{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,6]],"date-time":"2026-02-06T04:20:32Z","timestamp":1770351632780,"version":"3.49.0"},"reference-count":62,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"7","license":[{"start":{"date-parts":[[2024,2,12]],"date-time":"2024-02-12T00:00:00Z","timestamp":1707696000000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,2,12]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND<\/jats:title>\n                    <jats:p>Complex regional pain syndrome (CRPS) is typically described as a peripheral nerve disorder in which exaggerated allodynia and hyperalgesia follow a minor injury. Some researchers propose a central mechanism, although current evidence is lacking.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>OBSERVATIONS<\/jats:title>\n                    <jats:p>A 14-year-old female presented with classic CRPS symptoms of left upper-extremity weakness and hyperalgesia after a bout of sharp pain in her thumb while shoveling snow. A possible seizure prompted magnetic resonance imaging, revealing a right frontal Spetzler-Martin grade II arteriovenous malformation (AVM) adjacent to the primary motor cortex. Brodmann areas 1, 3a, and 3b, which are responsible for localizing and processing burning and painful sensations, were also involved. The patient underwent transarterial Onyx embolization in two sessions and microsurgical resection, after which her CRPS symptoms completely resolved.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>LESSONS<\/jats:title>\n                    <jats:p>To our knowledge, this is the first reported case of a cerebral AVM presenting as CRPS, which supports a central mechanism. The authors propose that rapid growth of the AVM led to a vascular steal phenomenon of surrounding parenchyma, which disrupted the patient\u2019s normal motor function and nociceptive processing. Further validation in other series is needed.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/case23576","type":"journal-article","created":{"date-parts":[[2024,2,12]],"date-time":"2024-02-12T18:56:15Z","timestamp":1707764175000},"source":"Crossref","is-referenced-by-count":0,"title":["Arteriovenous malformation presenting as complex regional pain syndrome: illustrative case"],"prefix":"10.3171","volume":"7","author":[{"given":"Dayna C","family":"Sloane","sequence":"first","affiliation":[{"name":"Department of Neurological Surgery, Loyola University Stritch School of Medicine, Maywood, Illinois;"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Diego D","family":"Luy","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Loyola University Stritch School of Medicine, Maywood, Illinois;"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Atul K","family":"Mallik","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Loyola University Stritch School of Medicine, Maywood, Illinois;"},{"name":"Department of Neuroradiology, Loyola University Medical Center, Maywood, Illinois; and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Joseph C","family":"Serrone","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Loyola University Stritch School of Medicine, Maywood, Illinois;"},{"name":"Department of Neuroradiology, Loyola University Medical Center, Maywood, Illinois; and"},{"name":"Department of Neurological Surgery, Edward Hines Jr. VA Hospital, Maywood, Illinois"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Douglas E","family":"Anderson","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Loyola University Stritch School of Medicine, Maywood, Illinois;"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1754","reference":[{"issue":"4","key":"b1","series-title":"Acta Neurol Scand","first-page":"262","article-title":"Neurological findings in complex regional pain syndromes\u2014analysis of 145 cases","volume":"101","author":"Birklein F","year":"2000","unstructured":"Birklein F, Riedl B, Sieweke N, Weber M, Neund\u00f6rfer B Neurological findings in complex regional pain syndromes\u2014analysis of 145 cases. Acta Neurol Scand. 2000;101(4):262\u2013269."