Observational Study

. 2023 Mar-Apr;39(2):150-155.

doi: 10.1097/IOP.0000000000002267. Epub 2022 Sep 9.

Outcomes of Patients With Thyroid Eye Disease Partially Treated With Teprotumumab

Tiffany C Ho¹, Robi N Maamari¹, Andrea L Kossler², Connie M Sears², Suzanne K Freitag³, Edith R Reshef³, Roman Shinder⁴, Daniel B Rootman⁵, Stefania B Diniz⁵, Alon Kahana^{6

7}, Dianne Schlachter⁶, Thai H Do⁶, Peter Kally⁶, Sara Turner⁷, Ali Mokhtarzadeh⁸, Andrew R Harrison⁸, Christopher J Hwang⁸, Hee Joon Kim⁹, Sarah A Avila⁹, Dilip A Thomas¹⁰, Maja Magazin¹⁰, Sara T Wester¹¹, Wendy W Lee¹¹, Kevin D Clauss¹¹, John B Holds^{12

13}, Matthew Sniegowski¹⁴, Christopher J Compton¹⁵, Christian Briggs¹⁵, Amina I Malik¹⁶, Mark J Lucarelli¹⁷, Cat N Burkat¹⁷, Luv G Patel¹⁸, Steven M Couch¹

Affiliations

¹ John F. Hardesty, MD, Department of Ophthalmology and Visual Sciences, Washington University School of Medicine, St. Louis, Missouri, U.S.A.
² Department of Ophthalmology, Byers Eye Institute, Palo Alto, California, U.S.A.
³ Department of Ophthalmology, Ophthalmic Plastic Surgery, Massachusetts Eye and Ear Infirmary, Boston, Massachusetts, U.S.A.
⁴ Department of Ophthalmology, SUNY Downstate Medical Center, Brooklyn, New York, U.S.A.
⁵ Division of Orbital and Ophthalmic Plastic Surgery, Jules Stein Eye Institute, University of California, Los Angeles, California, U.S.A.
⁶ Department of Ophthalmology, Oakland University William Beaumont School of Medicine, Rochester, Michigan, U.S.A.
⁷ Kahana Oculoplastic & Orbital Surgery, Rochester, Michigan, U.S.A.
⁸ Department of Ophthalmology, University of Minnesota, Minneapolis, Minnesota, U.S.A.
⁹ Department of Ophthalmology, Emory University School of Medicine, Atlanta, Georgia, U.S.A.
¹⁰ Department of Ophthalmology, Georgia Regents University, Augusta, Georgia, U.S.A.
¹¹ Bascom Palmer Eye Institute, Department of Ophthalmology, University of Miami Miller School of Medicine, Miami, Florida, U.S.A.
¹² Ophthalmic Plastic and Cosmetic Surgery Inc., Des Peres, Missouri, U.S.A.
¹³ Departments of Ophthalmology and Otolaryngology-Head and Neck Surgery, Saint Louis University, St. Louis, Missouri, U.S.A.
¹⁴ Department of Ophthalmology, University of Missouri Kansas City, Kansas City, Missouri, U.S.A.
¹⁵ Department of Ophthalmology and Visual Sciences, University of Louisville, Louisville, Kentucky, U.S.A.
¹⁶ Department of Ophthalmology, Blanton Eye Institute, Houston Methodist Hospital, Houston, Texas, U.S.A.
¹⁷ Oculoplastic, Facial Cosmetic & Orbital Surgery, Department of Ophthalmology and Visual Sciences, University of Wisconsin-Madison, Madison, Wisconsin, U.S.A.
¹⁸ Retina Center of Texas, Dallas, Texas, U.S.A.

PMID: 36095848
PMCID: PMC10771969
DOI: 10.1097/IOP.0000000000002267

Observational Study

Outcomes of Patients With Thyroid Eye Disease Partially Treated With Teprotumumab

Tiffany C Ho et al. Ophthalmic Plast Reconstr Surg. 2023 Mar-Apr.

. 2023 Mar-Apr;39(2):150-155.

doi: 10.1097/IOP.0000000000002267. Epub 2022 Sep 9.

Authors

Affiliations

¹ John F. Hardesty, MD, Department of Ophthalmology and Visual Sciences, Washington University School of Medicine, St. Louis, Missouri, U.S.A.
² Department of Ophthalmology, Byers Eye Institute, Palo Alto, California, U.S.A.
³ Department of Ophthalmology, Ophthalmic Plastic Surgery, Massachusetts Eye and Ear Infirmary, Boston, Massachusetts, U.S.A.
⁴ Department of Ophthalmology, SUNY Downstate Medical Center, Brooklyn, New York, U.S.A.
⁵ Division of Orbital and Ophthalmic Plastic Surgery, Jules Stein Eye Institute, University of California, Los Angeles, California, U.S.A.
⁶ Department of Ophthalmology, Oakland University William Beaumont School of Medicine, Rochester, Michigan, U.S.A.
⁷ Kahana Oculoplastic & Orbital Surgery, Rochester, Michigan, U.S.A.
⁸ Department of Ophthalmology, University of Minnesota, Minneapolis, Minnesota, U.S.A.
⁹ Department of Ophthalmology, Emory University School of Medicine, Atlanta, Georgia, U.S.A.
¹⁰ Department of Ophthalmology, Georgia Regents University, Augusta, Georgia, U.S.A.
¹¹ Bascom Palmer Eye Institute, Department of Ophthalmology, University of Miami Miller School of Medicine, Miami, Florida, U.S.A.
¹² Ophthalmic Plastic and Cosmetic Surgery Inc., Des Peres, Missouri, U.S.A.
¹³ Departments of Ophthalmology and Otolaryngology-Head and Neck Surgery, Saint Louis University, St. Louis, Missouri, U.S.A.
¹⁴ Department of Ophthalmology, University of Missouri Kansas City, Kansas City, Missouri, U.S.A.
¹⁵ Department of Ophthalmology and Visual Sciences, University of Louisville, Louisville, Kentucky, U.S.A.
¹⁶ Department of Ophthalmology, Blanton Eye Institute, Houston Methodist Hospital, Houston, Texas, U.S.A.
¹⁷ Oculoplastic, Facial Cosmetic & Orbital Surgery, Department of Ophthalmology and Visual Sciences, University of Wisconsin-Madison, Madison, Wisconsin, U.S.A.
¹⁸ Retina Center of Texas, Dallas, Texas, U.S.A.

