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TAO are classically described in Grave\u2019s disease (GD) however it may occur in euthyroid and hypothyroid patients. Those patients usually test positive for Thyroid Stimulating Hormone receptor antibodies (TRAb). For instance, only few cases of severe Hashimoto\u2019s thyroiditis (HT) associated orbitopathy with negative TRAb are reported to date.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case presentation<\/jats:title><jats:p>Herewith we report a rare case of a middle-aged female who presented with bilateral progressive upper and lower palpebral edema and a unilateral marked proptosis associated with asthenia, headache and decrease in visual acuity. Biological investigation was notable for high levels of anti-thyroid peroxidase antibodies (Anti-TPO) in an otherwise euthyroid patient with negative TRAb. Orbital Magnetic resonance imaging revealed edema of the extraocular muscles and inflammation of periorbital soft tissue. The patient received a treatment with intravenous methylprednisolone followed by oral treatment with prednisone. This regimen was both effective and safe with minimal metabolic side effects in our patient.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Minor ocular manifestations of HT are common; however, severe sight threatening ophtalmopathy in the absence of TRAb is rare. Multiple differential diagnosis should be considered and investigated before diagnosing this rare entity. Management of similar cases is currently based on reports and no clear guidelines have been elaborated, corticosteroids is the mainstream of treatment with a potential benefit of selenium supplementation in mild to moderate cases.<\/jats:p><\/jats:sec>","DOI":"10.1186\/s12902-020-00658-6","type":"journal-article","created":{"date-parts":[[2020,11,27]],"date-time":"2020-11-27T10:02:45Z","timestamp":1606471365000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["A case report of thyroid-associated Orbitopathy with elevated TPO antibodies"],"prefix":"10.1186","volume":"20","author":[{"given":"Radwan","family":"El Othman","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christelle","family":"Ephrem","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Elsie","family":"Touma","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6918-5689","authenticated-orcid":false,"given":"Souheil","family":"Hallit","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rola","family":"El Othman","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2020,11,27]]},"reference":[{"key":"658_CR1","doi-asserted-by":"publisher","first-page":"343","DOI":"10.1507\/endocrj.K11E-019","volume":"58","author":"A Yoshihara","year":"2011","unstructured":"Yoshihara A, Yoshimura Noh J, Nakachi A, et al. 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