Purpose. Arteriovenous malformations (AVMs) in the basal ganglia and thalamus have a more aggressive natural history with a higher morbidity and mortality than AVMs in other locations. Optimal treatment—complete obliteration without new neurological deficits—is often challenging. We present a patient with a large bilateral basal ganglia and thalamic AVM successfully treated with hypofractionated stereotactic radiosurgery (HFSRS) with intensity modulated radiotherapy (IMRT). Methods. The patient was treated with hypofractionated stereotactic radiosurgery to 30？Gy at margin in 5 fractions of 9 static fields with a minimultileaf collimator and intensity modulated radiotherapy. Results. At 10 months following treatment, digital subtraction angiography showed complete obliteration of the AVM. Conclusions. Large bilateral thalamic and basal ganglia AVMs can be successfully treated with complete obliteration by HFSRS with IMRT with relatively limited toxicity. Appropriate caution is recommended. 1. Introduction Arteriovenous malformations (AVMs) in the basal ganglia and thalamus comprise a small percentage of all AVMs [1–4]. These deep AVMs have a more aggressive natural history with a higher morbidity and mortality than AVMs in other locations [5]. Bilateral basal ganglia and thalamic AVMs are extremely rare. The majority of patients harboring these lesions present with hemorrhage and serious neurologic sequela. Optimal treatment—complete obliteration without new neurological deficits—is often challenging. Stereotactic radiosurgery (SRS) is generally accepted as the first option in treatment [3, 5, 6]. In this report, we present a patient with a large bilateral basal ganglia and thalamic AVM successfully treated with hypofractionated stereotactic radiosurgery (HFSRS) with intensity modulated radiotherapy (IMRT). There have been no other case reports of bilateral AVMs of this size successfully treated with radiosurgery. In particular, the complete obliteration of the AVMs after HFSRS with one year of treatment is noteworthy. 2. Case Report 2.1. History and Examination A 12-year-old male presented to his primary care physician with a 3-month history of progressive gait abnormality. On physical examination, he had a subtle left facial droop and hyperreflexia in the left lower extremity. His gait was mildly ataxic with circumduction of the left leg. He was unable to heel walk on the left, and he had sidestepping with tandem gait. Toe walking was preserved. His motor and sensory exam was otherwise unremarkable. During his initial hospitalization for further
References
[1]
P. M. Crawford, C. R. West, D. W. Chadwick, and M. D. M. Shaw, “Arteriovenous malformations of the brain: natural history in unoperated patients,” Journal of Neurology Neurosurgery and Psychiatry, vol. 49, no. 1, pp. 1–10, 1986.
[2]
B. A. Gross, E. A. M. Duckworth, C. C. Getch, B. R. Bendok, and H. H. Batjer, “Challenging traditional beliefs: microsurgery for arteriovenous malformations of the basal ganglia and thalamus,” Neurosurgery, vol. 63, no. 3, pp. 393–410, 2008.
[3]
H. Kano, D. Kondziolka, J. C. Flickinger et al., “Stereotactic radiosurgery for arteriovenous malformations, Part 4: management of basal ganglia and thalamus arteriovenous malformations: clinical article,” Journal of Neurosurgery, vol. 116, no. 1, pp. 33–43, 2012.
[4]
C. Stapf, H. Mast, R. R. Sciacca et al., “Predictors of hemorrhage in patients with untreated brain arteriovenous malformation,” Neurology, vol. 66, no. 9, pp. 1350–1355, 2006.
[5]
Y. M. Andrade-Souza, G. Zadeh, D. Scora, M. N. Tsao, and M. L. Schwartz, “Radiosurgery for basal ganglia, internal capsule, and thalamus arteriovenous malformation: clinical outcome,” Neurosurgery, vol. 56, no. 1, pp. 56–63, 2005.
[6]
C. S. Ogilvy, P. E. Stieg, I. Awad et al., “Recommendations for the management of intracranial arteriovenous malformations: a statement for healthcare professionals from a special writing group of the stroke council, American Stroke Association,” Stroke, vol. 32, no. 6, pp. 1458–1471, 2001.
[7]
The Arteriovenous Malformation Study Group, “Arteriovenous malformations of the brain in adults,” New England Journal of Medicine, vol. 340, no. 23, pp. 1812–1818, 1999.
[8]
W. J. Michelsen, “Natural history and pathophysiology of arteriovenous malformations,” Clinical Neurosurgery, vol. 26, pp. 307–313, 1979.
[9]
H. Olivecrona and J. Riives, “Arteriovenous aneurysms of the brain, their diagnosis and treatment,” Archives of Neurology & Psychiatry, vol. 59, no. 5, pp. 567–602, 1948.
[10]
R. Al-Shahi, J. J. Bhattacharya, D. G. Currie et al., “Prospective, population-based detection of intracranial vascular malformations in adults: the Scottish Intracranial Vascular Malformation Study (SIVMS),” Stroke, vol. 34, no. 5, pp. 1163–1169, 2003.
