Chikungunya viral (CHIKV) fever is often a self-limiting febrile illness associated with severe debilitating arthralgia. Neurological complications associated with CHIKV, although rare, have been reported in literature; however, longitudinally extensive transverse myelitis (LTEM) is rarely associated with it. We present a case of a middle-aged man with a 1-week history of low-grade fever and arthralgia followed by urinary retention and quadriplegia. A sensory level was noted at T2. On subsequent investigations, he was diagnosed with LETM. Although LETM is commonly seen in patients with neuromyelitis optica, the other possible etiologies are inflammatory and parainfectious. To date, only two cases of LETM are reported worldwide in association with CHIKV fever and this is the first case from Pakistan. With frequent chikungunya outbreaks, neurological complications are increasingly seen in clinical practice. The knowledge of these associations will result in their early diagnosis and treatment.