Purpose To describe a case series of enlarged extraocular muscles in young patients. Design Retrospective interventional case series. Methods Study involves 6 young patients who presented with atypical restrictive strabismus and reduced eye movements. Examination, neuroimaging, biochemistry results, and biopsy results are presented. The surgeries are described, and the follow-up over 1-20 years is presented. Results All 6 patients had enlarged extraocular muscles that caused restrictive strabismus. The patients had no significant medical history. There was no identifiable underlying pathology on biochemistry tests or muscle biopsy. Three of the patients required multiple surgeries. Conclusion Atypical presentations of strabismus should be investigated for systemic conditions. Neuroimaging of the orbit and brain and a biopsy of the affected muscles should be considered. The patients should be counseled that multiple surgeries may be necessary to improve the strabismus, and it is unlikely that orthotropia will be attained.
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