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Dysphonia and Dyspnea in Idiopathic Hypereosinophilic Syndrome Treated with Mepolizumab
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A2997 - Dysphonia and Dyspnea in Idiopathic Hypereosinophilic Syndrome Treated with Mepolizumab
Author Block: D. Kay, A. Ataya, D. R. Urbine; Pulmonary and Critical Care, University of Florida, Gainesville, FL, United States.
Introduction: Hypereosinophilic syndrome (HES) is characterized by a persistently elevated eosinophil count associated with eosinophil-related end-organ damage and thrombo-embolic events, in the absence of an identifiable cause. We present a case of idiopathic HES treated with the IL-5 inhibitor, Mepolizumab, with resolution of symptoms including dyspnea and dysphonia.
Case: 73-year-old woman presented with a three-year history of dyspnea on exertion, chest tightness, and dysphonia. Over the course of three years she had peripheral eosinophilia ranging from 1128-1907 cells/uL while on 40 mg prednisone daily. Her medical history was relevant for unprovoked venous thromboembolic disease. Her workup prior to presentation to our clinic included a bronchoscopy that did not show any eosinophilia, a negative connective tissue disease, vasculitis and parasite workup. Regarding her dysphonia, she had been evaluated by neurology and ENT. She was trialed on bronchodilators and inhaled steroids without any improvement. Based on her eosinophilia while on high dose steroids, history of thromboembolic disease, negative workup, and tissue hypereosinophilia on dermatology biopsy that showed superficial perivascular dermatitis with many eosinophils, the patient was diagnosed with idiopathic hypereosiniphilic syndrome. She was taken off steroids and started on Mepolizmab. After 3 doses she experienced significant improvement in her symptoms of dyspnea and chest tightness with complete resolution of her dysphonia.
Discussion: Persistent peripheral eosinophilia and end organ damage that is seen in HES may occur as a a result of myeloproliferative variant of the hypereosinophilic syndrome, or more commonly due to maturation, differentiation and mobilization of eosinophil from hematopoietic progenitors due to the influence interleukin-5. Mepolizumab, a humanized anti-interlukin 5 monoclonal antibody, reduces eosinophil counts in blood and tissues by blocking interleukin-5, an activating eosinophil cytokine, through binding to eosinophil surface receptors. Our patient probably had eosinophilic involvement of her laryngeal muscle or nerves manifesting as muscle tension dysphonia that resolved with therapy. Although treatment with Mepolizumab has been shown to be effective in a variety of eosinophilic diseases, there are limited studies on long-term use of the treatment.
Author Block: D. Kay, A. Ataya, D. R. Urbine; Pulmonary and Critical Care, University of Florida, Gainesville, FL, United States.
Introduction: Hypereosinophilic syndrome (HES) is characterized by a persistently elevated eosinophil count associated with eosinophil-related end-organ damage and thrombo-embolic events, in the absence of an identifiable cause. We present a case of idiopathic HES treated with the IL-5 inhibitor, Mepolizumab, with resolution of symptoms including dyspnea and dysphonia.
Case: 73-year-old woman presented with a three-year history of dyspnea on exertion, chest tightness, and dysphonia. Over the course of three years she had peripheral eosinophilia ranging from 1128-1907 cells/uL while on 40 mg prednisone daily. Her medical history was relevant for unprovoked venous thromboembolic disease. Her workup prior to presentation to our clinic included a bronchoscopy that did not show any eosinophilia, a negative connective tissue disease, vasculitis and parasite workup. Regarding her dysphonia, she had been evaluated by neurology and ENT. She was trialed on bronchodilators and inhaled steroids without any improvement. Based on her eosinophilia while on high dose steroids, history of thromboembolic disease, negative workup, and tissue hypereosinophilia on dermatology biopsy that showed superficial perivascular dermatitis with many eosinophils, the patient was diagnosed with idiopathic hypereosiniphilic syndrome. She was taken off steroids and started on Mepolizmab. After 3 doses she experienced significant improvement in her symptoms of dyspnea and chest tightness with complete resolution of her dysphonia.
Discussion: Persistent peripheral eosinophilia and end organ damage that is seen in HES may occur as a a result of myeloproliferative variant of the hypereosinophilic syndrome, or more commonly due to maturation, differentiation and mobilization of eosinophil from hematopoietic progenitors due to the influence interleukin-5. Mepolizumab, a humanized anti-interlukin 5 monoclonal antibody, reduces eosinophil counts in blood and tissues by blocking interleukin-5, an activating eosinophil cytokine, through binding to eosinophil surface receptors. Our patient probably had eosinophilic involvement of her laryngeal muscle or nerves manifesting as muscle tension dysphonia that resolved with therapy. Although treatment with Mepolizumab has been shown to be effective in a variety of eosinophilic diseases, there are limited studies on long-term use of the treatment.
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