• Shinya Nakao, Manabu Miyata, Mariko Hirai, Shusaku Mizoguchi, Hirokazu Ohashi, and Hirokazu Nishiwaki.
• Department of Ophthalmology, Tenri Hospital, Tenri, Japan.
• Medicine (Baltimore). 2022 Dec 30; 101 (52): e31864e31864.

RationalePulled-in-two syndrome is one of the significant complications of strabismus surgery. This study aimed to report a case of pulled-in-two syndrome of the contractured medial rectus muscle (MR) that occurred during strabismus surgery for strabismus fixus due to high myopia, and to describe a rescue of this complication.Patient ConcernsA woman in her 60s presented to our Ophthalmology Department with the main complaint of unilateral high myopia and severe myopic strabismus fixus. Esotropia exceeded 45° and hypotropia exceeded 15° in her right eye in the Hirschberg test. Right eye duction was markedly limited in every gaze direction. Orbital magnetic resonance images showed rupture of the superior rectus to lateral rectus band ligament and lengthening of the distance between the SR and LR muscles in the right eye.DiagnosisDue to the patient's ophthalmic examination and imaging results, she was diagnosed with high myopic strabismus fixus.InterventionsWe performed MR recession and Yokoyama surgery to correct right eye hypoesotropia. In the MR recession procedure, pulled-in-two syndrome (MR muscle tear) occurred. Thus, no additional procedure was performed on the MR. After the surgery, she presented 45 prism diopter exotropia and 18 prism diopter residual right hypotropia in a Krimsky test. We performed a second surgery, combining MR muscle advancement and inferior rectus (IR) muscle recession, 3 months after the first surgery.OutcomesOne and a half years after the second surgery, she presented exotropia of 14 prism diopters without hypotropia in the Krimsky test and was satisfied with her ocular position and improved motility.LessonsWe experienced pulled-in-two syndrome in a case with severe myopic strabismus fixus and achieved a good outcome by performing additional surgery 3 months later, in which the lost MR muscle was advanced. This case underscores that, if the lost muscle cannot be found during surgery, one should maintain composure and perform a reoperation a few months after the initial surgery, if necessary. This case report can aid in making rescue treatment decisions when pulled-in-two syndrome occurs.Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.

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