Abstract
Intracranial vertebral artery dissecting aneurysm is a rare type of intracranial aneurysm characterized by outward protrusion of the vessel wall due to damage to the intima and elastic membrane. Aplastic anemia is a relatively rare and severe syndrome involving bone marrow failure leading to pancytopenia. No previously reported cases exist of intracranial vertebral artery dissecting aneurysm complicated by aplastic anemia. Due to thrombocytopenia and coagulation dysfunction, managing concomitant vascular lesions during neurointerventional therapy poses significant challenges. We report the case of a 40-year-old man who with a history of aplastic anemia. Computed tomography angiography revealed a dissecting aneurysm in the right vertebral artery segment IV, with a distal branch to the posterior inferior cerebellar artery. The patient demonstrated favorable outcomes following individualized neurointerventional surgery with no complications. This case underscores the importance of developing personalized neurointerventional strategies for patients with intracranial aneurysms complicated by hematologic disorders. Priority should be given to controlling bleeding risks while maintaining posterior circulation patency, providing crucial guidance for clinicians managing such conditions.
Keywords
Introduction
Intracranial vertebral artery dissecting aneurysm (IVADA) is a rare type of intracranial aneurysm characterized by protrusion of the vascular wall due to damage to the intima and elastic membrane. 1 The annual incidence rate is 1–1.5 cases/100,000 population, making it a major cause of stroke in young and middle-aged adults, resulting in very high morbidity and mortality rates. 2 Due to hemodynamic changes, thromboembolism, or occlusion of perforating arteries, it typically manifests as subarachnoid hemorrhage or ischemic stroke. 3 Aplastic anemia (AA)4,5 is a rare and severe disorder characterized by bone marrow and peripheral blood pancytopenia. Due to thrombocytopenia and coagulation dysfunction, it poses significant challenges in managing associated vascular complications. Although AA has been reported to be associated with cardiac interventions,6,7 no literature has yet documented an association between AA and IVADA. This case report presents the first documented instance of AA coexisting with IVADA. In such scenarios, neurointerventional therapy requires an individualized treatment strategy, prioritizing the balancing of ischemic and hemorrhagic risks. Particular attention must be given to coordinating anticoagulation regimens with procedural planning. This report provides important reference data for endovascular treatment of cases involving intracranial aneurysms complicated by hematologic disorders (Supplemental Material).
We adhered to the Case Report guidelines during the preparation of this manuscript. 8
Case presentation
A 40-year-old male patient presented with recurrent occipital headaches over the past 2 months. Physical examination revealed stable vital signs with no neck pain, nausea, vomiting, or focal neurological deficits. Ten years prior, the patient was diagnosed with AA due to recurrent epistaxis. He had received multiple platelet transfusions for bleeding episodes and was on long-term oral maintenance therapy with cyclosporine (100 mg twice daily) and stanozolol (2 mg twice daily) to sustain platelet counts. These medications have never been discontinued at any point, as sustaining immunosuppressive and hematopoiesis-stimulating therapy was critical to prevent further deterioration of bone marrow function, and they do not inherently exacerbate procedural bleeding risks. Platelet counts remained stable around 25 × 109/L over the past 5 years. No recent transfusions were reported, and there was no relevant family history. At presentation, laboratory findings revealed leukopenia (white blood cells 1.6 × 109/L; normal range 3.5–9.5 × 109/L), anemia (hemoglobin 114 g/L; normal range 130–175 g/L), and thrombocytopenia (platelet count 22 × 109/L; normal range 150–450 × 109/L). Initial coagulation studies were unremarkable, but thromboelastography revealed hypocoagulability (coagulation index −6.9; normal range −3 to 3) and platelet dysfunction (maximum amplitude 29.6 mm; normal range 50–70 mm).
Head computed tomography angiography (CTA; Figure 1(a)) revealed a dissecting aneurysm in the right vertebral artery at the IV segment. The posterior inferior cerebellar artery (PICA) originated distally from the right vertebral artery, adjacent to the vertebrobasilar junction and near the aneurysm. The left vertebral artery showed hypoplasia compared with the right. Subsequent intraoperative digital subtraction angiography (Figure 1(b)) confirmed the right vertebral artery as the primary feeding vessel, with a dissecting aneurysm at the IV segment. The aneurysm neck measured 11.3 mm in width and 2.49 mm in height. Based on these anatomical findings, the patient’s headache symptoms, and the bleeding risk associated with AA, an endovascular intervention was ultimately chosen over conservative management.
