Background Visual snow syndrome is a phenomenon for which no effective treatment is known. It is highly comorbid with migraine, therefore we performed a retrospective chart review of patients with visual snow syndrome treated with a monoclonal antibody against calcitonin gene related peptide or its receptor. Findings We enrolled 15 patients with visual snow syndrome who received at least once a monoclonal antibody against calcitonin gene related peptide or its receptor. None of the patients reported relief of visual snow syndrome whereas those patients with comorbid migraine reported a very good efficacy of the antibody against the migraine headache but not against the migraine aura. Conclusion The data suggest that visual snow syndrome is not mediated by calcitonin gene related peptide in a relevant way and that the calcitonin gene related peptide receptor is not involved in the network underlying the visual snow syndrome.