Trochleitis is an easily treatable condition; however, it is often misdiagnosed by many clinicians because of its rare incidence. We report the case of a 14-year-old Saudi male patient, known to have type 1 diabetes mellitus (DM) and pansinusitis, who presented to the emergency department with a one-day history of severe right periorbital pain exacerbated by upgaze and supraduction. There was intense point tenderness on palpation over the trochlear region of the orbit with no underlying swelling or redness. Both eyes had a corrected visual acuity of 0.8. Mildly limited elevation (-1) of the right eye was noted. All other extraocular movements were normal in both eyes. Contrast-enhanced CT of the head and orbits showed mild trochlear swelling, inflammation, and pansinusitis. He was treated with a single intratrochlear injection of steroids, which immediately and significantly improved the symptoms. To the best of our knowledge, this is the second case of trochleitis associated with paranasal sinusitis in children. This suggests that a possible, but rare, association between these two conditions may exist in the pediatric population.