Extensive Airway Hemangiomas in Two Patients Without Beard Hemangiomas

Suh, Ki-Young; Rosbe, Kristina W.; Meyer, Anna K.; Frieden, Ilona J.

doi:10.1111/j.1525-1470.2010.01096.x

Cited by 5 publications

(4 citation statements)

References 7 publications

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“…This was an institutional review board–approved retrospective study of 32 infants with PHACE with cerebral or cervical arteriopathy evaluated between July 2008 and October 2011 and treated with oral propranolol. A minority of these patients have been reported in prior publications . Data were obtained from a group of pediatric dermatologists, all members of the Hemangioma Investigator Group with special expertise in the field of hemangiomas and PHACE syndrome who attended a multidisciplinary research conference on PHACE syndrome in Milwaukee, Wisconsin, September 15–16, 2010.…”

Section: Methodsmentioning

confidence: 99%

Propranolol Use in PHACE Syndrome with Cervical and Intracranial Arterial Anomalies: Collective Experience in 32 Infants

Metry

Frieden

Hess

et al. 2012

Pediatric Dermatology

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1. Objective Combine collective clinical experience using oral propranolol therapy in PHACE syndrome infants with cerebrovascular anomalies. 2. Design Retrospective study of patients evaluated between July 2008 and October 2011. 3. Setting Seven pediatric dermatology centers. 4. Patients 32 infants with definite PHACE syndrome and cervical and/or intracranial arterial anomalies. 5. Intervention Oral propranolol: average dose of 1.8 mg/kg/day divided t.i.d. or b.i.d., for an average duration of 12.3 months. 6. Main Outcome Measure Adverse neurologic events. 7. Results 7/32 (22%) patients were categorized as higher-risk for stroke, defined on MRA as severe, long-segment narrowing or non-visualization of major cerebral or cervical vessels without anatomic evidence for collateral circulation, often in the presence of concomitant cardiovascular comorbidities. Only 1 patient developed a change in neurologic status during propranolol treatment: a mild right hemiparesis that remained static and improved while propranolol was continued. An additional 3 patients had worsening hemangioma ulceration and/or tissue necrosis during therapy. 8. Conclusions This is the largest report thus far of PHACE patients treated with propranolol. While no catastrophic neurologic events occurred, serious complications, particularly severe ulcerations were seen in a minority of patients, and given the sample size we cannot negate the possibility that propranolol could augment the risk of stroke in this population. We continue to advise caution in using systemic beta-blockers, particularly for children with vascular anomalies at higher risk for stroke. Use of the lowest possible dosage, slow dosage titration, and t.i.d. dosing in order to minimize abrupt changes in blood pressure and close follow-up, including neurologic consultation as needed, are recommended.

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Section: Methodsmentioning

confidence: 99%

Propranolol Use in PHACE Syndrome with Cervical and Intracranial Arterial Anomalies: Collective Experience in 32 Infants

Metry

Frieden

Hess

et al. 2012

Pediatric Dermatology

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“…Further details of these 2 cases are reported elsewhere. 8 The other 2 patients with unilateral distribution had large hemangiomas (Ͼ200 cm 2 ), 1 of which was located in the left frontotemporal, left mandibular, and frontonasal regions and 1 in the left frontotemporal, left maxillary, left mandibular, and frontonasal regions. Three patients had hepatic hemangiomatosis, and 1 had a lumbosacral hemangioma.…”

Section: Cutaneous Hemangiomasmentioning

confidence: 99%

“…Subglottic hemangiomas and cutaneous infantile hemangiomas have identical histopathologic and immunophenotypic characteristics, which implies that their pathogenesis may be similar. 8 GLUT-1, an erythrocytetype glucose transporter expressed in infantile hemangiomas, 14 has also been shown to be expressed in most subglottic hemangiomas.…”

Section: Pathogenesismentioning

confidence: 99%

Clinical Spectrum and Risk of PHACE Syndrome in Cutaneous and Airway Hemangiomas

Haggstrom

Skillman

Garzon

et al. 2011

Arch Otolaryngol Head Neck Surg

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“…Bilateral presentation and involvement of multiple regions of the "beard area" increase this risk [ 43 ]. However, it should be kept in mind that airway hemangiomas have been demonstrated in association with extrafacial HI [ 44 ], with facial HI outside the S3 segment [ 45 ], and even in the absence of cutaneous HI.…”

Section: Airway Obstructionmentioning

confidence: 98%

Vascular Tumors/Birthmarks

Blei

2015

Pediatric Skin of Color

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Abstract"Vascular anomalies" encompass a heterogeneous group of vascular lesions, of unclear etiology and often with unpredictable behavior. Patients with vascular anomalies represent a unique population, in that they have focal (or generalized) aberrations of vascular development (in vascular malformations) or vascular proliferation (in vascular tumors). The etiology of these disorders is yet unclear, and likely represents a multifactorial process, although new discoveries into the genetic and molecular bases of these disorders have dominated the past decade.

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Extensive Airway Hemangiomas in Two Patients Without Beard Hemangiomas

Cited by 5 publications

References 7 publications

Propranolol Use in PHACE Syndrome with Cervical and Intracranial Arterial Anomalies: Collective Experience in 32 Infants

Propranolol Use in PHACE Syndrome with Cervical and Intracranial Arterial Anomalies: Collective Experience in 32 Infants

Clinical Spectrum and Risk of PHACE Syndrome in Cutaneous and Airway Hemangiomas

Vascular Tumors/Birthmarks

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