Clinically successful late recanalization of basilar artery occlusion in childhood: What are the odds?: Case report and review of the literature

doi:10.1016/j.jns.2007.03.028

Journal of the Neurological Sciences

Volume 260, Issues 1–2, 15 September 2007, Pages 256-260

https://doi.org/10.1016/j.jns.2007.03.028 Get rights and content

Abstract

Background and purpose

Acute basilar artery occlusion is an infrequent but potentially fatal cause of stroke, both in adults and children. We present our experience with a 6-year-old child and we investigate the rationality for late treatment of acute basilar occlusion in children.

Methods

We report the case of a 6-year-old boy with acute basilar artery occlusion presented with a full blown locked-in syndrome, admitted to the endovascular suite 44 h after the stroke onset, and we review all the reported cases of basilar artery occlusions presented with locked-in syndrome in children.

Results

Six hours following admission the basilar artery was partially recanalized by intra-arterial thrombolysis combined with mechanical clot angioplasty. After 12 h, the patient was awake, oriented, his speech function was fully restored and he had only a mild right hemiparesis that recovered completely after a month.

Conclusions

To our knowledge, this is the first report of complete clinical recovery after delayed (50 h) endovascular recanalization of basilar artery in a child. Intra-arterial thrombolysis combined with cerebral angioplasty, can successfully restore the patency of the basilar artery and the neurologic deficit of children with acute basilar artery occlusion, even after a considerable delay.

Introduction

Acute basilar artery occlusion (ABAO) is an infrequent but potentially fatal cause of stroke, both in adults and children [1]. Locked-in syndrome is a common clinical pattern of this condition and is attributed to the occlusion of the midportion of the basilar artery (BA) causing ischemic damage to the pontine pyramidal tracts [2]. It is characterized by upper motor neuron quadriplegia, paralysis of lower cranial nerves, bilateral paresis of horizontal gaze and anarthria, with preserved consciousness.

Traumatic vertebral artery dissection is one of the most common causes of ABAO in young patients and must be especially suspected in patients presenting with cervical pain preceding the dramatic neurological deterioration [3]. Delay in the diagnosis of ABAO is frequent due mainly to the misleading symptoms and signs and the rarity of this condition [4], [5]. In adults, intra-arterial fibrinolysis is considered as the most effective available treatment to achieve BA recanalization and clinical improvement. In children, this technique has been rarely reported. The aim of this paper it to report the first case of complete clinical recovery after delayed (50 h) endovascular recanalization of the BA in a child and to review the pediatric literature.

Section snippets

Case report

A 6-year-old child presented an episode of sudden loss of consciousness a few minutes after the arrival in his classroom. This initial attack was characterized by an unprovoked shout followed by prolonged “pathological cry,” fall to the ground and “spasms”. The first assessment took place at the regional hospital and involved a neurological examination from a pediatric neurologist. On examination, the patient's eyes were open but there wasn't any reaction to the environmental stimuli, he was

Discussion

In general, the annual incidence of pediatric stroke is estimated at 2.5 cases per 100,000 children [6]. Furthermore, the occlusion of BA in childhood is even rarer [7]. Our review of the literature yielded 14 cases of ABAO presented with locked-in syndrome in children between 4–18 years of age (Table 1).

One of the leading causes of BA occlusion in children is the vertebral artery (VA) dissection [3]. Very often a history of trivial neck or head trauma is identified and this was the case in our

Conclusions

The clinical diagnosis of ABAO in children is often delayed. The reasons for this are the rarity of this condition and the vague, non-specific presenting symptoms. Intra-arterial thrombolysis combined with cerebral angioplasty, can successfully restore the patency of the basilar artery and the neurologic deficit of children with ABAO, even after a considerable delay. To our knowledge, this is the first pediatric report of complete clinical recovery after delayed endovascular recanalization of

References (23)

G.S. Golden et al.
The “locked-in” syndrome in children
J Pediatr
(1976)
S. Chatkupt et al.
Cerebellar infarction in children
Pediatr Neurol
(1987)
Y. Ezaki et al.
Retrospective analysis of neurological outcome after intra-arterial thrombolysis in basilar artery occlusion
Surg Neurol
(2003)
R.E. Nordgren et al.
Seven cases of cerebromedullospinal disconnection. The “locked-in” syndrome
Neurology
(1971)
I. Hasan et al.
Vertebral artery dissection in children: a comprehensive review
Pediatr Neurosurg
(2002)
A.J. Larner
Basilar artery occlusion associated with pathological crying: “Folles larmes prodromiques?”
Neurology
(1998)
F.J. Kirkham
Stroke in childhood
Arch Dis Child
(1999)
B. Voetsch et al.
Basilar artery occlusive disease in the New England Medical Center Posterior Circulation Registry
Arch Neurol
(2004)
J.M. De Bray et al.
Extracranial and intracranial vertebrobasilar dissections: diagnosis and prognosis
J Neurol Neurosurg Psychiatry
(1997)
D.T. Cross et al.
Collateral circulation and outcome after basilar artery thrombolysis
AJNR
(1998)

T.A. Baird et al.

