Flow Diversion for Treatment of Growing A2 Aneurysm in a Child: Case Report and Review of Flow Diversion for Intracranial Aneurysms in Pediatric Patients

doi:10.1016/j.wneu.2016.09.078

World Neurosurgery

Volume 96, December 2016, Pages 607.e13-607.e17

https://doi.org/10.1016/j.wneu.2016.09.078 Get rights and content

Background

Intracranial flow diversion has gained increasing popularity since the approval of the Pipeline Embolization Device (PED). Although it is only approved for use in adult patients, the PED has been used to treat aneurysms in pediatric patients. We present the first reported case of the use of a PED in a pediatric patient to treat an unusual fusiform distal anterior cerebral artery aneurysm.

Case Description

A 12-year-old girl presented with new onset seizures and was found to have a distal left anterior cerebral artery aneurysm. Initially, this was managed conservatively, but follow-up imaging performed 4 months after presentation demonstrated enlargement of the aneurysm. The patient underwent endovascular embolization of her aneurysm with PED. This was successfully performed and the patient recovered from the procedure with no neurologic deficits.

Conclusions

Follow-up digital subtraction angiography and magnetic resonance angiography at 6 and 12 months, respectively, showed complete occlusion of the aneurysm. We also reviewed the literature on flow diversion for treatment of pediatric intracranial aneurysms.

Introduction

Intracranial flow diversion has gained increasing popularity since the approval of the Pipeline Embolization Device (PED [Medtronic Neurovascular, Minneapolis, Minnesota, USA]) for treatment of large, giant, or wide-necked aneurysms of the internal carotid artery. At present, the PED system is the sole Food and Drug Administration–approved flow diverter available in the United States; however, several others are under study. The PED consists of a braided mesh cylinder of platinum and cobalt microfilaments that provide 30%–35% metal surface coverage at nominal expansion. This high surface area coverage disrupts blood flow into the aneurysm and promotes aneurysm thrombosis, but it is porous enough to preserve the patency of normal branch vessels.¹

Although it is only approved for use in adult patients, the PED has been used to treat aneurysms in pediatric patients.2, 3 A recent case report has also demonstrated successful embolization of distal anterior circulation aneurysms with the PED.⁴ We present the first reported case, to our knowledge, of the use of a PED in a pediatric patient to treat an unusual fusiform distal anterior cerebral artery (ACA) aneurysm.

Section snippets

History and Examination

A 12-year-old girl presented with a new onset seizure and was found to have an aneurysm on magnetic resonance imaging. A subsequent cerebral angiogram showed a fusiform aneurysm of the left A2 segment of the ACA. A repeat angiogram performed 4 months later showed significant growth in the aneurysm, and she was subsequently referred to our center (Figure 1).

Endovascular Treatment with Flow Diversion

The patient was placed under general anesthesia in the angiography suite. A 6-French Raabe sheath (Cook Medical, Bloomington, Indiana, USA)

Discussion

Pediatric intracranial aneurysms are rare and thought to represent less than 5% of all intracranial aneurysms.⁵ Historically, these aneurysms have been treated with open surgical techniques, such as clip reconstruction, trapping, or vessel occlusion, with or without bypass. Endovascular treatments are generally not Food and Drug Administration approved for usage in pediatric patients, and there is concern as to how endovascular devices will fare in growing cerebral arteries. Kalani et al.⁶

Conclusions

We present an unusual case of a growing fusiform distal ACA aneurysm in a child successfully treated with a PED. Historically, pediatric intracranial aneurysms have been treated surgically. The literature on the treatment of pediatric aneurysms treated with flow diversion shows 19 patients with 21 aneurysms. Overall, 14 out of the 19 patients (74%) showed complete occlusion of their aneurysms with parent vessel preservation. One of the 5 patients with incomplete aneurysm occlusion required

Acknowledgments

The authors thank Andrew J. Gienapp, B.A. (Department of Medical Education, Methodist University Hospital, Memphis, Tennessee, USA and Department of Neurosurgery, University of Tennessee Health Science Center, Memphis, Tennessee, USA) for technical assistance, copyediting, preparation of the manuscript and figures for publishing, and publication assistance with this manuscript.

