Journal of clinical neuroscience : official journal of the Neurosurgical Society of Australasia · 2026
Epidemiology and treatment strategies of intracranial aneurysms associated with Loeys-Dietz syndrome: A systematic review, meta-analysis, and a case presentation.
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Shimizu Kampei, Tani Shoichi
Аннотация
BACKGROUND: Recent studies have suggested that intracranial aneurysms (IAs) may be a common arterial manifestation of Loeys-Dietz syndrome (LDS). However, the prevalence, characteristics, and treatment strategies remain unclear because of limited and fragmented evidence.
METHODS: We searched PubMed and Scopus through April 2026. Inclusion criteria were studies reporting epidemiological characteristics or surgical interventions of LDS-associated IAs. Studies with high risk of bias according to an adapted Newcastle-Ottawa Scale were excluded from the meta-analysis. Prevalence and risk factors of harboring IAs in patients with LDS were meta-analyzed using a random-effects model. Between-study heterogeneity was assessed by the I2 statistic. The review protocol was registered with PROSPERO (Registration ID: CRD420261374963).
RESULTS: Nine observational studies were eligible for the meta-analysis. The pooled IA prevalence was 23 % (64/282; 95 % confidence interval, 18-28 %) with low across-study heterogeneity (I2 = 6.4 %). Mean aneurysm size was 3.23 mm, and 78.1 % (50/64) of the aneurysms were saccular. Eighty-five percent (63/74) of IAs were located in the anterior circulation or the posterior communicating artery. The prevalence was significantly higher in women than in men (odds ratio, 2.49; 95 % confidence interval, 1.51-4.10, I2 = 0 %). Current smoking and prior arterial dissection were consistently associated with increased odds of harboring IAs (odds ratio > 4) across two studies. No statistically significant differences in IA prevalence were detected across pathogenic variant groups, although statistical power was limited. No rupture events were reported among 62 IAs during mean or median follow-up periods ranging from 2.0 to 8.9 years. Data about the safety and efficacy of surgical interventions were only available through case reports. A systematic review of 11 case reports, including the case presented herein, identified 16 surgically treated LDS-associated IAs. Angiographic occlusion was achieved in all 16 aneurysms, with no major complications clearly attributable to the underlying connective tissue disorder.
CONCLUSIONS: This study highlights the high prevalence and risk factors of IAs among patients with LDS. The available data do not suggest a high rupture rate in small LDS-associated IAs; however, evidence remains insufficient to define their natural history. Current management strategies are largely extrapolated from sporadic IAs and may not fully reflect LDS-specific vascular pathology.
METHODS: We searched PubMed and Scopus through April 2026. Inclusion criteria were studies reporting epidemiological characteristics or surgical interventions of LDS-associated IAs. Studies with high risk of bias according to an adapted Newcastle-Ottawa Scale were excluded from the meta-analysis. Prevalence and risk factors of harboring IAs in patients with LDS were meta-analyzed using a random-effects model. Between-study heterogeneity was assessed by the I2 statistic. The review protocol was registered with PROSPERO (Registration ID: CRD420261374963).
RESULTS: Nine observational studies were eligible for the meta-analysis. The pooled IA prevalence was 23 % (64/282; 95 % confidence interval, 18-28 %) with low across-study heterogeneity (I2 = 6.4 %). Mean aneurysm size was 3.23 mm, and 78.1 % (50/64) of the aneurysms were saccular. Eighty-five percent (63/74) of IAs were located in the anterior circulation or the posterior communicating artery. The prevalence was significantly higher in women than in men (odds ratio, 2.49; 95 % confidence interval, 1.51-4.10, I2 = 0 %). Current smoking and prior arterial dissection were consistently associated with increased odds of harboring IAs (odds ratio > 4) across two studies. No statistically significant differences in IA prevalence were detected across pathogenic variant groups, although statistical power was limited. No rupture events were reported among 62 IAs during mean or median follow-up periods ranging from 2.0 to 8.9 years. Data about the safety and efficacy of surgical interventions were only available through case reports. A systematic review of 11 case reports, including the case presented herein, identified 16 surgically treated LDS-associated IAs. Angiographic occlusion was achieved in all 16 aneurysms, with no major complications clearly attributable to the underlying connective tissue disorder.
CONCLUSIONS: This study highlights the high prevalence and risk factors of IAs among patients with LDS. The available data do not suggest a high rupture rate in small LDS-associated IAs; however, evidence remains insufficient to define their natural history. Current management strategies are largely extrapolated from sporadic IAs and may not fully reflect LDS-specific vascular pathology.