_______________________________________________________________________________________________________________________________ 794 https://www.id-press.eu/mjms/index ID Design Press, Skopje, Republic of Macedonia Open Access Macedonian Journal of Medical Sciences. 2019 Mar 15; 7(5):794-796. https://doi.org/10.3889/oamjms.2019.110 eISSN: 1857-9655 Case Report Angiolymphoid Hyperplasia with Eosinophilia Successfully Treated with Cryotherapy Nina Caca-Biljanovska, Irina Arsovska-Bezhoska * Clinic of Dermatology, Ss Cyril and Methodius University of Skopje, Skopje, Republic of Macedonia Citation: Caca-Biljanovska N, Arsovska-Bezhoska I. Angiolymphoid Hyperplasia with Eosinophilia Successfully Treated with Cryotherapy. Open Access Maced J Med Sci. 2019 Mar 15; 7(5):794-796. https://doi.org/10.3889/oamjms.2019.110 Keywords: Angiolymphoid hyperplasia with eosinophilia; Histopathology; Therapeutic approach *Correspondence: Irina Arsovska Bezhoska. Clinic of Dermatology, Ss Cyril and Methodius University of Skopje, Skopje, Republic of Macedonia. E-mail: irina.arsovska@yahoo.com Received: 27-Feb-2019; Revised: 06-Mar-2019; Accepted: 07-Mar-2019; Online first: 14-Mar-2019 Copyright: © 2019 Nina Caca-Biljanovska, Irina Arsovska-Bezhoska. This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 International License (CC BY-NC 4.0) Funding: This research did not receive any financial support Competing Interests: The authors have declared that no competing interests exist Abstract BACKGROUND: Angiolymphoid hyperplasia with eosinophilia is an uncommon, benign, vasoproliferative cutaneous neoplasm with uncertain origin. It preferably affects middle-aged adults, manifesting as plum-colored pruritic papules, nodules and plaques, which can persist indefinitely, relapsing over time. Different response/resistance to various therapeutic modalities and frequent recurrences impose a great therapeutic dilemma. CASE PRESENTATION: Herein, we present a 77-year-old male patient with a 7-month-history of unrecognized cutaneous manifestations on his left shoulder and flank. Based on the investigations, the diagnosis of angiolymphoid hyperplasia with eosinophilia was established. We applied cryotherapy as a treatment of choice. The complete regression of the skin lesions and three years disease-free period was achieved. CONCLUSION: Although surgery is the standard therapeutic approach, the disease recurs despite multiple surgical attempts. Therefore, we recommend cryotherapy as effective and safe treatment modalities for angiolymphoid hyperplasia with eosinophilia. Knowing the recurring nature of this disease, the patients with angiolymphoid hyperplasia should stay on short-term follow up in order to monitor if new lesions occur. Introduction Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare, benign vascular disease described for the first time by Wells and Whimster in 1969 [1]. It is characterized with prominent vascular proliferation with lymphocytic and eosinophilic infiltration of the skin [2]. This vascular tumor was also named epithelioid hemangioma (EH) by Weiss and Enzinger in 1982, in order to differentiate the lesion from the malignant vascular tumor, epithelioid hemangioendothelioma [3]. ALHE usually occurs in young to middle aged adults, and it usually affects women [4]. The most frequent involved sites are head and neck (face, ear and periauricular), rarely present in other areas and organs like shoulders, trunk, extremities, hands, breast, liver, spleen, orbit and bone [5]. Oral mucosa, conjunctiva and genital area can also be affected [6]. Clinically, ALHE shows solitary or multiple, dome-shaped, smooth, erythematous/violaceous papules/nodules [7]. There are no systemic symptoms, but sometimes, the lesions may be painful, pruritic or pulsatile due to its vascular nature [8]. Usually, there is no regional lymphadenopathy or peripheral eosinophilia, but approximately 20% of patients with ALHE may show eosinophilia [9]. ALHE grows slowly and can persist indefinitely [10]. Some cases clear up spontaneously, without any treatment, but more often, persistent and recurrent lesions require medical treatment [11]. We report a case of a male patient with a 7- months history of cutaneous lesions and histological characteristics compatible with ALHE. After cryotherapy, the complete healing of skin lesions was achieved. He is three years disease-free, without any recurrence.