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Pediatric Intracranial Aneurysms: Favorable Outcomes Despite Rareness and Complexity
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Citation Excerpt :
Flow diversion can be considered an effective treatment option, enabling reconstitution of the structural conformation of the parent vessel.91 It showed some favorable outcomes in pediatric patients96-98 but was limited to relatively few cases. Also, the long-term durability of flow diversion in pediatric patients has not been established.
Pediatric intracranial aneurysms (IAs) are rare and differ from their adult counterparts in terms of their aneurysmal characteristics, presentation, treatment, and outcomes. Their treatment is often more difficult and complex compared with that of adults. However, studies outlining the clinical effect of pediatric IAs remain sparse.
We retrospectively reviewed the data from patients aged ≤18 years admitted to our hospital from 2000 to 2017 with a diagnosis of IAs.
From the sample of 8207 patients with an IA diagnosis, 26 patients with 33 IAs were involved. Our cohort included 17 males and 9 females, with a mean age of 12.5 years. The mean follow-up duration was 4 years and 3 months. Seven patients (26.92%) were assumed to have a traumatic origin for their IAs. Ruptured aneurysms were more common than unruptured ones (61.53% vs. 38.46%). Complex features were observed in 14 aneurysms (42.42%). Initially, microsurgical and endovascular treatment were both performed in 10 patients (38.46%). A good recovery was obtained in 16 patients (61.54%) as determined by the Glasgow outcome scale scores at the 6-month follow-up visits. The complete obliteration of aneurysms was observed in 17 patients (65.38%). Endovascular treatment was the initial treatment in 3 patients with incomplete obliteration.
The treatment of pediatric IAs is challenging and technically demanding owing to their discrete nature compared with adult IAs and the need for greater surgical skills. We found a male predominance, with internal carotid artery bifurcation as the most frequent location of the aneurysms. Despite the greater incidence of ruptured and complex aneurysm cases, many patients had experienced a good recovery at the 6-month follow-up examinations.
Pipeline Embolization Device Flow Diversion for a Dissecting Ruptured Posterior Cerebral Artery Aneurysm in a Pediatric Patient
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Intracranial aneurysms (IAs) are rare in the pediatric population and are usually considered difficult to treat with traditional microsurgery owing to their complex morphology. Endovascular techniques have become the standard option for treating IAs in adults. More recently, flow diverters, such as the Pipeline embolization device (PED), are being widely adopted for unruptured IAs, with proven safety and efficacy in adults; however, their use in the pediatric population is not well defined. Here we report a pediatric patient with a ruptured posterior cerebral artery (PCA) aneurysm successfully treated with a PED, and provide a review of the literature on the current status of PED use in this subset of patients.
A previously healthy 4-year old boy presented to the emergency department with a subarachnoid hemorrhage. Magnetic resonance angiography (MRA) suggested a ruptured dissecting aneurysm in the right PCA. After discussing treatment options with the child's parents, off-label use of the PED device was chosen. A single PED device was successfully deployed within 24 hours of onset. At a 6-month follow-up, the patient was fully recovered, with a modified Rankin Scale score of 0, and MRA showed complete occlusion of the aneurysm and patency of the parent vessel.
Even though the PED has not received Food and Drug Administration approval to treat IAs in children, the literature reports favorable outcomes with this application. Thus, the PED may be a feasible option for treating challenging cases occurring more frequently in the pediatric population. Further studies in pediatric populations are needed to determine whether this technology is a viable and durable option for treating aneurysms in children.
Endovascular Management of Multiple Dysplastic Aneurysms in a Young Man with an Unknown Underlying Cause: A Case Report and Review of the Literature
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Flow diverters were originally introduced as a treatment option for large or fusiform aneurysms of the proximal internal carotid artery. The devices direct flow through the normal vessel lumen, leading to progressive thrombosis of the aneurysmal sac, and also provide a scaffold for endothelial growth and reconstruction of the normal parent vessel contour.21-23 Early success with flow diverters has led to their wider application in the treatment of various aneurysms difficult to treat both microsurgically and through traditional endovascular means.22
Intracranial aneurysms are the leading cause of nontraumatic subarachnoid hemorrhage and are most commonly associated with the anterior cerebral artery (ACA) and anterior communicating artery complex. We describe the presentation and management of a 27-year-old man with concurrent bilateral A1-2 junction aneurysms and fusiform intraorbital ophthalmic artery (OA) aneurysms.
