Journal of Clinical and Aesthetic Dermatology

MAY 2018

An evidence-based, peer-reviewed journal for practicing clinicians in the field of dermatology

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43 JCAD JOURNAL OF CLINICAL AND AESTHETIC DERMATOLOGY May 2018 • Volume 11 • Number 5 C A S E R E P O R T D Different lymphatic and angiomatous malformations of the skin rarely co-occur, and are all complex diseases. These malformations are mostly seen at birth, and they can be of a nevoid pattern, superficial, or with deeper involvement. 1 Two of the most widely recognized lesions are angioma serpiginosum (AS) 2,3 and lymphangioma circumscriptum (LC). 4 Occasionally, angiomatous and lymphamatous lesions overlap and are known as " hematolymphangioma." 5 Additionally, a hyperkeratosis can develop on the surface of angiomatous and lymphomatous lesions, and are known as angiokeratoma (AK) 6 or verrucous hemangioma (VH) 7 for angiomatous lesions and lymphangiokeratoma (LK) 8 or verrucous lymphangioma (VL) 9 for lymphangiomatous lesions. However, to the best of our knowledge, an overlapping of these lesions in a patient has not been reported in the literature. CASE PRESENTATION A 15-year-old girl presented to our outpatient clinic with vesicular leg lesions that had been present since birth. When the lesions were first noticed by her, they appeared only as erythematous punctate smooth patches with no vesicles or papules; however, when the patient was three years old, a few red bulges emerged and increased in number steadily as she aged. Over time, the bulges developed into bright red papules, with a few yellowish vesicles also appearing near or around the red patches. When the patient was nine years old, her parents observed some thickening and crusting in yellowish and red papules. The color of the papules also changed, with the yellowish ones turning a dirty, dull-colored yellow and the red ones turning a darker red. The lesions were serpiginous and were situated on an area with dimensions of 32cm×14cm on the upper and anterolateral part of the left leg. All of the lesions were symptomless, and the patient only complained of the cosmetic appearance of the lesions. Upon clinical examination, five different types of lesions were detected: 1) transparent, grouped or scattered yellowish vesicles; 2) keratotic, grouped, dark-yellowish papules; 3) bright red, grouped papules; 4) keratotic surfaced, grouped dark red papules; and 5) patchy, punctate, and erythematous red macules. All of the lesions were intertwined along the lines of Blaschko and were in the form of intricate and irregular serpiginous plaques (Figures 1A–C). A systemic physical A B S T R A C T We present the case of a 15-year-old girl who presented with an unusual grouping of lesions on her upper left leg. The lesions had been present since birth. The patient had five different types of lesions: 1) transparent grouped or scattered yellowish vesicles; 2) keratotic-surfaced, grouped dark-yellowish papules; 3) bright-red grouped papules; 4) keratotic-surfaced grouped dark-red papules; and 5) patchy, punctate, and erythematous red macules. All of the lesions were intertwined along the lines of Blaschko and were in the form of irregular serpiginous plaques. Histopathological examinations of the lesions showed three main histological features, and diagnoses of the lesions were made as lymphangioma circumscriptum, lymphangiokeratoma, and verrucous hemangioma. To the best of our knowledge, such intertwined lesions have never been reported in the literature. Hence, we suggest that the name of this unique combination of lesions be "congenital nevoid mixed hemato-lymphangio-keratoma serpiginosum." KEYWORDS: Lymphangioma, hemangioma, lymphangiokeratoma, verrucous hemangioma, angiokeratoma circumscriptum, angioma serpiginosum Blaschko-linear "Congenital Mixed Hemato-lymphangio-keratoma Serpiginosum" Naeviforme: A New Hybrid Entity or Various Morphological Reflections of the Two Different Vascular Abnormalities? by BETUL TAS, MD; AYSEL ÇAĞLAR, MD; and SERDAR ALTINAY, MD Dr. Tas is with the Health Sciences University, Istanbul Bagcilar Research and Training Hospital, Department of Dermatology in Istanbul, Turkey. Dr. Çağlar is with the Health Sciences University, Istanbul Bagcilar Research and Training Hospital, Department of Pathology in Istanbul, Turkey. Dr. Altinay is with the Health Sciences University, Bakirkoy Sadi Konuk Research and Training Hospital, Department of Pathology, in Istanbul, Turkey. J Clin Aesthet Dermatol. 2018;11(5):43–47 FUNDING: No funding was provided for this article. DISCLOSURES: The authors have no conflicts of interest relevant to the content of this article. CORRESPONDENCE: Betul Tas, MD; Email: betulavc@yahoo.com

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