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A Case of Ruptured Cerebral Arteriovenous Malformation Associated with Hereditary Hemorrhagic Telangiectasia Katsuhiro GOTO 1 , Yoichiro KAWAMURA 1 , Ataru NISHIMURA 1 , Koichi ARIMURA 1 , Masahiro MIZOGUCHI 1 , Koji IIHARA 1 1Department of Neurosurgery, Graduate School of Medical Sciences, Kyushu University Keyword: hereditary hemorrhagic telangiectasia , arteriovenous malformation , ruptured arteriovenous malformation , pediatric arteriovenous malformation pp.1281-1287
Published Date 2019/12/10
DOI https://doi.org/10.11477/mf.1436204116
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 BACKGROUND:Patients with hereditary hemorrhagic telangiectasia(HHT)are known to have high rates of cerebral arteriovenous malformations(AVMs). Compared to patients with sporadic AVMs, patients with HHT are less likely to present with ruptured AVMs.

 CASE REPORT:A 14-year-old male patient presented with headache that had lasted for 2 days. CT revealed an intracerebral hemorrhage in the right parietal lobe, and enhanced CT revealed an AVM in the upper part of the hematoma. The size of the nidus was 20 mm, and its feeders were the right superior internal parietal artery and a branch of the anterior cerebral artery. In addition, the AVM had no deep drainer. We also found another AVM in the right temporal lobe and identified telangiectasia of the nose using digital subtraction angiography. We suspected HHT and performed whole body CT, which revealed an arteriovenous fistula in the right lung and a hematoma-like lesion in the spleen. Thus, we diagnosed the patient with HHT. His ruptured AVM was removed electively.

 CONCLUSION:We report a case of HHT that presented as an intracerebral hemorrhage in a patient. Based on our case study findings, it is necessary to perform long-term follow-up not only for brain AVMs but also for visceral vascular malformations in such patients, as well as perform HHT screening for families. Although such cases are rare, some features of HHT must be considered to accurately diagnose suspected HHT.


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電子版ISSN 1882-1251 印刷版ISSN 0301-2603 医学書院

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