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Bilateral facial capillary malformation associated with eye and brain abnormalities
Juliette Mazereeuw-Hautier1, Samira Syed, John I Harper
1Paediatric Dermatology Department, Great Ormond Street Hospital for Children NHS Trust, London, England. mazereeuw-hautier.j@chu-toulouse.fr
Archives of Dermatology
|August 23, 2006
Summary
Bilateral facial capillary malformation (BFCM) indicates a worse prognosis than unilateral facial port-wine stains. Children with BFCM have a higher risk of associated conditions like glaucoma and leptomeningeal angiomatosis.
Area of Science:
- Dermatology
- Pediatrics
- Medical Genetics
Background:
- Facial capillary malformations (CM) are common vascular anomalies in children.
- Distinguishing between unilateral and bilateral presentations is crucial for prognosis.
- Bilateral facial capillary malformation (BFCM) requires further investigation due to potential associated syndromes.
Purpose of the Study:
- To compare the prognosis of bilateral facial capillary malformation (BFCM) with unilateral facial port-wine stains.
- To identify specific clinical and radiographic differences between BFCM and unilateral CM.
- To assess the association of BFCM with extrafacial lesions and neurological complications.
Main Methods:
- Retrospective study design.
- Inclusion of 350 children with facial CM, with 27 identified with BFCM.
- A control group of 27 children with unilateral CM was randomly selected for comparison.
Main Results:
- BFCM group showed significantly higher rates of extrafacial lesions (63% vs 22%).
- Higher frequency of glaucoma (78% vs 7%) and ipsilateral leptomeningeal angiomatosis (52% vs 7%) in BFCM patients.
- All BFCM patients with bilateral ophthalmic involvement had ipsilateral leptomeningeal angiomatosis.
Conclusions:
- Bilateral facial capillary malformation is associated with a significantly worse prognosis.
- Increased vigilance for associated conditions like glaucoma and CNS involvement is warranted in BFCM.
- BFCM should be considered a distinct entity with a more severe clinical course compared to unilateral CM.
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