(a) Preoperative CTA revealed a dissecting aneurysm in the right vertebral artery at the IV segment (white arrow). The PICA originates from the distal right vertebral artery, adjacent to the vertebrobasilar junction and in close proximity to the aneurysm (red arrow). (b) Intraoperative DSA confirmed the right vertebral artery as the primary feeding vessel, with a dissecting aneurysm in its IV segment (white arrow). The PICA originates adjacent to the vertebrobasilar junction and is close to the aneurysm (red arrow).
Surgical procedure
A multidisciplinary team discussion involving neurology, hematology, anesthesiology, and cardiology optimized management. Following informed consent discussion with the patient and family regarding risks (e.g. bleeding, ischemia), endovascular treatment was pursued. One day prior to the procedure, single-donor platelet apheresis (3 units) was transfused without adverse reactions; posttransfusion complete blood count showed platelet count elevation to 159 × 109/L with normal coagulation. Based on the Chinese expert consensus on perioperative management of thrombocytopenia, the target platelet level for high-risk neuro-interventional procedures should be >80–100 × 109/L. In the morning of the procedure, a repeat complete blood count showed her platelets were stable at 120 × 109/L, well above our target threshold. We determined that performing the procedure the day after transfusion falls within a safe window to ensure adequate platelet levels during the operation. Postoperatively, her platelet count was monitored every other day. As expected, it gradually decreased by ~20 × 109/L per test. She was managed with oral platelet-stimulating agents without needing further transfusions, and her platelets eventually stabilized near her baseline levels. No preoperative antiplatelet therapy was administered. On the procedure day, under general anesthesia with endotracheal intubation, catheter-based intracranial vertebral artery isolation embolization was performed via right radial artery access for embolization and left femoral artery access for intraoperative angiography. During the procedure, catheters were continuously flushed with heparinized saline at 4 U/mL, without intravenous heparin bolus.
The patient was positioned supine on the fluoroscopy table. After satisfactory general anesthesia, the groin and right wrist were prepped with three povidone–iodine applications and draped sterilely. Left femoral artery access was obtained via Seldinger technique with a 5 French sheath; right radial artery with a 6 French sheath. A 6 French intermediate catheter was advanced over a slippery guidewire via radial access to selectively engage the right vertebral artery. Right vertebral angiography (anteroposterior and lateral views) confirmed the IV segment aneurysm with distal PICA opacification. A 4 French diagnostic catheter via femoral sheath, guided by slippery wire, engaged the left vertebral artery for anteroposterior/lateral angiography and three-dimensional rotational reconstruction, demonstrating good left vertebral opacification.
Via radial access, an EV3 Echelon 10 microcatheter (Medtronic Neurovascular, Minneapolis, MN, USA) over Stryker Syncro Soft 014 microwire (Stryker Neurovascular, Fremont, CA, USA) was advanced into the right vertebral artery to the distal end of the aneurysm. A second EV3 Echelon 10 microcatheter (Medtronic) over Syncro Soft 014 microwire (Stryker) accessed the proximal aneurysm sac. Detachable coiling commenced: via the distal microcatheter, an EV3 Axium Prime coil (Medtronic, 7 × 30 mm 3D) was deployed in basket configuration but not detached, as it might shift during subsequent packing, posing a risk. Via the proximal microcatheter, EV3 Axium coils (Medtronic) were sequentially placed (6 × 20 mm 3D × 3, 5 × 15 mm 3D × 1, 4 × 12 mm HX × 2, 3 × 8 mm 3D × 1, 4 × 10 mm 3D × 1) to embolize the aneurysm. Post-embolization right vertebral artery angiography demonstrated delayed blood flow and contrast retention (Figure 2(a)). Left vertebral artery angiography confirmed adequate visualization of the inferior cerebellar artery with no evidence of thrombosis (Figure 2(b)). No complications occurred during the procedure.
