BACKGROUND: Baclofen can relax muscle spasticity by inhibiting the transmission of both mono- and polysynaptic reflexes at the spinal cord. It has been increasingly used off-label for the management of spinal cord lesions or neurologic disorders. However, the expansion use has led to an increase in baclofen-associated complications. CASE PRESENTATION: We report the case of a 60-year-old Chinese female patient who suffered from nocturnal choking and daytime sleepiness. Polysomnography identified central sleep apnea after initiation of low-dosage baclofen (10 mg two times a day) treatment, and a complete resolution of central sleep apnea was associated with baclofen withdrawal. CONCLUSION: This case reminds us that more attention should be paid to the adverse reactions on respiratory control of baclofen, even with low dosage. Regular polysomnography before and after prescription of baclofen is helpful to detect the effects of baclofen on sleep and breathing.