Pristine W Lee1, Ilona J Frieden2, Jenna L Streicher3, Timothy McCalmont4, Anita N Haggstrom5. 1. Department of Dermatology, Kaiser Permanente, San Rafael, California. 2. Department of Dermatology, University of California, San Francisco, California. 3. Department of Dermatology, Emory University, Atlanta, Georgia. 4. Department of Dermatology, University of California, San Francisco, California; Department of Pathology, University of California, San Francisco, California. 5. Department of Dermatology, Indiana University School of Medicine, Indianapolis, Indiana. Electronic address: ahaggstr@iu.edu.
Abstract
BACKGROUND: Noninvoluting congenital hemangioma (NICH) is a distinct vascular tumor of infancy. OBJECTIVE: We describe the clinical characteristics, histopathology, imaging, and natural history of NICH and compare our findings with previous reports. METHODS: We conducted a retrospective review of charts and photographic databases from 2 vascular anomaly centers over a 15-year period. RESULTS: Thirty cases of NICH were identified. All patients had fully formed vascular lesions at birth that demonstrated a nonprogressive course. The trunk and lower extremities were preferred sites and there was a female predominance. Thirteen of 30 patients reported pain. Focal necrosis and scarring was seen in a minority. Doppler studies, when performed, confirmed high vascular flow. Microscopic evaluation of 4 excised lesions showed lobular areas of endothelial cell proliferation directly adjacent to ectatic malformed vessels. Immunohistochemical studies demonstrated absence of glucose transporter-1 protein expression in every case. Wilms tumor-1 positivity was observed in lobular areas. The larger vessels did not stain with Wilms tumor-1, but some displayed D2-40 positivity. LIMITATIONS: Patients were referred to university-based pediatric vascular anomaly centers, with potential bias toward more severe or extensive cases. CONCLUSIONS: This retrospective study highlights the unique clinical and histopathologic features of NICH.
BACKGROUND: Noninvoluting congenital hemangioma (NICH) is a distinct vascular tumor of infancy. OBJECTIVE: We describe the clinical characteristics, histopathology, imaging, and natural history of NICH and compare our findings with previous reports. METHODS: We conducted a retrospective review of charts and photographic databases from 2 vascular anomaly centers over a 15-year period. RESULTS: Thirty cases of NICH were identified. All patients had fully formed vascular lesions at birth that demonstrated a nonprogressive course. The trunk and lower extremities were preferred sites and there was a female predominance. Thirteen of 30 patients reported pain. Focal necrosis and scarring was seen in a minority. Doppler studies, when performed, confirmed high vascular flow. Microscopic evaluation of 4 excised lesions showed lobular areas of endothelial cell proliferation directly adjacent to ectatic malformed vessels. Immunohistochemical studies demonstrated absence of glucose transporter-1 protein expression in every case. Wilms tumor-1 positivity was observed in lobular areas. The larger vessels did not stain with Wilms tumor-1, but some displayed D2-40 positivity. LIMITATIONS: Patients were referred to university-based pediatric vascular anomaly centers, with potential bias toward more severe or extensive cases. CONCLUSIONS: This retrospective study highlights the unique clinical and histopathologic features of NICH.
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