Acta paediatrica (Oslo, Norway : 1992) · 2010
Arteriovenous fistula of the vertebral artery in a female infant with hypotonia and cephalocorporal disproportion.
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Núñez F, Martínez-Costa Cecilia, Soler F, Guijarro-Martínez R, Castelló M L, Brines J
Аннотация
BACKGROUND: Congenital arteriovenous fistulas are exceptional in childhood and imply a therapeutic challenge.
CASE REPORT: A 9-month-old female infant was studied for cephalocorporal disproportion, hypotonia, progressive muscular atrophy and hyperreflexia. Computed tomography of the brain and electroencephalography were normal. Electromyographic patterns suggested proximal myopathic involvement. A continuous murmur with systolic reinforcement was audible in the neck. Angioresonance detected intracranial aneurysmal dilatations behind the bulbo-medullary junction and cerebral panangiography evidenced a direct vertebrovertebral fistula with extra- and intra-cranial varices and extreme medullary compression. Occlusion of the afferent vessel to the aneurismal sack was successfully achieved with a mixture of Histoacryl and 75% lipiodol via a microcatheter.
CONCLUSION: Clinical signs of an arteriovenous fistula may be atypical. Considerable cephalocorporal disproportion and a bruit in the cervical or retromastoidal regions must suggest its existence. Complete obliteration with endovascular embolization permits somatic and neurological recovery.
CASE REPORT: A 9-month-old female infant was studied for cephalocorporal disproportion, hypotonia, progressive muscular atrophy and hyperreflexia. Computed tomography of the brain and electroencephalography were normal. Electromyographic patterns suggested proximal myopathic involvement. A continuous murmur with systolic reinforcement was audible in the neck. Angioresonance detected intracranial aneurysmal dilatations behind the bulbo-medullary junction and cerebral panangiography evidenced a direct vertebrovertebral fistula with extra- and intra-cranial varices and extreme medullary compression. Occlusion of the afferent vessel to the aneurismal sack was successfully achieved with a mixture of Histoacryl and 75% lipiodol via a microcatheter.
CONCLUSION: Clinical signs of an arteriovenous fistula may be atypical. Considerable cephalocorporal disproportion and a bruit in the cervical or retromastoidal regions must suggest its existence. Complete obliteration with endovascular embolization permits somatic and neurological recovery.