(a) Right vertebral artery angiography after coil embolization shows complete occlusion. (b) Left vertebral artery angiography shows patent vessels.
Sheaths were removed post-procedure; radial and femoral sites were bandaged. Head CT showed no intracranial hemorrhage. The patient awoke without distress and returned to the ward. Ceftriaxone (2 g intravenous) was administered for infection prophylaxis.
Outcome and follow-up
Postoperative recovery proceeded smoothly with no episodes of ischemia or hemorrhage. On the day following surgery, the patient reported mild headache but no symptoms of dizziness, vertigo, or limb weakness. On postoperative third day, the patient developed fever (temperature 38.5 °C). White blood cell count and C-reactive protein levels were normal, suggesting an upper respiratory tract infection. After symptomatic treatment with antipyretics and fluid replacement, the fever resolved within 24 h without requiring additional antibiotic therapy. A follow-up cranial CTA on the fourth day post-operation (Figure 3) showed patency of the PICA, compensatory dilation of the left vertebral artery, and no evidence of infarction or hemorrhage. Early CTA confirmed preserved blood flow in the inferior temporal artery and compensatory dilation of the contralateral vertebral artery. By postoperative day 5, the patient’s headache had largely resolved. Multiple complete blood counts during hospitalization demonstrated stable pancytopenia without requiring transfusion. On postoperative day 11, the patient was discharged in good condition and continued cyclosporine and stanozolol therapy for AA, with follow-up scheduled at the outpatient hematology clinic.
Postoperative CTA shows that the right vertebral artery aneurysm is not visualized, and no occlusion of the small cerebellar posterior inferior artery.
At the 1-month outpatient follow-up, the patient was asymptomatic with stable vital signs. The follow-up platelet count remained stable at 28 × 109/L. At the 3-month postoperative follow-up, cranial CTA revealed persistent occlusion of the V4 segment of the right vertebral artery; the PICA was patent; and the left vertebral artery showed compensatory dilation, indicating that blood flow in the posterior circulation was unobstructed.
Discussion
IVADA is rare but carries an extremely high risk of rupture. Endovascular treatment has become the preferred approach due to its efficacy and safety.9,10 Literature11–13 review indicates that transcatheter interventions (including coil embolization or flow diversion) achieve complete occlusion in 80%–95% of cases, with perioperative complication rates of 5%–10%, particularly in unruptured or ruptured IVADAs involving the PICA. There have been no previous reports of IVADA in patients with AA or severe thrombocytopenia, highlighting the uniqueness of this case.
The exact link between AA and IVADA is still unclear. Instead of an underlying connective tissue defect, this association might be driven by endothelial dysfunction. It is well known that severe, prolonged thrombocytopenia deprives the endothelium of essential platelet-derived growth factors, making the blood vessels more fragile.14,15 Moreover, the chronic inflammation typically seen in AA can also damage the endothelial lining. 16 Together, these factors could make the arterial wall more vulnerable to tearing and dissection under normal blood pressure.
In this case, the uncommon association between non-severe AA and IVADA presents unique challenges for surgical decision-making and perioperative management. We summarized the available treatment modalities, highlighting their advantages and disadvantages in Table 1. While reconstructive endovascular techniques (such as stent-assisted coiling or flow diverters) preserve the parent artery, they mandate strict dual antiplatelet therapy (DAPT). In patients with severe thrombocytopenia, DAPT carries an unacceptably high risk of fatal systemic or intracranial hemorrhage. Open neurosurgery is similarly contraindicated due to the extreme risks of perioperative bleeding and infection. Therefore, the endovascular trapping technique with coils emerges as the optimal choice. Its most significant advantage in this scenario is that it provides immediate, definitive prevention of rebleeding without the need for DAPT, provided the patient has adequate collateral circulation from the contralateral vertebral artery. The main disadvantage is the sacrifice of the parent artery, which requires careful preoperative evaluation of the collateral blood flow to avoid brainstem or cerebellar ischemia. Furthermore, thrombocytopenia and platelet dysfunction necessitated preoperative platelet transfusion to mitigate hemorrhagic risk, enabling safe endovascular treatment without antiplatelet therapy, which could exacerbate bleeding in AA patients. 17 We did not use perioperative platelet reactivity unit (PRU) testing in this case, but it could be a useful tool in the future. Checking PRU helps doctors see whether the platelets are actually working properly, giving a better idea of the real bleeding risk and helping tailor transfusion needs. The multidisciplinary approach balanced rupture risk against ischemic potential to the PICA territory, resulting in successful aneurysm isolation and preserved PICA patency. Postoperative complications were minor and self-limited, supporting the feasibility of individualized endovascular treatment in high-risk hematologic conditions.
