Background/Purpose Recovery of upper extremity (UE) function is a priority for individuals with multiple sclerosis (MS) whose disease sequalae often results in loss of UE strength and motor control. Acute Intermittent Hypoxia (AIH) is a novel intervention where an individual breathes bouts of air with low oxygen (∼9% O2) to set off a cascade of cellular events that may induce neuroplasticity and improve motor function. This intervention has shown promising effects in individuals with motor incomplete spinal cord injury (iSCI), even after a single bout. The purpose of this case report was to assess the ability of a single-bout AIH protocol to improve UE strength and function in an individual with chronic, relapsing-remitting MS. Methods A 30-year-old, right-handed female with a 10-year history of MS (Expanded Disability Status Scale score 3.0), who presented with LUE weakness and increased LUE tone participated in two treatment sessions. In the first session, she received a sham intervention where she was delivered normal room air, through the hypoxia generator for the entire treatment. The following week she received AIH. The 9-Hole Peg Test (9HPT) and Hand-held dynamometry (HHD) were performed immediately before and after the intervention Results Following the single bout of AIH, compared to baseline, left shoulder abduction strength increased 8% immediately after treatment and 21% at the 30-minute assessment. Left wrist extension strength increased 26% and 30%, respectively. Left shoulder flexion and elbow extension did not change. Comparatively, right shoulder abduction strength increased 27% and 7%. Right wrist extension strength increased 12% and 1%. Functionally, the participant's 9HPT time decreased from 44.88 to 41.41 seconds on her left but increased from 15.69 to 16.06 seconds on her right. Discussion AIH treatment was well tolerated by our participant and a single bout of AIH resulted in positive changes in UE strength and function on her more involved side. These results are promising, and further research is required to further assess safety, feasibility, and potential efficacy of AIH in people with MS. Recovery of upper extremity (UE) function is a priority for individuals with multiple sclerosis (MS) whose disease sequalae often results in loss of UE strength and motor control. Acute Intermittent Hypoxia (AIH) is a novel intervention where an individual breathes bouts of air with low oxygen (∼9% O2) to set off a cascade of cellular events that may induce neuroplasticity and improve motor function. This intervention has shown promising effects in individuals with motor incomplete spinal cord injury (iSCI), even after a single bout. The purpose of this case report was to assess the ability of a single-bout AIH protocol to improve UE strength and function in an individual with chronic, relapsing-remitting MS. A 30-year-old, right-handed female with a 10-year history of MS (Expanded Disability Status Scale score 3.0), who presented with LUE weakness and increased LUE tone participated in two treatment sessions. In the first session, she received a sham intervention where she was delivered normal room air, through the hypoxia generator for the entire treatment. The following week she received AIH. The 9-Hole Peg Test (9HPT) and Hand-held dynamometry (HHD) were performed immediately before and after the intervention Following the single bout of AIH, compared to baseline, left shoulder abduction strength increased 8% immediately after treatment and 21% at the 30-minute assessment. Left wrist extension strength increased 26% and 30%, respectively. Left shoulder flexion and elbow extension did not change. Comparatively, right shoulder abduction strength increased 27% and 7%. Right wrist extension strength increased 12% and 1%. Functionally, the participant's 9HPT time decreased from 44.88 to 41.41 seconds on her left but increased from 15.69 to 16.06 seconds on her right. AIH treatment was well tolerated by our participant and a single bout of AIH resulted in positive changes in UE strength and function on her more involved side. These results are promising, and further research is required to further assess safety, feasibility, and potential efficacy of AIH in people with MS.