Abstract
Background: Pure arterial malformations (PAMs) are extremely rare abnormalities defined as dilated, overlapping, and tortuous arteries with a coil-like appearance in the absence of venous components. Over the last half century, only seven published reports have described cases of patients with PAMs who received treatment.Methods: Here, we report two cases of women with PAMs who received surgical treatment, and we present a systematic review of the literature. We searched the PubMed, Embase, Web of Science, and Medline databases (up until October 1, 2021) for relevant publications. We performed independent-sample t-tests and Fisher's exact tests to compare continuous and categorical characteristics among the available cases.Results: Our first patient was a 43-year-old woman with PAM of the left internal carotid artery (ICA), who received an ICA-radial artery (RA)-M2 bypass. Post-operative digital subtraction angiography (DSA) revealed the disappearance of the left ICA PAM without ischemic events during follow-up. The second patient was a 53-year-old woman with PAMs of the right ICA and posterior cerebral artery. The P1 lesion was treated by proximal occlusion combined with a superficial temporal artery-P2 bypass. During the 12-month follow-up period, the size of the PAMs decreased significantly as indicated by the post-operative DSA showing the absence of hemorrhages. Our systematic review, which includes 56 PAMs, shows that the reported PAMs were more common in the anterior circulation (33/56, 58.9%) than in the posterior circulation (11/56, 19.7%). Bilateral PAMs were more likely to affect bilateral anterior cerebral arteries (ACA) (ACAbilateral vs. ACAunilateral: 63.6 vs. 26.2%, p = 0.02). In addition, PAMs involving the anterior circulation were likely to affect multiple arteries (anteriormulti vs. posteriormulti: 30.3 vs. 0%, p = 0.038).Conclusion: We found very few reports on treated PAMs; further studies with large sample sizes and long follow-up periods are required to explore the appropriate treatment strategy for PAMs.
Highlights
Pure arterial malformations (PAMs) are extremely rare abnormalities defined as dilated, overlapping, and tortuous arteries with a coil-like appearance in the absence of any venous component
We searched the electronic PubMed, Embase, Web of Science, and Medline databases using the following search terms: “pure arterial anomalies” OR “pure arterial malformation” OR “dilated” OR “overlapping” OR “tortuous” OR “coil-like” OR “arterial loops” AND “ACA” OR “anterior cerebral artery” OR “MCA” OR “middle cerebral artery” OR “ICA” OR “internal carotid artery” OR “PCA” OR “posterior cerebral artery” OR “PCoA” OR “posterior communicating artery.”
The results of the DWI MRI revealed the absence of an acute ischemic stroke (Figure 1H). These results demonstrated that PAM of the left ICA led to decreased blood flow velocity in the left hemisphere, which caused the right-sided weakness and repeated TIAs in our patient
Summary
Pure arterial malformations (PAMs) are extremely rare abnormalities defined as dilated, overlapping, and tortuous arteries with a coil-like appearance in the absence of any venous component. These lesions are first defined by McLaughlin et al [1] and are often mistaken for arteriovenous malformations (AVMs) or dissecting intracranial aneurysms. We performed a detailed review of the existing literature to better characterize the clinical characteristics of PAMs. Pure arterial malformations (PAMs) are extremely rare abnormalities defined as dilated, overlapping, and tortuous arteries with a coil-like appearance in the absence of venous components. Over the last half century, only seven published reports have described cases of patients with PAMs who received treatment
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