[11]
H. T. ApSimon, H. Reef, R. V. Phadke, and E. A. Popovic, “A population-based study of brain arteriovenous malformation: long-term treatment outcomes,” Stroke, vol. 33, no. 12, pp. 2794–2800, 2002.
[12]
R. D. Brown Jr., D. O. Wiebers, J. C. Torner, and W. M. O'Fallon, “Incidence and prevalence of intracranial vascular malformations in Olmsted County, Minnesota, 1965 to 1992,” Neurology, vol. 46, no. 4, pp. 949–952, 1996.
[13]
J. Hillman, “Population-based analysis of arteriovenous malformation treatment,” Journal of Neurosurgery, vol. 95, no. 4, pp. 633–637, 2001.
[14]
G. A. Jessurun, D. J. Kamphuis, F. H. R. Van der Zande, and J. C. Nossent, “Cerebral arteriovenous malformations in the Netherlands Antilles. High prevalence of hereditary hemorrhagic telangiectasia-related single and multiple cerebral arteriovenous malformations,” Clinical Neurology and Neurosurgery, vol. 95, no. 3, pp. 193–198, 1993.
[15]
R. G. Auger and D. O. Wiebers, “Management of unruptured intracranial arteriovenous malformations: a decision analysis,” Neurosurgery, vol. 30, no. 4, pp. 561–569, 1992.
[16]
R. D. Brown Jr., D. O. Wiebers, G. Forbes et al., “The natural history of unruptured intracranial arteriovenous malformations,” Journal of Neurosurgery, vol. 68, no. 3, pp. 352–357, 1988.
[17]
D. M. Forster, L. Steiner, and S. H？kanson, “Arteriovenous malformations of the brain. A long-term clinical study,” Journal of Neurosurgery, vol. 37, no. 5, pp. 562–570, 1972.
[18]
D. Fults and D. L. Kelly Jr., “Natural history of arteriovenous malformations of the brain: a clinical study,” Neurosurgery, vol. 15, no. 5, pp. 658–662, 1984.
[19]
C. J. Graf, G. E. Perret, and J. C. Torner, “Bleeding from cerebral arteriovenous malformations as part of their natural history,” Journal of Neurosurgery, vol. 58, no. 3, pp. 331–337, 1983.
[20]
H. Kano, D. Kondziolka, J. C. Flickinger et al., “Stereotactic radiosurgery after embolization for arteriovenous malformations,” Progress in Neurological Surgery, vol. 27, pp. 89–96, 2013.
[21]
L. Kihlstr？m, W. Guo, B. Karlsson, C. Lindquist, and M. Lindqvist, “Magnetic resonance imaging of obliterated arteriovenous malformations up to 23 years after radiosurgery,” Journal of Neurosurgery, vol. 86, no. 4, pp. 589–593, 1997.
[22]
H. Mast, J. P. Mohr, A. Osipov et al., “‘Steal’ is an unestablished mechanism for the clinical presentation of cerebral arteriovenous malformations,” Stroke, vol. 26, no. 7, pp. 1215–1220, 1995.
[23]
R. H. Wilkins, “Natural history of intracranial vascular malformations: a review,” Neurosurgery, vol. 16, no. 3, pp. 421–430, 1985.
[24]
I. G. Fleetwood, M. L. Marcellus, R. P. Levy, M. P. Marks, and G. K. Steinberg, “Deep arteriovenous malformations of the basal ganglia and thalamus: natural history,” Journal of Neurosurgery, vol. 98, no. 4, pp. 747–750, 2003.
[25]
T. Sasaki, H. Kurita, I. Saito et al., “Arteriovenous malformations in the basal ganglia and thalamus: management and results in 101 cases,” Journal of Neurosurgery, vol. 88, no. 2, pp. 285–292, 1998.
[26]
C. G. Drake, “Cerebral arteriovenous malformations: considerations for and experience with surgical treatment in 166 cases,” Clinical Neurosurgery, vol. 26, pp. 145–208, 1979.
[27]
T. Matsushima, M. Fukui, K. Kitamura, K. Fujii, and K. Hasuo, “Arteriovenous malformations in the basal ganglia. Surgical indications and approaches,” Neurologia Medico-Chirurgica, vol. 28, no. 1, pp. 49–56, 1988.
[28]
H. Kano, D. Kondziolka, J. C. Flickinger et al., “Stereotactic radiosurgery for arteriovenous malformations, Part 2: management of pediatric patients: clinical article,” Journal of Neurosurgery, vol. 9, no. 1, pp. 1–10, 2012.
[29]
B. E. Pollock, D. A. Gorman, and P. D. Brown, “Radiosurgery for arteriovenous malformations of the basal ganglia, thalamus, and brainstem,” Journal of Neurosurgery, vol. 100, no. 2, pp. 210–214, 2004.
[30]
E. de Oliveira, H. Tedeschi, and J. Raso, “Comprehensive management of arteriovenous malformations,” Neurological Research, vol. 20, no. 8, pp. 673–683, 1998.
[31]
J. M. Tew Jr., A. I. Lewis, K. W. Reichert, B. M. Stein, and R. C. Heros, “Management strategies and surgical techniques for deep-seated supratentorial arteriovenous malformations,” Neurosurgery, vol. 36, no. 6, pp. 1065–1072, 1995.
