In vivo characterization of solitary angiokeratoma by reflectance confocal microscopy and high definition optical coherence tomography Ivette Alarcon, MD,a,b Johanna Brito, MD,c Llucia Alos, MD,c Josep Malvehy, MD, PhD,a,b,d and Susana Puig, MD, PhDa,b,d,e Barcelona, Spain
CLINICAL PRESENTATION A 62-year-old man presented with a 9-month history of a bleeding lesion. Clinical evaluation showed a 5-mm red-to-black keratotic papule on the first digit of the left foot (Fig 1, A).
Fig1. Angiokeratoma. A, Clinical appearance showing a blue-black and dark red papule with a crust. B, Dermoscopy showing keratotic scale, hemorrhagic crusts, and rainbow pattern. C, Mosaic of reflectance confocal microscopy showing epidermal cells surrounding the prominent ectatic vascular lacunae (asterisk) forming septa. D, En face mode of high-definition optical coherence tomography showing a branch vessel of the subpapillary plexus (arrow) with a dilatation at the end forming a lacunae (asterisk).
DERMOSCOPIC APPEARANCE Dermoscopy showed keratotic scale and hemorrhagic crusts. Black and violaceous areas with poorly defined borders and some shiny white streaks were also seen. Additionally, a multicolored rainbow pattern associated with Kaposi sarcoma was present (Fig 1, B). We could not identify any specific criteria for melanocytic lesion. S43
S44 Alarcon et al
J AM ACAD DERMATOL
JANUARY 2015
CONFOCAL MICROSCOPY APPEARANCE AND HIGH-DEFINITION OPTICAL COHERENCE TOMOGRAPHY At the epidermal level we observed highly reflective superficial scale present focally or in more extensive areas. Dermoepidermal junction displayed an abnormal architecture with epidermal cells surrounding the prominent ectatic vascular lacunae forming septa (Fig 1, C ). The contents of these lacunae are described in Fig 2, A. High-definition optical coherence tomography (HD-OCT) provided the objective in vivo determination of epidermal thickness and showed dilatation of the branches of the subpapillary plexus (Fig 1, D).
Fig 2. Angiokeratoma. A, Under reflectance confocal microscopy, structures morphologically consistent with erythrocytes and leukocytes ( yellow arrow) and endothelial cells inside the lacunae were organized, displaying a rhomboidal mesh (asterisk) and resembling a fishnet. B, Histology showing a tumor composed of vascular channels occupying the papillary lamina propria; some of them were partly or completely enclosed by the elongated rete pegs. (Hematoxylin and eosin stain; original magnification: 31.)
HISTOLOGIC DIAGNOSIS Histology identified dilated thin-walled blood vessels, lined by a layer of endothelial cells, in the papillary dermis and hyperkeratosis, leading to the diagnosis of angiokeratoma (Fig 2, B).
KEY MESSAGE The lesion was diagnosed as nodular melanoma and the case was referred. Dermoscopy, in this case, did not show a clear-cut diagnosis, because dark lacunae, the most specific finding in solitary angiokeratomas,1 were absent. In vivo reflectance confocal microscopy and HD-OCT improved the diagnostic accuracy of angiokeratoma, allowing the better characterization of the microvascular network and the morphology of this tumor.
REFERENCE 1. Zaballos P, Daufı C, Puig S, Argenziano G, Moreno-Ramırez D, Cabo H, et al. Dermoscopy of solitary angiokeratomas: a morphological study. Arch Dermatol 2007;143:318-25. From the Melanoma Unit, Dermatology Department,a and the Pathology Department,c Hospital Clinic of Barcelona, the August Pi i Sunyer Biomedical Research Institute (IDIBAPS),b the Centre of Biomedical Research on Rare Diseases (CIBERER), ISCIII,d and the Department of Medicine, Universitat de Barcelona,e Spain. Publication of this article was supported by 3Gen. Funding sources: This study was supported in part by grants from Fondo de Investigaciones Sanitarias PI 09/01393 and 12/00840, Spain by the CIBER de Enfermedades Raras of the Instituto de Salud Carlos III, Spain.
