Abstract

<h3>Introduction</h3> Chronic spontaneous urticaria (CSU) has been well described in autoimmune thyroid disorders, but less associated with Graves' Disease (GD), and the mechanism of their relationship is still unknown. Thyroid-associated CSU has been shown to be more difficult to manage. <h3>Case Description</h3> We present a 10-year-old female with difficult to control CSU with associated facial angioedema that started six months after diagnosis of Graves' disease that was being treated with methimazole. Her symptoms presented multiple times each week as bodily hives, angioedema of her cheeks, tongue, and throat with difficulty breathing, which caused her to be treated multiple times with epinephrine for concern of anaphylaxis. Therapy was rapidly escalated with H1 and H2 antihistamines, leukotriene receptor antagonist, chronic steroids and omalizumab without resolution. She was positive for thyroid stimulating immunoglobulins and p-ANCA antibodies; and negative for anti-TPO and anti-FcεRI antibodies. Due to the concern of vasculitic conversion, methimazole was stopped and she underwent radioiodine ablation for her GD to eventually become euthyroid. She was subsequently started on cyclosporine, but symptoms continue intermittently. <h3>Discussion</h3> Thyroid-associated CSU remains difficult to control from standard therapies, even when the patient becomes euthyroid. Several reports show that controlling the thyroid levels would resolve the CSU and a few only after thyroidectomy. Most studies note that anti-TPO antibodies have been associated with CSU, but only a few report an association with GD. Methimazole can cause p-ANCA positivity for concern of vasculitic conversion. Further studies are needed to evaluate the mechanism of GD with CSU to better manage these complicated patients.

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