},{"issue":"2","key":"b2","series-title":"Spinal Cord","first-page":"61","article-title":"Complex regional pain syndrome\u2014diagnostic, mechanisms, CNS involvement and therapy","volume":"41","author":"Wasner G","year":"2003","unstructured":"Wasner G, Schattschneider J, Binder A, Baron R Complex regional pain syndrome\u2014diagnostic, mechanisms, CNS involvement and therapy. Spinal Cord. 2003;41(2):61\u201375."},{"issue":"2","key":"b3","series-title":"Indian J Plast Surg","first-page":"298","article-title":"Complex regional pain syndrome","volume":"44","author":"Sebastin SJ","year":"2011","unstructured":"Sebastin SJ Complex regional pain syndrome. Indian J Plast Surg. 2011;44(2):298\u2013307."},{"issue":"8","key":"b4","series-title":"Pain Med","first-page":"1224","article-title":"A hypothesis for the cause of complex regional pain syndrome-type I (reflex sympathetic dystrophy): pain due to deep-tissue microvascular pathology","volume":"11","author":"Coderre TJ","year":"2010","unstructured":"Coderre TJ, Bennett GJ A hypothesis for the cause of complex regional pain syndrome-type I (reflex sympathetic dystrophy): pain due to deep-tissue microvascular pathology. Pain Med. 2010;11(8):1224\u20131238."},{"issue":"11","key":"b5","series-title":"J Pain","first-page":"1270","article-title":"Primary motor cortex function in complex regional pain syndrome: a systematic review and meta-analysis","volume":"14","author":"Di Pietro F","year":"2013","unstructured":"Di Pietro F, McAuley JH, Parkitny L,  Primary motor cortex function in complex regional pain syndrome: a systematic review and meta-analysis. 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Khirurgiia (Mosk). 2019;6:111\u2013116."},{"issue":"6","key":"b8","series-title":"J Vasc Access","first-page":"1535","article-title":"Complex regional pain syndrome following vascular access creation for hemodialysis","volume":"24","author":"Sathiavageesan S","year":"2023","unstructured":"Sathiavageesan S, Annamalai I, Karki A, Kamalanathan M, Annadurai A, Chandrasekaran A Complex regional pain syndrome following vascular access creation for hemodialysis. J Vasc Access. 2023;24(6):1535\u20131537."},{"issue":"4","key":"b9","series-title":"Hemodial Int","first-page":"344","article-title":"Reflex sympathetic dystrophy syndrome due to arteriovenous fistula","volume":"9","author":"Unek IT","year":"2005","unstructured":"Unek IT, Birlik M, Cavdar C,  Reflex sympathetic dystrophy syndrome due to arteriovenous fistula. Hemodial Int. 2005;9(4):344\u2013348."},{"issue":"4","key":"b10","series-title":"Eura Medicophys","first-page":"283","article-title":"Shoulder-hand syndrome after stroke. A complex regional pain syndrome","volume":"41","author":"Pertoldi S","year":"2005","unstructured":"Pertoldi S, Di Benedetto P Shoulder-hand syndrome after stroke. A complex regional pain syndrome. Eura Medicophys. 2005;41(4):283\u2013292."},{"issue":"2-3","key":"b11","series-title":"Pain","first-page":"313","article-title":"A case of reflex sympathetic dystrophy (complex regional pain syndrome, type I) resolved by cerebral contusion","volume":"79","author":"Shibata M","year":"1999","unstructured":"Shibata M, Nakao K, Galer BS, Shimizu T, Taniguchi H, Uchida T A case of reflex sympathetic dystrophy (complex regional pain syndrome, type I) resolved by cerebral contusion. Pain. 1999;79(2-3):313\u2013315."},{"issue":"2","key":"b12","series-title":"J Neurosurg","first-page":"167","article-title":"Red cerebral veins and the cerebral steal syndrome. 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Neurosurg Focus. 2017;43(VideoSuppl1):V3."},{"issue":"4","key":"b24","series-title":"J Neurosurg","first-page":"929","article-title":"Fatal rupture of a brain arteriovenous malformation flow-related aneurysm during microcatheter removal: a rare complication","volume":"122","author":"Gabrieli J","year":"2015","unstructured":"Gabrieli J, Claren\u00e7on F, Di Maria F,  Fatal rupture of a brain arteriovenous malformation flow-related aneurysm during microcatheter removal: a rare complication. 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