Cerebral vasculopathy and neurologic sequelae in infants with cervicofacial hemangioma: report of eight patients.

Burrows, Patricia E.; Robertson, Richard L.; Mulliken, JB; Beardsley, Diana S.; Chaloupka, John C.; Ezekowitz, R. A. B.; Scott, R. Michael

doi:10.1148/radiology.207.3.9609880

Cited by 157 publications

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“…Contrast-enhancing masses involving the cerebellopontine angles, cerebellar meninges, hypothalamus, and perichiasmatic cisterns have also been described and are presumed to represent intracranial hemangiomas, since their growth and involu- tion parallel that of the accompanying extracranial hemangiomas (Fig 13) (33). Specific arterial abnormalities include persistent primitive fetal arteries-most commonly a persistent trigeminal artery-and agenesis of the internal carotid or vertebral arteries, anomalies that are more commonly seen ipsilateral to the hemangioma (34). Progressive bilateral vasculopathy with stenoses, aneurysm formation about the vessels of the circle of Willis, and a moya-moya type of collateral vessel formation have also been noted (33,34).…”

Section: Radiographicsmentioning

confidence: 99%

“…Specific arterial abnormalities include persistent primitive fetal arteries-most commonly a persistent trigeminal artery-and agenesis of the internal carotid or vertebral arteries, anomalies that are more commonly seen ipsilateral to the hemangioma (34). Progressive bilateral vasculopathy with stenoses, aneurysm formation about the vessels of the circle of Willis, and a moya-moya type of collateral vessel formation have also been noted (33,34).…”

Section: Radiographicsmentioning

confidence: 99%

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Lumps and Bumps on the Head in Children: Use of CT and MR Imaging in Solving the Clinical Diagnostic Dilemma

et al. 2004

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Section: Radiographicsmentioning

confidence: 99%

Section: Radiographicsmentioning

confidence: 99%

Lumps and Bumps on the Head in Children: Use of CT and MR Imaging in Solving the Clinical Diagnostic Dilemma

et al. 2004

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“…Magnetic resonance imaging (MRI) with angiography of the head and neck region is usually indicated in such infants. [6][7][8] Laryngeal involvement can occur if haemangiomas are present on the cervicofacial, mandibular or the 'beard' distribution. Affected infants should be watched for signs and symptoms of airway obstruction (stridor, hoarseness) and referred for a laryngeal examination.…”

Section: Methodsmentioning

confidence: 99%

Pulse steroid therapy in infantile facial hemangiomas: the early experience

et al. 2018

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Background: To investigate the efficacy and safety of Pulse steroid therapy in the management of Infantile facial hemangioma in five subjects.Methods: Consecutive patients who presented with facial hemangioma were prospectively enrolled in this study between January 2014 till August 2017. All subjects underwent treatment with 30mg/kg/day I/V methyl prednisolone for 6days and then weekly for 6weeks and then followed by oral daily dosing. All subjects underwent systemic, and radiologic evaluations before treatment and at periodic intervals after starting therapy. Side effects from therapy were also evaluated.Results: Five subjects under 6 months of age, with facial hemangioma were enrolled in this study. After hospital admission, I/V steroid therapy was initiated in all subjects under monitoring by a pediatric cardiologist. Subsequent oral therapy after 6 weeks was performed with periodic out-patient monitoring. All subjects had excellent response to treatment, with regression of facial hemangioma. There were no side effects from therapy.Conclusions: I/V methyl prednisolone pulse steroid therapy for facial hemangioma was effective in all the five subjects.

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“…Little literature exists describing in detail treatment options for progressive steno-occlusive cerebrovascular disease in PHACE syndrome with, to our knowledge, only 1 prior report mentioning pial synangiosis. 5 Thus personal communication was also sought from other centers (Dr. M. Scott and Dr. E. Smith, Boston Children's Hospital, and Dr. P. Dirks, The Hospital for Sick Children [SickKids], Toronto). A staged bilateral pial synangiosis was planned for 1 month poststroke, allowing the patient an initial period of recovery from the stroke.…”

Section: Surgical Treatmentmentioning

confidence: 99%

“…The patient was just under 18 months old at this time, in keeping with acute neurological deterioration documented in patients with PHACE syndrome. 5 The patient was found to have a dense left hemiparesis and decreased level of consciousness. An emergent CT followed by MRI of the head revealed an infarct in the right frontal and parietal lobes ( Fig.…”

mentioning

confidence: 98%

Bilateral pial synangiosis in a child with PHACE syndrome

Jack¹,

Chow²,

Fiorillo

et al. 2016

PED

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The acronym PHACE has been used to denote a constellation of abnormalities: posterior fossa anomalies, facial hemangiomas, arterial anomalies, cardiac anomalies, and eye abnormalities. Approximately 30% of patients with large facial hemangiomas have PHACE syndrome, with the vast majority having intracranial arteriopathy. Few reports characterize neurological deterioration from this intracranial arteriopathy, and even fewer report successful treatment thereof. The authors report on a case of a child with PHACE syndrome who presented with an ischemic stroke from a progressive intracranial arteriopathy and describe her successful treatment with bilateral pial synangiosis. An 8-month old girl diagnosed with PHACE syndrome was found to have bilateral internal carotid artery stenosis. Although initially asymptomatic, a few months after diagnosis she suffered a right frontal and parietal stroke. MRI and cerebral angiography investigations demonstrated progressive intracranial arterial stenosis and occlusion. The patient then underwent indirect cerebral revascularization surgery. At 2-year follow-up, she exhibited clinical improvement with persistent speech and motor developmental delay. Follow-up MRI and cerebral angiography showed no new ischemic events and robust extensive vascular collateralization from surgery. PHACE syndrome is an uncommon disease, and affected patients often have cerebral arteriopathy. Although the underlying natural history of cerebral arteriopathy in PHACE remains unclear, cerebral revascularization may represent a potential therapy for symptomatic patients.

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Cerebral vasculopathy and neurologic sequelae in infants with cervicofacial hemangioma: report of eight patients.

Cited by 157 publications

References 0 publications

Lumps and Bumps on the Head in Children: Use of CT and MR Imaging in Solving the Clinical Diagnostic Dilemma

Lumps and Bumps on the Head in Children: Use of CT and MR Imaging in Solving the Clinical Diagnostic Dilemma

Pulse steroid therapy in infantile facial hemangiomas: the early experience

Bilateral pial synangiosis in a child with PHACE syndrome

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