Elsevier

Surgical Neurology

Volume 63, Issue 5, May 2005, Pages 424-432
Surgical Neurology

Aneurysm-Pediatric
Intracranial aneurysms in the pediatric population: case series and literature review

https://doi.org/10.1016/j.surneu.2004.11.023Get rights and content

Abstract

Background

Intracranial aneurysms in children (≤18 years old) are rare, and their epidemiology is poorly understood. We present a prospective 14-year experience at our institution and review the literature to clarify the characteristics and outcomes of pediatric patients with intracranial aneurysms.

Methods

Review of medical records in the Johns Hopkins aneurysm prospective database and review of the literature since 1939 were performed.

Results

Of 1377 intracranial aneurysm cases at our institution from 1991 to 2004, 19 (1.4%) pediatric patients were treated using microsurgical or endovascular techniques. Male/female ratio was 2.2:1. Eleven percent occurred at the internal carotid artery (ICA) bifurcation, and 42% were located in the posterior circulation. Seven (37%) were giant lesions; 58% presented with subarachnoid hemorrhage. Patients in good Hunt and Hess grade (I-III) comprised 42%, and poor-grade (IV-V) patients comprised 16%. At a mean follow-up duration of 3.0 ± 3 years, 95% of patients demonstrated favorable outcomes with Glasgow Outcome Scale score of 4 or 5. In the literature, a total of 706 pediatric intracranial aneurysm cases have been described since 1939. The male/female ratio is 1.8:1. The ICA bifurcation was the location in 26%, and only 17% were in the posterior circulation. Twenty percent were giant lesions, and 80% presented with subarachnoid hemorrhage. Good- and poor-grade patients comprise 49% and 36% of the total cases, respectively. Favorable outcome was reported in 60%.

Conclusions

During recent decades, good outcomes after the treatment of ruptured and unruptured pediatric aneurysms have increased, reaching 95% in the current series. Pediatric intracranial aneurysms occur more commonly in male patients and have a predilection for the terminal ICA bifurcation.

Introduction

Intracranial aneurysms are rare in the pediatric population, defined as patients 18 years old or younger, with a reported prevalence ranging from 0.5% to 4.6% [22], [48], [54], [56], [61], [65]. Only 41 of 6368 ruptured aneurysms were found in patients younger than 19 years in the Cooperative Study reported in 1966, with an incidence of only 0.6% [44]. In a multicenter series consisting entirely of children treated after 1986, they comprise 1% of all surgically aneurysms treated at 3 institutions in France during the same period [59]. It is estimated that most academic neurosurgical centers treat one pediatric aneurysm each year [51].

It appears that several epidemiological features of aneurysms occurring in young people make them unique in comparison to the adult counterpart, such as a predilection for male patients, location at the internal carotid bifurcation, and association with other diseases. Controversy remains as to whether relatively greater proportions of pediatric aneurysms are giant (>25 mm) or occur in the posterior circulation as compared with adult aneurysms. Discrepancies exist not only in the characterization of their features but also with respect to clinical outcome, with good results ranging from 40% to 92% [2], [3], [26], [27], [31], [48], [55]. This is caused by the fact that the case series in the literature include patients treated over the course of the previous half-century, with heterogeneous reporting of outcomes and length of follow-up.

We report the experience at our institution with 19 cases treated using contemporary microsurgical and endovascular techniques since 1991 and summarize all previously reported cases to more accurately describe this rare clinicopathological entity.

Section snippets

Patients and methods

Between 1991 and 2004, 19 (1.4%) of a total of 1377 cerebral aneurysm cases treated at the Johns Hopkins Hospital and the Johns Hopkins Bayview Medical Center occurred in patients aged 18 years or younger. Review of a prospectively accrued clinical database and hospital records was performed to identify and characterize these young patients with the diagnosis of intracranial aneurysm.

Radiographic studies, operative reports, and hospital records were retrospectively reviewed for each patient (

Patient characteristics

Nineteen intracranial aneurysms were treated in 19 patients (age range, 1 month to 18 years, mean 9.8 ± 5.7 years, Table 1). Only 3 patients were younger than 1 year, 8 were children between the ages of 5 and 12 years, and the remaining 8 patients were adolescents from 13 to 18 years old. Thirteen patients were boys, and 6 were girls, resulting in a male/female predominance of 2.2:1.

Presentation and associated medical conditions

Presentation with SAH occurred in 11 (58%) patients. According to clinical criteria defined by Hunt and Hess, 7

Historical perspective

The first definitive account of an intracranial aneurysm was published by Biumi of Milan in 1765 and later reprinted by Sandifort at Leyden in 1778. In this case, an ICA aneurysm was found at necropsy in a woman of advanced age who had severe rheumatic attacks. Many years later in his work entitled “Dropsy,” Blackhall reported a basilar tip aneurysm in a woman who developed sudden coma and death in 1814. In 1834, Nebel reported the first aneurysm series of 13 cases. This was followed by a

Conclusions

Intracranial aneurysms that occur in infancy, childhood, and adolescence are rare and account for less than 2% of all cerebral aneurysms. Compilation of the reports published since 1939 allows for the following conclusions: (1) major improvements in clinical outcomes have been achieved with contemporary microsurgical and endovascular techniques, (2) there is a male/female preponderance of 1.8:1, (3) SAH is the most common mode of presentation, (4) a quarter of all pediatric aneurysms occur at

References (77)

  • H. Arai et al.

