Rationale: Tendinitis of the longus colli muscle is an aseptic inflammatory process leading to acute posterior neck pain, neck stiffness, and dysphagia or odynophagia. We present a patient exhibiting an infrequent symptom, vertigo. This is the first description of the occurrence of vertigo symptoms caused by longus colli tendinitis. Patient concerns: A 38-year-old man was diagnosed with vertigo, presenting with a 1-month history of dizziness, palpitations, and numbness in the hands. Diagnosis: Longus colli tendinitis. The diagnosis was established using magnetic resonance imaging fat-suppression sequences. Interventions: Treatment with corticosteroid injections and acupotomy. Outcomes: The symptoms relieved immediately after the treatment and complete resolution of the symptoms was observed after 1 week. Lessons: Longus colli tendinitis with vertigo is an under-reported condition in the literature and physicians should be aware of its existence. A lack of familiarity with the anatomy of the prevertebral space and its variable radiographic appearance makes the diagnosis of longus colli tendinitis clinically difficult. Misdiagnosis of this condition may lead to unnecessary interventions in vertigo.