Comparison between available treatment methods for IVADA.
DAPT: dual antiplatelet therapy; IVADA: intracranial vertebral artery dissecting aneurysm; PICA: posterior inferior cerebellar artery.
The choice of aneurysm isolation embolization (aneurysm and parent artery occlusion via coiling)) over stent-assisted coiling or flow diversion was driven by the need to avoid DAPT, essential for stent-based methods to prevent thrombosis but contraindicated here due to thrombocytopenia (platelet count 22 × 109/L) and bleeding diathesis. DAPT, typically required for 3–6 months post-stenting, carries a heightened risk of hemorrhagic complications—estimated at 2%–5% for major bleeding in coagulopathic patients—which was unacceptable given the patient’s AA-related hypocoagulability.18,19 Literature20,21 supports this strategy in vertebral artery dissecting aneurysm with adequate collateral circulation, as parent artery occlusion achieves comparable occlusion rates to stent-assisted approaches while minimizing such risks, validated by preserved perfusion via the left vertebral artery post-procedure. This aligns with guidelines emphasizing individualized treatment to prioritize safety in comorbidities.22,23
Conclusion
In conclusion, this case report documents the first instance of endovascular treatment for a patient with non-severe AA complicated by IVADA. A personalized approach was established: “preoperative platelet transfusion to elevate the safety threshold + endovascular aneurysm exclusion embolization.” It emphasizes the need for optimized hematologic management and avoidance of routine antiplatelet therapy in patients with thrombocytopenia complicated by neurovascular lesions. The pathogenesis of IVADA may involve hemodynamic stress or connective tissue fragility. Vascular fragility associated with AA may exacerbate this lesion, though the causal relationship remains speculative and warrants further investigation. This report underscores the importance of personalized interventional strategies and meticulous perioperative management, providing a critical reference paradigm for diagnosing and treating such rare conditions while expanding therapeutic approaches for IVADA in specialized hematologic patient populations.
Supplemental Material
sj-pdf-1-sco-10.1177_2050313X261454851 – Supplemental material for Endovascular treatment of intracranial vertebral artery dissecting aneurysm in a patient with aplastic anemia: A case report and literature review
Supplemental material, sj-pdf-1-sco-10.1177_2050313X261454851 for Endovascular treatment of intracranial vertebral artery dissecting aneurysm in a patient with aplastic anemia: A case report and literature review by Hualong Shen, Jinqing Hu, Tiantian Gao, Yaohua Qian, Yuling Cai, Diheng Gu, Minfang Sheng, Lei Hao, Xiangyu Zhang, Yaping Feng and Jinghua Chen in SAGE Open Medical Case Reports
Footnotes
Ethical considerations
Our institution does not require ethics approval for reporting individual cases or case series.
Consent to participate
The participant provided their written informed consent to participate in this study.
Consent for publication
Verbal informed consent was obtained from the patient(s) for their anonymized information to be published in this article.
Author contributions
Hualong Shen: conceptualization, writing—original draft. Jinqing Hu: conceptualization, writing—original draft. Tiantian Gao: methodology, writing—review and editing. Yaohua Qian: supervision, writing—review and editing. Yuling Cai: writing—review and editing. Diheng Gu: writing—review and editing. Minfang Sheng: writing—review and editing. Lei Hao: writing—review and editing. Xiangyu Zhang: writing—review and editing. Yaping Feng: conceptualization, writing—review and editing. Jinghua Chen: conceptualization, writing—review and editing.
Funding
The authors disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This study was funded by the Suzhou Science and Technology Development Plan (SYW2025163) and Taicang Science and Technology Development Plan (TC2023JCYL18).
Declaration of conflicting interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Supplemental material
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References
Supplementary Material
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