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Case: A 29-year-old woman with acute peroneal tendon subluxation underwent superior retinacular repair. On exposure, a single peroneal myotendinous unit was encountered, as opposed to the usual presence of independent peroneal tendons arising from separate muscle bellies. At 3-year follow-up, she has had no recurrence with full return to activity and no limitations. Conclusion: Multiple peroneal myotendinous variants have been described; however, this report is the first to describe direct intraoperative observation of a single peroneal myotendinous unit. Whether this anatomic variant contributed to the patient’s problem or has other potential clinical sequelae remains to be elucidated.