Background: Involvement of trochlear nerve during Varicella Zoster Virus (VZV) Infection has been rarely described, and always in association with skin rash.
Case presentation: We describe the case of a patient with VZV infection presenting as isolated diplopia due to fourth cranial nerve palsy. The diagnosis has been obtained through the application of a standardized molecular diagnostic panel, and diplopia resolved after specific antiviral and corticosteroid therapy.
Conclusion: This case evidences that clinicians should be aware of atypical VZV infection, even in the absence of the typical skin rash.