    Intracranial aneurysm in an infant

    Surg. Neurol.

    (1973)
  • S.S. Baeesa et al.

    Unusual association of intractable temporal lobe seizures and intracranial aneurysms in an adolescent: is it a coincidence?

    Pediatr. Neurosurg.

    (1998)
  • S. Batnitzky et al.

    Infantile and juvenile cerebral aneurysms

    Neuroradiology

    (1978)
  • Brinton W. Die Krankheiten des Magens nebest einer anatomisch-physiologischen Einleitung. Verlesungen gehalten im St....
  • E. Bull

    Acute brain aneurysm, occulomotor palsy, meningeal apoplexy

    Norsk. Mag. Laegeridenski.

    (1877)
  • M.J. Burke

    Occult aneurysmal hemorrhage in a child. Case report and literature review

    Pediatr. Neurosurg.

    (2000)
  • R.W. Byard et al.

    Sudden and unexpected death due to hemorrhage from occult central nervous system lesions. A pediatric autopsy study

    Pediatr. Neurosurg.

    (1991)
  • V.L. Chiang et al.

    Routine intraoperative angiography during aneurysm surgery

    J. Neurosurg.

    (2002)
  • J.Y. Chun et al.

    Current multimodality management of infectious intracranial aneurysms

    Neurosurgery

    (2001)
  • K.A. Diab et al.

    Cerebral mycotic aneurysm in a child with Down's syndrome: a unique association

    J. Child Neurol.

    (2001)
  • A. Dorfler et al.

    Endovascular treatment of a giant aneurysm of the internal carotid artery in a child with visual loss: case report

    Neuropediatrics

    (2000)
  • R.A. Dudley et al.

    Selective referral to high-volume hospitals: estimating potentially avoidable deaths

    JAMA

    (2000)
  • H. Eppinger

    Histogenesis und aetiologie der aneurysmen einschliesslich des aneurysma equi verminosum

    Arch. Klin. Chir.

    (1887)
  • H. Eppinger

    Stenosis aortae congenita seu isthmus persistens

    Vjschr. prakt. Heilk.

    (1871)
  • F.M. Forster et al.

    Aneurysm of circle of Willis associated with congenital polycystic disease of the kidneys

    Arch. Neurol. Psych.

    (1943)
  • C. Garcia-Chavez et al.

    Cerebral artery aneurysm in infancy: association with agenesis of the corpus callosum

    J. Neuropathol. Exp. Neurol.

    (1965)
  • M. Gerosa et al.

    Intracranial aneurysms of childhood

    Childs Brain

    (1980)
  • R.J. Gewirtz et al.

    Fusiform P1 segment artery aneurysm in a pediatric patient: technical case report

    Pediatr. Neurosurg.

    (1998)
  • S. Ghosh et al.

    Intraoperative angiography in the management of pediatric vascular disorders

    Pediatr. Neurosurg.

    (1999)
  • K.A. Greene et al.

    Cardiopulmonary bypass, hypothermic circulatory arrest and barbiturate cerebral protection for the treatment of giant vertebrobasilar aneurysms in children

    Pediatr. Neurosurg.

    (1994)
  • O. Heiskanen et al.

    Intracranial arterial aneurysms in children and adolescents

    Acta Neurochir. (Wien)

    (1981)
  • J.M. Herman et al.

    Pediatric intracranial aneurysms: simple and complex cases

    Pediatr. Neurosurg.

    (1991)
  • K. Hermann et al.

    Cerebral haemorrhage from rupture of a congenital intracerebral aneurysm in a child

    BMJ

    (1940)
  • L.T. Huang et al.

    Posterior cerebral artery aneurysm in a two-year-old girl

    J. Formos. Med. Assoc.

    (1996)
  • S. Hulsmann et al.

    Management of a ruptured cerebral aneurysm in infancy. Report of a case of a ten-month-old boy

    Neurosurg. Rev.

    (1998)
  • R.P. Humphreys et al.

    Childhood aneurysms—atypical features, atypical management

    Concepts Pediatr. Neurosurg.

    (1985)
  • I. Iob et al.

    Traumatic cerebral aneurysm in pediatric age. Case report

    J. Neurosurg. Sci.

    (1983)
  • J.A. Jane

    A large aneurysm of the posterior inferior cerebellar artery in a 1-year-old

    J. Neurosurg.

    (1961)
  • Cited by (192)

    • Reported incidence and treatment modalities of giant cerebral aneurysms in the pediatric population: A systematic review and illustrative case report

      2021, Journal of Clinical Neuroscience
      Citation Excerpt :

      After screening by abstract and title, 68 studies were accessed as full-text and ultimately 33 met inclusion criteria. Eleven of the included studies were case series or reported more than one patient [1,4,13–21]. The remaining 22 studies were reports of single cases [22–43].

    • A case report on dissecting giant middle cerebral artery aneurysm in a six years old patient: Combined approach

      2021, Interdisciplinary Neurosurgery: Advanced Techniques and Case Management
    View all citing articles on Scopus
    View full text