Rationale: Dengue is the most important human arboviral disease. Neurological manifestations occur rarely in dengue. To the best of our knowledge, there is only one reported case of dengue-associated sudden sensorineural hearing loss (SSNHL) in Brazil untill now.Patient concerns: A 42-year-old man was presented to the emergency department with unconsciousness and generalized tonic-clonic seizures. Diagnosis: Dengue-associated aseptic meningitis, acute kidney injury, and SSNHL. Interventions: The patient was treated with anticonvulsants and thiamine and underwent mechanical ventilation. He received combined ceftriaxone and acyclovir, which were later switched to meropenem, acyclovir and ampicillin empirically until culture results became available. He also required hemodialysis and plasmapheresis sessions and fresh frozen plasma and buffy coat transfusions until definitive diagnosis. Outcomes: The patient was discharged after improvement of his general condition and of his blood test results, but hearing loss remained. A six-month follow-up visit showed persistent deafness. Lessons: Dengue should be included in the differential diagnosis of patients from dengue-endemic areas presenting an acute febrile disease with neurological manifestations. To the best of our knowledge, this is the second reported case of dengue-associated SSNHL, suggesting an association between dengue and development of SSNHL.