A 27-year-old man with no past medical history presented with 3 months of headaches. Imaging showed a large dysplastic left A1-2 junction aneurysm and a smaller saccular right A1-2 junction aneurysm, with potentially adherent domes. Two fusiform aneurysms of the intraorbital segment of the left OA were also identified. The patient underwent coil-assisted pipeline embolization of the left A1-A2 aneurysm, with complete obliteration and reconstitution of the normal parent vessel. The patient underwent coil embolization of the right A1-2 aneurysm 3 weeks later, which was found to have grown significantly at the time of treatment. Three-month follow-up showed spontaneous resolution of the OA aneurysms, persistent obliteration of the left aneurysm, and significant recurrence of the right aneurysm, which was treated with stent-assisted coil embolization. A second recurrence 3 months later was successfully treated with repeat coiling. At the time of this treatment, the patient was also found to have 2 de novo distal middle cerebral artery and ACA dysplastic aneurysms, which were not treated. Follow-up angiography 6 weeks later showed stable complete obliteration of the right A1-2 aneurysm and interval complete resolution of the dysplastic middle cerebral artery aneurysm. The distal ACA aneurysm was observed to have minimally increased in size; however, the parent vessel showed signs of interval partial thrombosis with contrast stasis within the aneurysm. This final aneurysm is being followed with serial imaging. The patient remains neurologically intact with complete resolution of his headaches.
We report the case of a young man with no past medical history who presented with multiple dysplastic aneurysms. Successful staged endovascular intervention resulted in obliteration of aneurysms with spontaneous obliteration of the intraorbital OA aneurysms observed at 3 months. We present this case to review the multiple challenges of managing complex ACA aneurysms and to highlight the usefulness of endovascular intervention in their treatment.
Multimodal Treatment of Intracranial Aneurysms in Children: Clinical Case Series and Review of the Literature
2018, World Neurosurgery
Citation Excerpt :
Stent-assisted coiling carries a slightly higher thromboembolic risk than primary coiling in addition to the bleeding risk from the requisite postoperative antiplatelet therapy, but it can offer a denser coil packing and lower risk of recurrence. Multiple case reports describe the successful use of flow diversion in children.78-82 However, the long-term durability of flow diversion in growing children is not established.
The management of intracranial aneurysms in the pediatric population presents unique challenges. Cases are rare and tend to be of higher complexity compared with aneurysms in adults. Outcomes in long-term follow-up are not well-characterized. Here we present illustrative case examples to demonstrate key concepts in managing these lesions in the context of the modern neurovascular era.
Four institutional databases of neurovascular procedures from 2012 to 2017 were reviewed. Patients <18 years old who underwent treatment for intracranial aneurysms were included. Patient characteristics, aneurysm details, treatment information, and angiographic and clinical outcomes were recorded.
Ten cases of intracranial aneurysms in 9 children were identified. Management included direct clipping, trapping and bypass, endovascular coil embolization, endovascular vessel sacrifice, and flow diversion.
The management of intracranial aneurysms in pediatric patients requires special considerations, from the diagnostic phase to treatment methods and follow-up regimen. These are ideally considered by a multidisciplinary team, with expertise from pediatric neurosurgeons, cerebrovascular neurosurgeons, and neurointerventionalists.
Endovascular flow diversion
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Coil embolization has been the standard endovascular treatment for intracranial aneurysms (IAs) since the early 1990s. Now, the innovative concept of flow-diverting stents with high-metal coverage and low porosity offers a unique intraluminal approach to IA management. In addition to the commonly used pipeline embolization device (Medtronic), which is in widespread use in the United States, many more devices are available in Europe or Asia. The flow diverters have been well studied in several large trials and registries and have been shown to be effective in the treatment of many types of aneurysms. In this chapter, we review key concepts of flow-diverting devices as well as major studies and discuss expanded indications, complication management, and areas for further study.
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Citation Excerpt :
More frequent follow-up is advised for fusiform and dissecting IAs and IAs treated endovascularly rather than microsurgically.164 DSA is the recommended initial follow-up imaging modality at 6 months for endovascular treatment.1,16,164,165 For patients treated with endovascular therapy with aneurysmal exclusion, Jian et al.164 recommended DSA be performed at 6 months followed by annual MRA, to be decreased to every 5 years.
Pediatric intracranial aneurysms (IAs) are rare. Compared with adult IAs, they are more commonly giant, fusiform, or dissecting. Treatment often proves more complex, and recurrence rate and de novo aneurysmogenesis incidence are higher. A consensus regarding the most appropriate algorithm for following pediatric IAs is lacking.
We sought to generate recommendations based on the reported experience in the literature with pediatric IAs through a thorough review of the PubMed database, discussion with experienced neurointerventionalists, and our own experience.
Digital subtraction angiography (DSA) was utilized immediately post-operatively for microsurgically-clipped and endovascularly-treated IAs, at 6-12 months postoperatively for endovascularly-treated IAs, and in cases of aneurysmal recurrence or de novo aneurysmogenesis discovered by non-invasive imaging modalities. Computed tomographic angiography was the preferred imaging modality for long-term follow-up of microsurgically clipped IAs. Magnetic resonance angiography (MRA) was the preferred modality for following IAs that were untreated, endovascularly-treated, or microsurgically-treated in a manner other than clipping.
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: Conflict of interest statement: J. Vachhani, C. Nickele, and P. Klimo have no financial relationships to disclose. L. Elijovich has served as a consultant for Codman, Medtronic/Covidien, and Stryker. A. S. Arthur has served as a consultant for Codman, Medtronic, Microvention, Penumbra, Sequent, Siemens, and Stryker and has received research support from Siemens and Sequent.