Conflicts of interest: None declared. Reprint requests: Susana Puig, MD, Melanoma Unit, Dermatology Department, Hospital Clinic Barcelona, Villarroel 170 08036, Barcelona, Spain. E-mail: [email protected]. J Am Acad Dermatol 2015;72:S43-4. 0190-9622/$36.00 ª 2014 by the American Academy of Dermatology, Inc. http://dx.doi.org/10.1016/j.jaad.2014.06.001
CLINICAL PRESENTATION A 62-year-old man presented with a 9-month history of a bleeding lesion. Clinical evaluation showed a 5-mm red-to-black keratotic papule on the first digit of the left foot (Fig 1, A).
Fig1. Angiokeratoma. A, Clinical appearance showing a blue-black and dark red papule with a crust. B, Dermoscopy showing keratotic scale, hemorrhagic crusts, and rainbow pattern. C, Mosaic of reflectance confocal microscopy showing epidermal cells surrounding the prominent ectatic vascular lacunae (asterisk) forming septa. D, En face mode of high-definition optical coherence tomography showing a branch vessel of the subpapillary plexus (arrow) with a dilatation at the end forming a lacunae (asterisk).
DERMOSCOPIC APPEARANCE Dermoscopy showed keratotic scale and hemorrhagic crusts. Black and violaceous areas with poorly defined borders and some shiny white streaks were also seen. Additionally, a multicolored rainbow pattern associated with Kaposi sarcoma was present (Fig 1, B). We could not identify any specific criteria for melanocytic lesion. S43
S44 Alarcon et al
J AM ACAD DERMATOL
JANUARY 2015
CONFOCAL MICROSCOPY APPEARANCE AND HIGH-DEFINITION OPTICAL COHERENCE TOMOGRAPHY At the epidermal level we observed highly reflective superficial scale present focally or in more extensive areas. Dermoepidermal junction displayed an abnormal architecture with epidermal cells surrounding the prominent ectatic vascular lacunae forming septa (Fig 1, C ). The contents of these lacunae are described in Fig 2, A. High-definition optical coherence tomography (HD-OCT) provided the objective in vivo determination of epidermal thickness and showed dilatation of the branches of the subpapillary plexus (Fig 1, D).
Fig 2. Angiokeratoma. A, Under reflectance confocal microscopy, structures morphologically consistent with erythrocytes and leukocytes ( yellow arrow) and endothelial cells inside the lacunae were organized, displaying a rhomboidal mesh (asterisk) and resembling a fishnet. B, Histology showing a tumor composed of vascular channels occupying the papillary lamina propria; some of them were partly or completely enclosed by the elongated rete pegs. (Hematoxylin and eosin stain; original magnification: 31.)
HISTOLOGIC DIAGNOSIS Histology identified dilated thin-walled blood vessels, lined by a layer of endothelial cells, in the papillary dermis and hyperkeratosis, leading to the diagnosis of angiokeratoma (Fig 2, B).
KEY MESSAGE The lesion was diagnosed as nodular melanoma and the case was referred. Dermoscopy, in this case, did not show a clear-cut diagnosis, because dark lacunae, the most specific finding in solitary angiokeratomas,1 were absent. In vivo reflectance confocal microscopy and HD-OCT improved the diagnostic accuracy of angiokeratoma, allowing the better characterization of the microvascular network and the morphology of this tumor.
REFERENCE 1. Zaballos P, Daufı C, Puig S, Argenziano G, Moreno-Ramırez D, Cabo H, et al. Dermoscopy of solitary angiokeratomas: a morphological study. Arch Dermatol 2007;143:318-25. From the Melanoma Unit, Dermatology Department,a and the Pathology Department,c Hospital Clinic of Barcelona, the August Pi i Sunyer Biomedical Research Institute (IDIBAPS),b the Centre of Biomedical Research on Rare Diseases (CIBERER), ISCIII,d and the Department of Medicine, Universitat de Barcelona,e Spain. Publication of this article was supported by 3Gen. Funding sources: This study was supported in part by grants from Fondo de Investigaciones Sanitarias PI 09/01393 and 12/00840, Spain by the CIBER de Enfermedades Raras of the Instituto de Salud Carlos III, Spain.
Conflicts of interest: None declared. Reprint requests: Susana Puig, MD, Melanoma Unit, Dermatology Department, Hospital Clinic Barcelona, Villarroel 170 08036, Barcelona, Spain. E-mail: [email protected]. J Am Acad Dermatol 2015;72:S43-4. 0190-9622/$36.00 ª 2014 by the American Academy of Dermatology, Inc. http://dx.doi.org/10.1016/j.jaad.2014.06.001
