Immunoreactivity of Wilms tumor 1 (WT1) as an additional evidence supporting hemangiomatous rather than inflammatory origin in the etiopathogenesis of angiolymphoid hyperplasia with eosinophilia
Keywords:
angiolymphoid hyperplasia with eosinophilia, Wilms tumor 1, GLUT-1, hemangiomaAbstract
Background: Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare vascular proliferative disorder mainly located in the periauricular region. The etiopathogenesis of ALHE is unknown, and it is still controversial as to whether the entity represents a benign vascular neoplasm or an inflammatory process.
Aim: Recently, the intracytoplasmic staining pattern of Wilms tumor 1 (WT1) on immunohistochemistry has highlighted true vascular neoplasms, such as microvenular hemangioma, tufted angioma, and spindle cell hemangioma, which has made it helpful to distinguish ALHE from vascular malformations, as there is a negative staining pattern in the other entities. We aimed to investigate the immunoreactivity of ALHE specimens for WT1 as well as glucose transporter protein 1 (GLUT1) immunohistochemistry, an important and sensitive marker for the diagnosis of infantile hemangioma, which recently has been described to label other hemangiomas, such as verrucous hemangioma.
Material and methods: Clinical data and histopathological specimens from patients diagnosed with ALHE were reviewed, and immunohistochemical staining and microscopic analysis for WT-1 and GLUT1 were performed.
Results: Intracytoplasmic endothelial staining of WT1 was detected in 19 of 20 ALHE specimens. GLUT1 was not detected in any ALHE specimen.
Conclusions: We conclude that ALHE may represent a true hemangioma (i.e., benign vascular neoplasia) characterized by an eosinophil- and lymphocyte-rich inflammatory component as opposed to the reactive inflammatory dermatosis with a positive intracytoplasmic staining pattern for WT1. As far as we are aware, WT1 staining for ALHE has not been described to date.
References
Trindade F, Tellechea O, Torrelo A, Requena L, Colmenero I. Wilms Tumor 1 expression in vascular neoplasms and vascular malformations. Am J Dermatopathol 2011;33(6):569-672.
Trindade F, Kutzner H, Requena L, Tellechea O, Colmenero I. Microvenular hemangioma: an immunohistochemical study of 9 cases. Am J Dermatopathol 2012;34(8):810-812.
Trindade F, Torrelo A, Requena L et al. An immunohistochemical study of verrucous hemangiomas. J Cutan Pathol 2013;40(5):472-476.
Sezer E, Erbil H, Koseoglu D, Filiz N, Kurumlu Z. Angiolymphoid hyperplasia with eosinophilia mimicking Bowenoid papulosis. Clin Exp Dermatol 2007;32(1):281-283.
Busquets A, Sanchez JL. Angiolymphoid hyperplasia with eosinophilia induced by trauma. Int J Dermatol 2006;45(10):1211-1214.
Calonje E, Brenn T, Lazar A, McKee PH. Tumors of vascular origin. In: Calonje E, ed. McKee's Pathology of the Skin. 4th ed. London: Elsevier Saunders, 2010
Onishi Y, Ohara K. Angiolymphoid hyperplasia with eosinophilia associated with arteriovenous malformations: a clinicopathological correlation with angiography and serial estimamtion of serum levels renin, eosinophil cationic protein and interleukin 5. Br J Dermatol 1999; 140(6):1153-1156.
Manton RN, Itinteang T, de Jong S, Brasch HD, Tan ST. Angiolymphoid hyperplasia with eosinophilia developing within a port wine stain. J Cutan Pathol 2016;43(1):53-56.
Bakan V, Aliagaoglu C, Yildiz A, Emsen A. Multiple pyogenic granulomas on the face after landmine injury. Pediatr Dermatol 2008; 25(3): 397-398.
Stewart N, Zagarella S, Mann S. Angiolymphoid hyperplasia with eosinophilia occurring after venipuncture trauma. J Dermatol 2013;40(5):393-395.
Hollo P, Marschalko M, Sikos G, Harsing J, Horwart A. Angiolymphoid hyperplasia with eosinophilia in pregnancy. J Eur Acad Dermatol Venereol 2005; 19(5): 645-646.
Chou S, Subramanian V, Lau HM, Achan A. Renal anastomosing hemangiomas with a diverse spectrum: report of two cases and review of the literature. Int J Surgical Pathol 2014; 22(4):369-373.
Wang L, Gao T, Wang G. Verrucous hemangioma: a clinicopathological and immunohistochemical analysis of 74 cases. J Cutan Pathol 2014; 41(11): 823-830.
Ali FR, Madan V. Facial angiolymphoid hyperplasia with eosinophilia: sustained remission following treatment with carbondioxide laser. Clin Exp Dermatol 2016; 41(1): 96-98.
Garrido-Rios AA, Sanz-Munoz C, Torrero-Anton MV, Martinez-Garcia Miranda-Romero A. Angiolymphoid hyperplasia with eosinophilia on the tongue. Clin Exp Dermatol 2009;34: (8):729-731.
Sotiriou E, Apalla Z, Patsatsi A, Panagiotidou DD, Ooannides D. Angiolymphoid hyperplasia with eosinophilia: good response to photodynamic therapy. Clin Exp Dermatol 2009;34(8):629-631.
Su HH, Shan SJ, Elston DM, Guo Y, Men JL. Congenital Blaschkoid angiolymphoid hyperplasia with eosinophilia of the anogenital region. Am J Dermatopathol 2016;38(4):305-306.
Sebok B, Batai I, Anga B, Schneider I. Angiolymphoid hyperplasia with eosinophilia. Orv Hetil 1998; 139(12):1-15.
Adler BL, Krausz AE, Minuti A, Silverberg JI, Lew-Tov H. Epidemiology and treatment of angiolymphoid hyperplasia with eosinophilia (ALHE): a systemic review. J Am Acad Dermatol 2016;74(3):506-512.
Isohisa T, Masuda K, Nakai N, Takenaka H, Katoh N. Angiolymphoid hyperplasia with eosinophilia treated successfully with imiquimod. Int J Dermatol 2014;53(1):43-44.
Nobeyama Y, Ishiuji Y, Nakagawa H. Retiform hemangioendothelioma treated with conservative therapy: report of a case and review of the literature. Int J Dermatol 2016;55(2): 238-243.
Ahrens WA, Ridenour RV, Caron BL, Caron BL, Miller DV, Folpe AL. Human Pathol 2008;39(10):1519-1526.
Wang L, Liu L, Gao T. Congenital disseminated tufted angioma. J Cutan Pathol 2013; 40(4):40-48.
Johann AC, Salla JT, Gomez RS et al. GLUT-1 in oral benign vascular lesions. Oral Diseases 2007; 13: 51-5.
Published
Issue
Section
License
Dermatology Practical & Conceptual applies a Creative Commons Attribution License (CCAL) to all works we publish (http://creativecommons.org/licenses/by-nc/4.0/). Authors retain the copyright for their published work.
How to Cite
Share
