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      Early experience with teprotumumab for chronic thyroid eye disease

      case-report

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          Abstract

          Purpose

          To report the first case of a patient with chronic thyroid eye disease (TED) treated with teprotumumab.

          Observations

          A 50-year-old female with a 3-year history of Graves’ disease presented with bilateral exophthalmos greatest on the left side. She was followed for 2 years with stable proptosis measurements (23mm OD, 28mm OS). Her clinical activity score (CAS) was 1 and there were no examination findings reflective of active inflammation. The patient underwent systemic treatment with teprotumumab and despite chronic TED and low CAS, she had notable improvement in proptosis (18mm OD, 22mm OS) and decrease in extraocular muscle volume as noted on orbital imaging.

          Conclusion and importance

          This case report suggests that teprotumumab may be used in patients with chronic TED and low CAS. Improvement in the proptosis and reduction in extraocular muscle volume suggest that teprotumumab may alter disease course even in patients with inactive or quiescent TED.

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          Most cited references11

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          Teprotumumab for the Treatment of Active Thyroid Eye Disease

          Thyroid eye disease is a debilitating, disfiguring, and potentially blinding periocular condition for which no Food and Drug Administration-approved medical therapy is available. Strong evidence has implicated the insulin-like growth factor I receptor (IGF-IR) in the pathogenesis of this disease.
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            Efficacy and safety of three different cumulative doses of intravenous methylprednisolone for moderate to severe and active Graves' orbitopathy.

            Optimal doses of i.v. glucocorticoids for Graves' orbitopathy (GO) are undefined.
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              Clinical features of Graves' ophthalmopathy in an incidence cohort.

              To determine the clinical characteristics of an incidence cohort of patients with Graves' ophthalmopathy. We reviewed the community medical records of 120 patients residing in Olmsted County, Minnesota, in whom Graves' ophthalmopathy was diagnosed between 1976 and 1990. Among 120 patients with Graves' ophthalmopathy, 108 (90%) patients had Graves' hyperthyroidism, one (1%) had primary hypothyroidism, four (3%) had Hashimoto's thyroiditis, and seven (6%) were euthyroid. At some point in their clinical course, eyelid retraction was present in 108 patients, whereas the approximate frequency of exophthalmos was 62% (73 patients); restrictive extraocular myopathy, 43% (51 patients); and optic nerve dysfunction, 6% (seven patients). Only six (5%) patients had eyelid retraction, exophthalmos, optic nerve dysfunction, extraocular muscle involvement, and hyperthyroidism. At the time of diagnosis of ophthalmopathy, upper eyelid retraction and eyelid lag were documented in 85 and 52 patients, respectively, and the most frequent ocular symptom was pain (36 patients, 30%). Diplopia was noted at the initial examination by 20 patients, lacrimation was present in 25 patients, 19 patients had photophobia, and nine patients had blurred vision. Decreased vision from optic neuropathy was present in less than 2% of eyes at the time of diagnosis. Thyroid dermopathy and acropachy accompanied Graves' ophthalmopathy in five patients (4%) and one (1%) patient, respectively. Myasthenia gravis occurred in only one patient. Eyelid retraction is the most common clinical sign of Graves' ophthalmopathy. The complete constellation of typical features (hyperthyroidism, eyelid retraction, exophthalmos, restrictive extraocular myopathy, and optic nerve dysfunction) occurs relatively infrequently.
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                Author and article information

                Contributors
                Journal
                Am J Ophthalmol Case Rep
                Am J Ophthalmol Case Rep
                American Journal of Ophthalmology Case Reports
                Elsevier
                2451-9936
                15 May 2020
                September 2020
                15 May 2020
                : 19
                : 100744
                Affiliations
                [a ]Division of Oculofacial Plastic and Reconstructive Surgery, UC San Diego Viterbi Family Department of Ophthalmology, La Jolla, CA, USA
                [b ]Division of Plastic and Reconstructive Surgery, UC San Diego Department of Surgery, La Jolla, CA, USA
                Author notes
                []Corresponding author. Division of Oculofacial Plastic and Reconstructive Surgery, UC San Diego Viterbi Family Department of Ophthalmology, Shiley Eye Institute, La Jolla, CA, USA. bkorn@ 123456health.ucsd.edu
                Article
                S2451-9936(20)30095-5 100744
                10.1016/j.ajoc.2020.100744
                7243051
                32462101
                e2d9d4ae-d795-4361-af9b-d84e980976a8
                © 2020 The Authors

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                : 27 April 2020
                : 6 May 2020
                : 11 May 2020
                Categories
                Case Report

                thyroid eye disease (ted),proptosis,clinical activity score (cas),teprotumumab,inactive disease,quiescent disease,fibrotic disease

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