PMID: 36095848
PMCID: PMC10771969
DOI: 10.1097/IOP.0000000000002267

Abstract

Purpose: In response to the coronavirus (COVID-19) pandemic, teprotumumab production was temporarily halted with resources diverted toward vaccine production. Many patients who initiated treatment with teprotumumab for thyroid eye disease were forced to deviate from the standard protocol. This study investigates the response of teprotumumab when patients receive fewer than the standard 8-dose regimen.

Methods: This observational cross-sectional cohort study included patients from 15 institutions with active or minimal to no clinical activity thyroid eye disease treated with the standard teprotumumab infusion protocol. Patients were included if they had completed at least 1 teprotumumab infusion and had not yet completed all 8 planned infusions. Data were collected before teprotumumab initiation, within 3 weeks of last dose before interruption, and at the visit before teprotumumab reinitiation. The primary outcome measure was reduction in proptosis more than 2 mm. Secondary outcome measures included change in clinical activity score (CAS), extraocular motility restriction, margin reflex distance-1 (MRD1), and reported adverse events.

Results: The study included 74 patients. Mean age was 57.8 years, and 77% were female. There were 62 active and 12 minimal to no clinical activity patients. Patients completed an average of 4.2 teprotumumab infusions before interruption. A significant mean reduction in proptosis (-2.9 mm in active and -2.8 mm in minimal to no clinical activity patients, P < 0.01) was noted and maintained during interruption. For active patients, a 3.4-point reduction in CAS ( P < 0.01) and reduction in ocular motility restriction ( P < 0.01) were maintained during interruption.

Conclusions: Patients partially treated with teprotumumab achieve significant reduction in proptosis, CAS, and extraocular muscle restriction and maintain these improvements through the period of interruption.

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Conflict of interest statement

The following authors are consultant/advisor for Horizon: Christopher Compton, Suzanne Freitag, Andrew Harrison, John Holds, Andrea Kossler, Wendy Lee, Amina I Malik, Daniel Rootman, Sara Wester, Dianne Schlachter, Roman Shindler, Matthew Sniegowski. Suzanne Freitag: Poriferous LLC—consultant/advisor; WL Gore and Associates—consultant/advisor. Andrew Harrison: RVL Pharmaceuticals—consultant/advisor. John Holds: Revance Therapeutics—ownership interests; Cypris Medical—ownership interests. Alan Kahana: Stryker Corporation—consultant/advisor, BioTissue Inc.—consultant/advisor; Genentech, Inc Researcher. Andrea Kossler: RVL pharmaceuticals—consultant/advisor; Axogen Inc—consultant/advisor; Immunovant Inc—consultant/advisor. Wendy Lee: Allergan—consultant/advisor, Galderma—consultant/advisor, Revance—consultant/advisor, RVL pharmaceuticals—consultant/advisor, Mallinckrodt Pharmaceuticals—consultant/advisor, Evolus—consultant/advisor; RoC—consultant/advisor; Osmotica—consultant/advisor. Daniel Rootman: Celularity—consultant/advisor. The other authors authors have no financial or conflicts of interest to disclose.

Figures

**FIG. 1.**
Scatterplot of number of teprotumumab infusions against proptosis change. Larger data points indicate more than 1 patient represented.

See this image and copyright information in PMC

References

1. Bahn RS. Graves’ ophthalmopathy. N Engl J Med 2010;362:726–738. - PMC - PubMed
1. Douglas RS, Kahaly GJ, Patel A, et al. Teprotumumab for the treatment of active thyroid eye disease. N Engl J Med 2020;382: 341–352. - PubMed
1. Smith TJ, Kahaly GJ, Ezra DG, et al. Teprotumumab for thyroid-associated ophthalmopathy. N Engl J Med 2017;376:1748–1761. - PMC - PubMed
1. Horizon Therapeutics plc Announces Short-Term TEPEZZA (teprotumumab-trbw) Supply Disruption Due to Government-Mandated (Operation Warp Speed) COVID-19 Vaccine Production. Available at: https://ir.horizontherapeutics.com/news-releases/news-release-details/ho.... Accessed February 15, 2022.
1. Statement on TEPEZZA (teprotumumab-trbw) Availability. Available at: https://www.horizontherapeutics.com/media-center/horizon-resources/state.... Accessed February 15, 2022.

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Outcomes of Patients With Thyroid Eye Disease Partially Treated With Teprotumumab

Affiliations

Outcomes of Patients With Thyroid Eye Disease Partially Treated With Teprotumumab

Authors

Affiliations

Abstract

Conflict of interest statement

Figures

References

Publication types

MeSH terms

Substances

Grants and funding

LinkOut - more resources

Full Text Sources