Vitiliginous lesions during contact immunotherapy for alopecia in a patient with autoimmune thyroiditis

Abstract

Letter to the Editor Sir,Squaric acid dibutylester (SADBE) is frequently used for the treatment of alopecia, but sometimes unwanted side effects occur. Herein we report a case which developed vitiliginous lesions induced by topical SADBE application in a patient with autoimmune thyroiditis. A 60-year-old female visited our department, complaining of diffuse alopecia of the scalp. She was suffering from chronic autoimmune thyroiditis over several years, and taking thyradin (90mg per day). After obtaining written informed consent, topical application of SADBE solution was started. Two months later, a sufficient effect was obtained for hair regrowth; however, when she was treated with 0.05% SADBE, depigmentation appeared and rapidly spread on the scalp. On physical examination, diffuse depigmentation along with gray hairs was found on the scalp, forehead, and nape ( Fig. 1). Laboratory examination showed normal liver and renal function, anti-nuclear antibody (1:160, speckled), anti-thyroid antibody (1:100), anti-microsome antibody (1:6400), anti-thyroid peroxidase antibody (139.6 IU/ ml, normal <16), but anti-thyroid stimulating hormone receptor antibody, free T3 and T4 levels were normal. A skin biopsy showed decrease of melanocytes and melanin deposition in the epidermis (Fig. 2A). Results of immunohistochemistry revealed a predominant CD8-positive T-cell infiltration (Fig. 2B). SADBE therapy was stopped, and betamethasone butyrate propionate and carpronium chrolide lotion were applied, which showed a dramatical effect within 9 months (Fig. 3). Adverse effects of topical immunomodulatory therapy include local irritation, blister formation, persistent dermatitis, lymphadenopathy, generalized eczema, and urticarial reaction. Mechanism of contact immunotherapy is suggested to modulate cytokine gene expression balance, and interferon-γ expression was reduced while interleukin-2 (IL-2), IL-8, IL-10 and tumor necrosis factor-α levels were increased in the lesional skin [1].Further, recent studies demonstrate that Th17 cells are involved in the pathogenesis of contact hypersensitivity. Skin biopsies of hypersensitivity reactions to contact sensitizers demonstrate the presence of CD8+ T-cells, Th1 CD4+ effector cells and regulatory T-cells [2]. T-cells play an important role in the pathogenesis of vitiligo, and IL-10 is supposed to play a role by inhibiting T-cells. Also, active Th17 cells are increased in vitiligo skin [3], and thus those mediators may play a role in the autoimmune mechanisms of vitiligo. Our case developed vitiliginous lesions not only on the scalp, but the forehead and nape were also involved beyond the application areas. Three months later from the start of immunotherapy, depigmentation was suddenly

Severe pustular lesions induced by topical immunotherapy with squaric acid dibutylester for alopecia totalis

“…The most common adverse effect of topical immunomodulatory therapy is eczema, and other rare forms include local irritation, blister formation, persistent dermatitis, lymphadenopathy, generalized eczema, urticarial reaction, and vitiliginous depigmentation. [1][2][3][4] We herein reported two cases of alopecia totalis, who developed a severe pustular reaction on the total scalp after topical application of 0.05% and 0.001% SADBE, respectively. Case 1 showed lymphadenopathy on the postauricular areas, and Case 2 developed upper eyelid edema.…”

Report of a rare case of alopecia areata coexisting with trichotillomania

“…Alopecia areata (AA) is a common dermatological disease characterized by patchy areas of nonscarring alopecia. [ 1 2 ] Trichotillomania (TT) is an expression of obsessive-compulsive disorder defined by the urge to pull out his/her hair. [ 3 ]…”