Basilar artery occlusion

Neurocrit Care

(2004)

Cited by (50)

Endovascular thrombectomy for basilar-artery occlusion: A meta-analysis with trial sequential analysis
2023, Heliyon
Basilar-artery occlusion (BAO) usually accounts for devastating neurologic sequelae, poor prognosis, and even death. While endovascular thrombectomy (EVT) is the most successful treatment for anterior circulation stroke with large vessel occlusion, its effectiveness in treating acute BAO is still debatable. Our aim is to compare the efficacy and safety between EVT and conservative medical treatment (CMT) in BAO.
Up until May 2022, relevant literature was gathered using searches in Embase, PubMed, and the Cochrane Library. The primary outcomes were defined as good functional outcome (modified Rankin Scale 0–2) and favorable functional outcome (modified Rankin Scale 0–3) at 3 months between EVT and CMT groups. The secondary outcomes included mortality at 3 months, symptomatic intracerebral hemorrhage (ICH), and any ICH.
Eight studies involving 3733 patients with BAO were enrolled 2573 individuals underwent EVT, and the remaining 1160 patients received CMT. Compared with CMT, EVT achieved more favorable functional outcome (odds ratio (OR) 1.26, 95% CI 1.03–1.55, I² = 54%, p = 0.05) in BAO. The good functional outcome showed a similar tendency (OR 1.23, 95% CI 0.97–1.57, I² = 63%, p = 0.02) as well. EVT decreased mortality at 3 months (OR 0.81, 95% CI 0.70–0.93, I² = 31 %, p = 0.19), although having a tendency to cause symptomatic ICH (OR 2.91, 95% CI 1.38–6.18, I² = 22 %, p = 0.27).
EVT in BAO provides superior functional outcomes and less mortality compared with CMT. Even though EVT has the propensity to cause symptomatic ICH, EVT nevertheless improved posterior circulation stroke.
Acute ischemic stroke endovascular therapy
2021, Handbook of Clinical Neurology
Citation Excerpt :
Similarly, Kumar et al. reported 1.5 times decreased dependency and two times decreased mortality with recanalization (Kumar et al., 2015). Although large variability exists in the time limits for thrombectomy of an acute basilar artery LVO, good clinical outcomes have been demonstrated up to 50 h from LKW (Grigoriadis et al., 2007). Stroke severity at presentation is an independent predictor of patient outcome (Yoon et al., 2015a).
Endovascular therapy (EVT) has become the standard treatment for large-vessel occlusion (LVO) acute ischemic stroke (AIS). EVT is now indicated in patients up to 24 h from their last known well, provided that the patient meets specific clinical and imaging criteria. Improvements in thrombectomy devices, techniques, and operator experience have allowed successful EVT of ICA terminus, M1-MCA occlusions as well as proximal M2-MCA, basilar artery occlusions, and revascularization of tandem lesions. Mechanical thrombectomy failures still occur due to several factors, however, highlighting the need for further device and technical improvements. An ongoing debate exists regarding the need for pre-EVT thrombolytic agents, thrombectomy techniques, distal occlusions, anesthesia methods, the role of advanced neuroimaging, the treatment of patients with larger infarct core, and those presenting with milder stroke symptoms. Many of these questions are the subject of current or upcoming clinical trials. This review aims to provide an outline and discussion about the established recommendations and emerging topics regarding EVT for LVO AIS.
Vertebrobasilar recanalization techniques before the placement of stent-retrievers: Reopening is not synonymous with good outcome
2014, Radiologia
Evaluar la tasa de recanalización y la evolución clínica a los 3 meses tras el tratamiento endovascular de la oclusión vertebrobasilar, antes de la introducción de las endoprótesis recuperadoras («stentrievers»)
Recogida consecutiva de todos los casos de trombosis basilar tratados mediante técnicas endovasculares en nuestro centro. Se revisó de forma retrospectiva su evolución clínica. Nuestro objetivo primario fue evaluar la frecuencia de recanalizaciones alcanzadas, y, como objetivo secundario, valorar la evolución con la escala modificada de Rankin (mRS) a los 3 meses. Valoramos variables clínicas y angiográficas correlacionándolas con la evolución y las complicaciones.
Se recogieron un total de 27 pacientes, con una edad media de 58,1 y desviación estándar de ± 15,5 años, la mediana de la escala National Institutes of Health Stroke Scale (NIHSS) fue 21, rango intercuartílico 18-29, y la mediana de la escala de coma de Glasgow (GCS) fue 7, rango intercuartílico 4-9,5. El tiempo medio y la desviación estándar desde el comienzo del cuadro clínico hasta el tratamiento intraarterial fue de 26,3 ± 41,7 horas. Se consiguió una recanalización parcial o completa en 23 enfermos (85,1%). A los 3 meses 16 pacientes (59,2%) habían fallecido y 6 (22,2%) presentaban una buena evolución (mRS ≤ 2).
