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In contrast to fCCMs, sCCMs are rarely linked to mutations in CCM genes and are instead commonly and peculiarly associated with developmental venous anomalies (DVAs), suggesting distinct origins and common pathogenic mechanisms. Case report: A hemorrhagic sCCM in the right frontal lobe of the brain was surgically excised from a symptomatic 3 year old patient, preserving intact and pervious the associated DVA. MRI follow-up examination performed periodically up to 15 years after neurosurgery intervention demonstrated complete removal of the CCM lesion and no residual or relapse signs. However, 18 years after surgery, the patient experienced acute episodes of paresthesia due to a distant recurrence of a new hemorrhagic CCM lesion located within the same area as the previous one. A new surgical intervention was, therefore, necessary, which was again limited to the CCM without affecting the pre-existing DVA. Subsequent follow-up examination by contrast-enhanced MRI evidenced a persistent pattern of signal-intensity abnormalities in the bed of the DVA, including hyperintense gliotic areas, suggesting chronic inflammatory conditions. Conclusions: This case report highlights the possibility of long-term distant recurrence of hemorrhagic sCCMs associated with a DVA, suggesting that such recurrence is secondary to focal sterile inflammatory conditions generated by the DVA.<\/jats:p>","DOI":"10.3390\/ijms232314643","type":"journal-article","created":{"date-parts":[[2022,11,24]],"date-time":"2022-11-24T02:25:36Z","timestamp":1669256736000},"page":"14643","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Distant Recurrence of a Cerebral Cavernous Malformation in the Vicinity of a Developmental Venous Anomaly: Case Report of Local Oxy-Inflammatory Events"],"prefix":"10.3390","volume":"23","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1014-0731","authenticated-orcid":false,"given":"Andrea","family":"Bianconi","sequence":"first","affiliation":[{"name":"Division of Neurosurgery, Department of Neurosciences \u201cRita Levi Montalcini\u201d, City of Health and Science and University of Turin, 10124 Torino, Italy"},{"name":"CCM Italia Research Network, National Coordination Center, Department of Clinical and Biological Sciences, University of Turin, 10124 Orbassano, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8593-2736","authenticated-orcid":false,"given":"Luca Francesco","family":"Salvati","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, Santa Corona Hospital, 17027 Pietra Ligure, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8512-6080","authenticated-orcid":false,"given":"Andrea","family":"Perrelli","sequence":"additional","affiliation":[{"name":"CCM Italia Research Network, National Coordination Center, Department of Clinical and Biological Sciences, University of Turin, 10124 Orbassano, Italy"},{"name":"Department of Clinical and Biological Sciences, School of Medicine and Surgery, University of Turin, Regione Gonzole 10, 10124 Orbassano, Italy"},{"name":"Department of Pharmacology and Physiology, University of Rochester Medical Center, Rochester, NY 14602, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5432-5386","authenticated-orcid":false,"given":"Chiara","family":"Ferraris","sequence":"additional","affiliation":[{"name":"CCM Italia Research Network, National Coordination Center, Department of Clinical and Biological Sciences, University of Turin, 10124 Orbassano, Italy"},{"name":"Department of Clinical and Biological Sciences, School of Medicine and Surgery, University of Turin, Regione Gonzole 10, 10124 Orbassano, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Armando","family":"Massara","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, Department of Neurosciences \u201cRita Levi Montalcini\u201d, City of Health and Science and University of Turin, 10124 Torino, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4792-7260","authenticated-orcid":false,"given":"Massimiliano","family":"Minardi","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, Department of Neurosciences \u201cRita Levi Montalcini\u201d, City of Health and Science and University of Turin, 10124 Torino, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gelsomina","family":"Aruta","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, Department of Neurosciences \u201cRita Levi Montalcini\u201d, City of Health and Science and University of Turin, 10124 Torino, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3852-0186","authenticated-orcid":false,"given":"Miriam","family":"Rosso","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, Department of Neurosciences \u201cRita Levi Montalcini\u201d, City of Health and Science and University of Turin, 10124 Torino, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Barbara","family":"Massa Micon","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, Department of Neurosciences \u201cRita Levi Montalcini\u201d, City of Health and Science and University of Turin, 10124 Torino, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2692-7133","authenticated-orcid":false,"given":"Diego","family":"Garbossa","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, Department of Neurosciences \u201cRita Levi Montalcini\u201d, City of Health and Science and University of Turin, 10124 Torino, Italy"},{"name":"CCM Italia Research Network, National Coordination Center, Department of Clinical and Biological Sciences, University of Turin, 10124 Orbassano, 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