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Case ReportFlow Diversion for Treatment of Growing A2 Aneurysm in a Child: Case Report and Review of Flow Diversion for Intracranial Aneurysms in Pediatric Patients

Background

Case Description

Conclusions

Introduction

Section snippets

History and Examination

Endovascular Treatment with Flow Diversion

Discussion

Conclusions

Acknowledgments

Surg Neurol

Pediatr Neurol

Definitive reconstruction of circumferential, fusiform intracranial aneurysms with the pipeline embolization device

Neurosurgery

Successful endovascular reconstruction of a recurrent giant middle cerebral artery aneurysm with multiple telescoping flow diverters in a pediatric patient

Pediatr Neurosurg

Flow diversion for complex intracranial aneurysms in young children

J Neurosurg Pediatr

Treatment of distal anterior circulation aneurysms with the pipeline embolization device: a US multicenter experience

Neurosurgery

Revascularization and pediatric aneurysm surgery

J Neurosurg Pediatr

Treatment of cerebral aneurysms in children: analysis of the Kids' Inpatient Database

J Neurosurg Pediatr

Pipeline for uncoilable or failed aneurysms: results from a multicenter clinical trial

Radiology

Case Report
Flow Diversion for Treatment of Growing A2 Aneurysm in a Child: Case Report and Review of Flow Diversion for Intracranial Aneurysms in Pediatric Patients