[32]
R. W. Hurst, A. Berenstein, M. J. Kupersmith, M. Madrid, and E. S. Flamm, “Deep central arteriovenous malformations of the brain: the role of endovascular treatment,” Journal of Neurosurgery, vol. 82, no. 2, pp. 190–195, 1995.
[33]
R. D. Paulsen, G. K. Steinberg, A. M. Norbash, M. L. Marcellus, and M. P. Marks, “Embolization of basal ganglia and thalamic arteriovenous malformations,” Neurosurgery, vol. 44, no. 5, pp. 991–997, 1999.
[34]
B. E. Pollock, “Stereotactic radiosurgery for arteriovenous malformations,” Neurosurgery Clinics of North America, vol. 10, no. 2, pp. 281–290, 1999.
[35]
J. L. Smith and B. Garg, “Treatment of arteriovenous malformations of the brain,” Current Neurology and Neuroscience Reports, vol. 2, no. 1, pp. 44–49, 2002.
[36]
M. S？derman, T. Andersson, B. Karlsson, M. C. Wallace, and G. Edner, “Management of patients with brain arteriovenous malformations,” European Journal of Radiology, vol. 46, no. 3, pp. 195–205, 2003.
[37]
C. S. Ogilvy, “Radiation therapy for arteriovenous malformations: a review,” Neurosurgery, vol. 26, no. 5, pp. 725–735, 1990.
[38]
B. Karlsson, C. Lindquist, and L. Steiner, “Prediction of obliteration after gamma knife surgery for cerebral arteriovenous malformations,” Neurosurgery, vol. 40, no. 3, pp. 425–431, 1997.
[39]
K. Maruyama, N. Kawahara, M. Shin et al., “The risk of hemorrhage after radiosurgery for cerebral arteriovenous malformations,” New England Journal of Medicine, vol. 352, no. 2, pp. 146–153, 2005.
[40]
B. E. Pollock, J. C. Flickinger, L. D. Lunsford, D. J. Bissonette, and D. Kondziolka, “Hemorrhage risk after stereotactic radiosurgery of cerebral arteriovenous malformations,” Neurosurgery, vol. 38, no. 4, pp. 652–661, 1996.
[41]
N. A. Kiran, S. S. Kale, M. K. Kasliwal et al., “Gamma knife radiosurgery for arteriovenous malformations of basal ganglia, thalamus and brainstem—a retrospective study comparing the results with that for AVMs at other intracranial locations,” Acta Neurochirurgica, vol. 151, no. 12, pp. 1575–1582, 2009.
[42]
T. Koga, M. Shin, K. Maruyama, A. Terahara, and N. Saito, “Long-term outcomes of stereotactic radiosurgery for arteriovenous malformations in the thalamus,” Neurosurgery, vol. 67, no. 2, pp. 398–403, 2010.
[43]
A. Nicolato, R. Foroni, A. Crocco et al., “Gamma knife radiosurgery in the management of arteriovenous malformations of the basal ganglia region of the brain,” Minimally Invasive Neurosurgery, vol. 45, no. 4, pp. 211–223, 2002.
[44]
D. I. Friedman, J. Jankovic, and L. A. Rolak, “Arteriovenous malformation presenting as hemidystonia,” Neurology, vol. 36, no. 12, pp. 1590–1593, 1986.
[45]
J. K. Krauss, G. D. Kiriyanthan, and J. J. Borremans, “Cerebral arteriovenous malformations and movement disorders,” Clinical Neurology and Neurosurgery, vol. 101, no. 2, pp. 92–99, 1999.
[46]
H. Kurita, T. Sasaki, I. Suzuki, and T. Kirino, “Basal ganglia arteriovenous malformation presenting as 'writer's cramp',” Child's Nervous System, vol. 14, no. 6, pp. 285–287, 1998.
[47]
S. Papapetropoulos, C. Singer, C. Sengun, and B. V. Gallo, “Giant arteriovenous malformation presenting as pediatric task-specific dystonia,” Neurology, vol. 70, no. 15, p. 1294, 2008.
[48]
M. Yamamoto, M. Hara, M. Ide, Y. Ono, M. Jimbo, and I. Saito, “Radiation-related adverse effects observed on neuro-imaging several years after radiosurgery for cerebral arteriovenous malformations,” Surgical Neurology, vol. 49, no. 4, pp. 385–398, 1998.
[49]
M. Yamamoto, M. Jimbo, M. Hara, I. Saito, and K. Mori, “Gamma knife radiosurgery for arteriovenous malformations: long-term follow-up results focusing on complications occurring more than 5 years after irradiation,” Neurosurgery, vol. 38, no. 5, pp. 906–914, 1996.
[50]
P. Lindvall, P. Bergstr？m, P. L？froth et al., “Hypofractionated conformal stereotactic radiotherapy for arteriovenous malformations,” Neurosurgery, vol. 53, no. 5, pp. 1036–1043, 2003.
