title: [Treatment of angiolymphoid hyperplasia with eosinophilia (ALHE) using topical tacrolimus: Two cases].
abstract: BACKGROUND
  Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare disease, currently considered a benign vascular proliferation of unknown etiology, and whose treatment is still unclear and challenging.
  
  
  METHODS
  Two women in their thirties consulted for itchy lesions of the right ear. Both presented with a reddish bleeding papulonodular infiltration of the auricle, with a nodule at the entrance to the external auditory canal in the first patient. Laboratory tests showed no abnormalities and in particular no hypereosinophilia or elevated serum immunoglobulin E. In both cases, histology of lesional skin showed vascular proliferation with thick-walled vessels lined by plump endothelial cells, protruding into the lumen, together with a mixed dermal inflammatory infiltrate consisting primarily of eosinophils and lymphocytes. A diagnosis of ALHE was made in both patients based on clinical and histological features. MRA revealed no underlying vascular malformation in both cases. Patients started treatment with 0.1% tacrolimus ointment twice daily. The pruritic sensation and bleeding had completely subsided within two weeks and the reddish infiltration and nodules had practically disappeared after two months of topical tacrolimus. Continuous application resulted in no recurrence at 6 months of follow-up.
  
  
  CONCLUSIONS
  Treatment of ALHE is still poorly standardized due to doubts concerning the pathophysiology of this rare condition and the small number of available studies. Topical tacrolimus was originally developed for the treatment of moderate to severe atopic dermatitis because of its anti-inflammatory and immunomodulatory properties. Recent studies suggest that this drug may be effective in treating other forms of inflammatory dermatosis. Our two observations suggest that tacrolimus ointment also represents potentially valuable treatment in AHLE.
authors.affiliation: Service de dermatologie, hôpital Saint-Louis, AP-HP, 1, avenue Claude-Vellefaux, 75010 Paris, France. Electronic address: agathe.nouchi@gmail.com.
authors.forename: A
authors.initials: A
authors.lastname: Nouchi
authors.affiliation: Service de dermatologie, hôpital Saint-Louis, AP-HP, 1, avenue Claude-Vellefaux, 75010 Paris, France.
authors.forename: G
authors.initials: G
authors.lastname: Hickman
authors.affiliation: Service d'anatomie pathologique, hôpital Saint-Louis, AP-HP, 1, avenue Claude-Vellefaux, 75010 Paris, France.
authors.forename: M
authors.initials: M
authors.lastname: Battistella
authors.affiliation: Service de dermatologie, centre hospitalier régional d'Orléans, 1, rue Porte-Madeleine, 45000 Orléans, France.
authors.forename: E
authors.initials: E
authors.lastname: Estève
authors.affiliation: Service de dermatologie, hôpital Saint-Louis, AP-HP, 1, avenue Claude-Vellefaux, 75010 Paris, France.
authors.forename: M
authors.initials: M
authors.lastname: Bagot
authors.affiliation: Service d'anatomie pathologique, hôpital Saint-Louis, AP-HP, 1, avenue Claude-Vellefaux, 75010 Paris, France.
authors.forename: D
authors.initials: D
authors.lastname: Vignon-Pennamen
authors.affiliation: Service de dermatologie, hôpital Saint-Louis, AP-HP, 1, avenue Claude-Vellefaux, 75010 Paris, France.
authors.forename: A
authors.initials: A
authors.lastname: Petit
mesh_terms: D000287:Administration, Topical; D000328:Adult; D000796:Angiolymphoid Hyperplasia with Eosinophilia / Q000150:complications / Q000188:drug therapy*; D005260:Female; D006801:Humans; D007166:Immunosuppressive Agents / Q000627:therapeutic use*; D011537:Pruritus / Q000188:drug therapy / Q000209:etiology; D016559:Tacrolimus / Q000627:therapeutic use*
chemical_list: D007166:Immunosuppressive Agents; D016559:Tacrolimus
keywords: ALHE; Angiolymphoid hyperplasia with eosinophilia; HALE; Hyperplasie angiolymphoïde avec éosinophilie; Oreille; Outer ear; Tacrolimus; Traitement; Treatment