Here we described the case of a 60 yr old non-smoker woman, with a history of Graves' disease (GD) associated to papillary thyroid carcinoma. After tumor removal, during the diagnostic follow-up for thyroid cancer, there was evidence of severe Graves' ophthalmopathy (GO) successfully treated with iv glucocorticoids. After this treatment, GO entered inactive status. Patient was then re-evaluated for thyroid cancer with human recombinant TSH (rTSH). Orbital [¹¹¹In-DTPA-D-phe1]octreotide scan was also performed and resulted to be negative. Shortly after rTSH administration, a moderate and transient pain behind the eye globes at rest and during eye movement was reported, with an increase in the activity score, but without further GO progression. Twenty-four hours after rTSH administration, the patient was submitted to a second [¹¹¹In-DTPA-D-phe1]octreotide scan revealing significant orbital uptake, likely related to rapid accumulation of activated lymphocytes with inflammatory cytokines, or fibroblasts expressing SSR in the orbital tissue, or interstitial edema due to the inflammation process. At last follow-up performed after 1 yr, there was no evidence for active thyroid cancer or changes in GO severity and/or activity, and orbital [¹¹¹In-DTPA-D-phe1]octreotide uptake was negative. This case further supports the involvement of TSH-R in the pathogenesis of GO. It also confirms the usefulness of orbital [¹¹¹In-DTPA-D-phe1]octreotide scan to evaluate GO activity.