Background: Vascular compression is an extremely rare cause of mononeuropathy and compression of selective tibial nerve branches is an additionally a rare finding and makes diagnosis difficult. Case Description: Here within, we describe the case of a 41-year-old male who presented with isolated mononeuropathy of the medial gastrocnemius (MG) branch of the tibial nerve presented as persistent fasciculations and atrophy. After electromyography and clinical evaluation, surgical exploration was recommended. A vascular bundle was found to be compressing the MG branch of the tibial nerve and thus was ligated to decompress the nerve. Postoperatively, all fasciculations improved and muscle atrophied improved. Conclusion: Vascular compression resulting in mononeuropathy of the peripheral nerves is a rare clinical entity. Clinicians should include these etiologies on their differential when considering surgical exploration of mononeuropathies.