El tratamiento endovascular consigue una alta tasa de recanalizaciones en las oclusiones de la arteria basilar. Sin embargo la evolución fue negativa en un porcentaje elevado de enfermos. Es de esperar que el uso de nuevos materiales mejore el pronóstico de estos pacientes.
To evaluate the recanalization rate and clinical outcome three months after endovascular treatment for vertebrobasilar occlusion before the placement of stentrievers.
We reviewed all cases of basilar thrombosis treated with endovascular techniques at our center. We reviewed the clinical outcomes with the main objective of determining the recanalization rate and the secondary objective of evaluating the outcome using the modified Rankin scale (mRS) three months after treatment. We assessed clinical and angiographic variables and correlated them with outcome and complications.
We reviewed a total of 27 consecutive patients (mean age, 58.1 ± 15.5 y; median National Institutes of Health Stroke Scale (NIHSS), 21, interquartile range, 18-29; median Glasgow coma score (GCS) 7, interquartile range, 4-9.5). The mean time between the onset of symptoms and endovascular treatment was 26.3 ± 41.7 hours. Complete or partial recanalization was achieved in 23 (85.1%) patients. Three months after treatment, 16 (59.2%) had died and 6 (22.2%) had good outcome (mRS ≤ 2).
Endovascular treatment achieved a high rate of recanalization of occlusions of the basilar artery. Nevertheless, a high percentage of the patients did not have a good outcome. New materials might improve the prognosis in these patients.
Pediatric ischemic stroke: Acute management and areas of research
2013, Journal of Pediatrics
Citation Excerpt :
One lethal case has been reported,50 but this child suffered from carotid bifurcation occlusion, which also is known to have an unfavorable outcome in adults. In total, 8 children underwent IAT for basilar artery occlusion.51-56 All children survived, 6 with no or minimal deficits after a follow-up ranging from 1 to 12 months.
Endovascular therapy in children with acute ischemic stroke: Review and Recommendations
2012, Neurology
This review provides a summary of the currently available data pertaining to the interventional management of acute ischemic stroke in children. The literature is scarce and is lacking much-needed prospective trials. No study in the literature on the well-established systemic or local thrombolysis trials has included children. Mechanical thrombectomy trials using clot retriever devices have also excluded patients younger than 18 years. The current review is limited to case series of interventional acute ischemic stroke therapy in children and the potential future of endovascular ischemic stroke therapy in this patient population. Recommendations in this review represent the opinion of the authors, based on review of the limited literature covering endovascular acute ischemic stroke therapy in children.
AIS = acute ischemic stroke; IA = intra-arterial; ICA = internal carotid artery; MCA = middle cerebral artery; TIPS = Thrombolysis in Pediatric Stroke; tPA = tissue plasminogen activator
Delayed revascularization in acute ischemic stroke patients
2023, Frontiers in Pharmacology

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Short communicationClinically successful late recanalization of basilar artery occlusion in childhood: What are the odds?: Case report and review of the literature

Abstract

Background and purpose

Methods

Results

Conclusions

Introduction

Section snippets

Case report

Discussion

Conclusions

J Pediatr

Pediatr Neurol

Retrospective analysis of neurological outcome after intra-arterial thrombolysis in basilar artery occlusion

Surg Neurol

Seven cases of cerebromedullospinal disconnection. The “locked-in” syndrome

Neurology

Vertebral artery dissection in children: a comprehensive review

Pediatr Neurosurg

Basilar artery occlusion associated with pathological crying: “Folles larmes prodromiques?”

Neurology

Stroke in childhood

Arch Dis Child

Basilar artery occlusive disease in the New England Medical Center Posterior Circulation Registry

Arch Neurol

Extracranial and intracranial vertebrobasilar dissections: diagnosis and prognosis

J Neurol Neurosurg Psychiatry

Collateral circulation and outcome after basilar artery thrombolysis

AJNR

Basilar artery occlusion

Neurocrit Care

Short communication
Clinically successful late recanalization of basilar artery occlusion in childhood: What are the odds?: Case report and review of the literature