Cerebral proliferative angiopathy depicted by four-dimensional computed tomographic angiography: A case report

Ito, Shuichi; Kanagaki, Mitsunori; Yoshimoto, Naoya; Hijikata, Yoichiro; Shimizu, Mineo; Kimura, Hiroyuki

doi:10.1016/j.radcr.2022.03.104

Cited by 2 publications

(2 citation statements)

References 19 publications

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“…CPA is a rare phenomenon characterized by diffuse abnormal vessels within the brain parenchyma, supplied by several arteries and drained to several veins, without the presence of high-flow arteriovenous shunts. 4 This phenomenon was commonly observed among children and young adults with the mean age at presentation being 22-23 years and female predominance (60-67%). 1,2 Lasjaunias et al (2008) emphasized that the presence of non-focal angiogenetic activity was a major feature of CPA, distinguishing it from "classical" cerebral AVMs.…”

Section: Discussionmentioning

confidence: 98%

“…2 Based on a prior study, CPA was identified in 2-4 % of patients initially diagnosed with cerebral AVM. 3,4 The relatively benign course of disease progression and the lack of distinctive clinical feature may cause CPA to be overlooked and misdiagnosed. 5 Based on the literature search conducted, there were no case reports of CPA from Indonesia.…”

Section: Introductionmentioning

confidence: 99%

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Cerebral Proliferative Angiopathy as the Cause of Symptomatic Epilepsy in a Young Adult Male: A First Case Report from Indonesia

Swatan,

Sani,

Islamiyah

et al. 2024

magna-neurologica

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Background: Cerebral proliferative angiopathy (CPA) is a rare and distinct vascular malformation that was once considered a subset of cerebral arteriovenous malformation (AVM). Due to its relatively benign course with no distinctive clinical feature, CPA may often be overlooked and misdiagnosed with other diseases. We report a case of CPA as the underlying cause of symptomatic epilepsy. Case: A 31-year-old male presented to the outpatient clinic with a history of focal to bilateral tonic-clonic seizure for 2 years. Following conservative management with an oral antiepileptic agent, the seizure frequency significantly decreased from once daily to once or twice monthly. The patient was lost to follow-up; however, he was incidentally referred back to our clinic two years later for further evaluation. A head Magnetic Resonance Imaging and Magnetic Resonance Angiography revealed a suspicion of giant AVM in the left hemisphere. Cerebral digital subtraction angiography (DSA) was performed and revealed a CPA in the left frontal area. The patient was managed conservatively and during the 6-month follow-up period, the patient did not have any seizures. Discussion: In young adults, seizures may be caused by an underlying vascular abnormality. Cerebral DSA remained the gold standard for distinguishing various etiologies of vascular malformation, including CPA. Conservative treatment using oral antiepileptic agents was effective in controlling the seizure frequency in CPA. However, a complete diagnostic evaluation is still warranted to determine the most appropriate treatment, revealing some peculiar and unexpected etiologies in the process.

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Section: Discussionmentioning

confidence: 98%

Section: Introductionmentioning

confidence: 99%

Cerebral Proliferative Angiopathy as the Cause of Symptomatic Epilepsy in a Young Adult Male: A First Case Report from Indonesia

Swatan,

Sani,

Islamiyah

et al. 2024

magna-neurologica

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Proliferative Angiopathy: A Systematic Review

Brown,

Lien,

Ehresman

et al. 2024

SVIN

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BACKGROUND First described over 4 decades ago as a diffuse form of arteriovenous malformation, cerebral proliferative angiopathy (CPA) is now categorized as a distinct vascular anomaly. Unlike arteriovenous malformation, which typically forms a well‐developed vascular nidus with feeder vessels and distinct venous outflow, CPA is the product of disorganized angiogenesis; thus, it lacks a true nidus. Its characteristic cycle of ischemia, angiogenesis, and aberrant perfusion can lead to abnormal blood flow patterns that characterize the disease. Treatment of CPA has historically relied on conservative management (antiepileptic drugs), and efforts to effectively manage this pathology have been hindered by an incomplete understanding of its natural history. METHODS A systematic search of 4 databases was performed. The following variables were extracted when present: study sample size, year of publication, age(s) of patient(s), sex, presenting signs and symptoms, neurological deficits (pre‐ and postoperative), type of intervention, average follow‐up time, and patient‐reported as well as functional outcomes at last follow‐up. RESULTS Following the search and screen, 48 studies reporting 105 CPA cases remained eligible for inclusion. These studies consisted of 41 case reports, 6 case series, and 1 retrospective cohort study. The most common modality used in treatment of CPA was conservative management (53.4%). The second most common treatment modality was endovascular embolization (36.9%). The least common treatment modality was gamma knife radiosurgery, which was used in only 2 patients. Eleven patients experienced intracranial hemorrhage as a complication of CPA; 10 of the 11 patients underwent decompressive craniectomy. Finally, revascularization surgery was reportedly used in 5 patients (4.9%). CONCLUSION Although there remains a dearth of studies reporting CPA, several clearly defined traits have been identified that characterize this pathology and distinguish it from arteriovenous malformation. Nonetheless, the verdict has not yet been reached regarding the effectiveness of surgical interventions.

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Cerebral proliferative angiopathy depicted by four-dimensional computed tomographic angiography: A case report

Cited by 2 publications

References 19 publications

Cerebral Proliferative Angiopathy as the Cause of Symptomatic Epilepsy in a Young Adult Male: A First Case Report from Indonesia

Cerebral Proliferative Angiopathy as the Cause of Symptomatic Epilepsy in a Young Adult Male: A First Case Report from Indonesia

Proliferative Angiopathy: A Systematic Review

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