, Acahrya Dhonde Marg, Parel, Mumbai - 400 012, India, akshaybaheti@gmail.com Code Number: cr10078 PMID: 21119268 DOI: 10.4103/0973-1482.73348 Abstract Prophylactic cranial irradiation has been a part of multimodality management of acute lymphoblastic leukemia (ALL). With optimum treatment and the resultant long-term cure rates, long-term side effects of radiation including radiation-induced neoplasms have been increasingly unearthed. We report a rare case of development of both a meningioma and a cavernous angioma following prophylactic cranial irradiation as a part of treatment of ALL. Regular follow-up and high index of suspicion for late radiation sequelae after treatment are therefore justifiable in leukemia survivors with history of prophylactic cranial irradiation.Keywords: Cavernous angioma, meningioma, radiation induced neoplasms, radiotherapy Introduction Radiotherapy is an integral part of multimodality management of various malignancies. Prophylactic cranial irradiation has been shown to result in increased survival outcome of children with acute lymphoblastic leukemia (ALL). [1] Although long-term sequelae of cranial prophylaxis have not yet been well defined, we report a rare case of development of both a meningioma and a cavernous angioma following prophylactic cranial irradiation as a part of treatment of ALL. Case Report A 30-year-old right handed male, driver by occupation, presented with one year history of multiple episodes of focal convulsions involving the left upper limb with secondary generalization. He complained of seven-eight such episodes in the past six months. He also gave history of occasional episodes of paraesthesia over the left upper limb since the past six months. There were no other significant associated complaints. He gave past history of acute lymphoblastic leukemia at the age of nine years for which he was treated with chemotherapy. Prophylactic cranial irradiation (PCI) was given as a part of treatment protocol. PCI regimen consisted of radiotherapy to the whole brain with a dose of 18 Gy in 10 fractions over two weeks. In view of the patient′s history, a plain and contrast enhanced MRI of the brain was performed. It showed a well defined, lobulated, extra-axial, right high frontal convexity space occupying lesion. The lesion was isointense to mildly hyperintense to grey matter on T1 weighted images and isointense to grey matter on T2 weighted images. It showed moderate perilesional vasogenic edema. The lesion showed intense homogenous post contrast enhancement with associated enhancement of the adjacent dura [Figure - 1]. In view of these characteristic findings, the lesion was diagnosed as a meningioma. In addition, a well-defined lobulated lesion was also observed in the pons with central T2 hyperintensity and a surrounding complete hemosiderin rim with characteristic blooming on gradient echo sequence suggestive of a cavernous angioma [Figure - 2] and [Figure - 3]. The patient was operated for his high frontal convexity meningioma as it was the symptomatic lesion. The histopathology revealed a transitional meningioma. Patient and relatives did not give consent for removal of cavernous angioma in view of its eloquent location and the potential complications associated with surgical removal of the brainstem cavernoma. Patient is under regular follow-up and is asymptomatic. Discussion Prophylactic cranial irradiation has been a part of multimodality management of acute lymphoblastic leukemias (ALL). [2] With the optimum treatment, the long-term cure rates for the patients with ALL approaches 95-98%. [2] Exposure to ionizing radiation has been implicated as an etiology for brain tumors like meningiomas, gliomas, cavernous angiomas and sarcomas. [1],[3] To sustain a diagnosis of neoplasia induced by radiation, the tumor must occur within the irradiated field and after a latent period sufficient to exclude its having being present at the time of radiotherapy. The tumor should differ histologically from the original lesion and neurocutaneous syndromes predisposing to malignancy must be excluded. [1],[3] The exact mechanism of the role of radiation in carcinogenesis has not been well established in humans and experimental animals. Radiation-induced neoplasms often develop as a result of mutagenic capacity, chromosome aberration and DNA injury caused by the radiation. It is a multi-stage process rather than a single hit process. [1],[3] Radiation injury can also manifest as vasculopathy with hyalinization and fibrinoid necrosis of the vessel walls, resulting in occlusion and infarction as well as vascular proliferative lesions such as capillary telangiectasias and cavernous angiomas. [4],[5],[6] Capillary telangiectasias usually occur three-nine months after irradiation. [7] Cavernous angiomas take a longer time to develop; latency periods ranging from 1 to 26 years, [4] and are more commonly seen to develop in children receiving radiation therapy. [4] Radiation-induced meningiomas have a tendency for aggressive biological behavior as compared to their de-novo counterparts. [2],[8] There is a slight male preponderance, multiple meningiomas often occur, and the recurrence rates are higher. [2],[8] The mean latency period for induction of a meningioma in most series is 18.7 ± 10.2 years. [9] There are no reports that the CT and MRI findings in cases of radiation-induced meningiomas are any different from those for spontaneous meningiomas. Post irradiation cavernous angiomas have certain special features. These have a higher tendency for clinically overt bleeding, [10] although this has not occurred in our case as yet. Histologically, the endothelium lined vascular spaces have only minimal stroma between them, and calcifications and organized thrombi are not seen. [5],[10] MR imaging of the tumor is characteristic, showing a reticulated core of heterogeneous signal intensity with a dark peripheral rim of hemosiderin, giving a ′popcorn-like′ appearance, with characteristic blooming on gradient echo imaging due to magnetic susceptibility effects. [11] Our patient gives a characteristic history of radiation therapy to brain in childhood, which is associated with the development of both meningiomas and cavernous angiomas. The latency period of the detection of the tumors in the presented case correlates with the available data. Conclusion While the prognosis of acute childhood leukemia has improved, long-term survivors may experience the rare late effects of treatment. Regular follow-up and high index of suspicion for late radiation sequelae after treatment are therefore justifiable in cranially irradiated leukemia survivors. References
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