A case of severe refractory chronic urticaria: A novel method for evaluation and treatment

Abstract

With cholinergic urticaria (ChU), the ultimate diagnosis often depends on the demonstration of characteristic urticaria by appropriate provocation. Several treatment options may be helpful but traditional options (antihistamines, leukotriene inhibitors, and immunosuppressives) may be exhausted by the refractory ChU patient. Here, we describe such a case. Demonstration of immediate hypersensitivity to autologous sweat skin testing (ASwST) may provide a rationale for use of omalizumab (Xolair, Genentech Novartis, South San Francisco, CA). Patients with severe ChU may have difficulty producing sufficient quantities of sweat for ASwST given that the very effort that produces the sample exacerbates ChU. Generation of sweat by iontophoresis with pilocarpine nitrate can be performed at many large medical centers. The procedure is simple, safe, and produces varying amounts of sweat depending on the individual. This sweat can then be used for ASwST. Our patient had a positive ASwST with appropriate positive and negative controls. Our testing methods were validated by negative ASwST, saline control, and positive histamine control in a nonatopic, nonurticarial control patient. By the patient's second injection of omalizumab, her quality of life score was significantly improved, as were her daily medication scores and exercise tolerance. We describe the first case of a patient with severe refractory ChU who had a positive ASwST by a novel collection method who has been successfully treated with omalizumab. We present a novel tool for the evaluation and demonstration of sweat-specific IgE in ChU patients who are unable to provide sweat by more traditional means.

Omalizumab for Chronic Urticaria: A Case Series and Overview of the Literature

“…Particularly, by December 2011, to our knowledge, a total of 59 cases of chronic urticaria treated with omalizumab have been reported in the literature comprising five cases of solar urticaria, two cases of heat urticaria, two cases of cold urticaria, three cases of delayed pressure urticaria, three cases of urticaria factitia, three cases of cholinergic urticaria, 40 cases of chronic idiopathic urticaria, and 16 cases of chronic autoimmune urticaria. Furthermore, a total of 139 patients have been enrolled in two randomized controlled trials comparing omalizumab with placebo ( table 2 ) [5, 6, 7, 8, 9, 10, 11, 12, 13, 14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27, 28, 29, 30]. The first randomized trial was restricted to patients with IgE against thyroperoxidase; 27 patients were randomized to omalizumab and 22 to placebo [29].…”

Partial improvement of solar urticaria after omalizumab

“…We hypothesize that other well described variables regarding omalizumab response including variations in receptor expression, specific/total IgE ratios, and cellular sensitivity may have accounted for his lack of dramatic improvement on repeat phototesting. 7 Two other reports of omalizumab with physical urticaria demonstrated complete resolution after a single dose for cholinergic urticaria, 8 whereas a case of cold urticaria had marked improvement after 2 months with complete resolution at 5 months. 9 Further study is warranted to determine the role of anti-IgE for solar urticaria for different action spectrums (UV-A, UV-B, visible light).…”

Evaluation of Different Combined Regimens in the Treatment of Cholinergic Urticaria

“…Both topically applied benzoyl scopolamine and oral scopolamine butylbromide may be helpful in blocking the appearance of cholinergic urticarial lesions after challenge[14]. Traditional options are antihistamines, leukotriene inhibitors, and immunosuppressive agents [15]. However, in some patients cholinergic urticaria may be refractory.…”

“…Benzoyl scopolamine administered topically and scopolamine butylbromide administered orally may be helpful in blocking the appearance of cholinergic urticaria lesions after challenge [14]. The traditional options are antihistamines, leukotriene inhibitors, and immunosuppressive agents [15]. Rapid sweat desensitization with autologous sweat has been reported in patients resistant to conventional therapy who have sweat hypersensitivity [16].…”