Introduction: Amyotrophic Lateral Sclerosis (ALS) is a fatal and progressive disease characterized by the deterioration of motor neurons within the body. This degeneration leads to bulbar dysfunction in the form of an impaired ability to communicate and swallow. Currently, bulbar dysfunction is treated via compensatory methods aimed at maximizing comfort and safety that include environmental adaptations, augmentative and assistive communication and gastrostomy tube placement to replace speech and oral feeding. The role of active intervention is controversial in this patient population and no investigations have examined the potential role lingual exercise might play in ALS bulbar management. The purpose of this study was to explore the impact of an eight week lingual resistance training program on lingual strength and lingual endurance, speech, swallowing, global disease progression and patient-reported outcomes in two individuals with ALS. Methods: Two individuals with ALS (El Escorial criteria) were enrolled across three different time points, each separated by eight weeks (Baseline 1, Baseline 2, Post-Treatment) with a delayed intervention design utilized to benchmark bulbar disease progression. At each evaluation, tongue strength, endurance, swallowing, speech and patient-reported outcomes were collected. Following Baseline 2, participants completed lingual resistance training five days a week for eight weeks (40 sessions). Outcome measures included maximum anterior isometric tongue pressure (MIP), maximum endurance hold time MHT), speech intelligibility, airway safety and patient-reported outcomes. Results: On average, MIPs decreased by 2% during the lead in period and increased by 13% across both participants. On average, MHT declined by 25% between baseline 1 and 2 and increased by 144% following lingual resistance training. No improvements were noted for speech intelligibility or airway safety during swallowing. Patient-perceptions of communicative effectiveness, swallowing impairment and quality of life remained relatively stable over the four-month period. Conclusion: Although no improvements were noted in lingual strength, improvements in lingual endurance hold times were noted in both participants. Further investigation is warranted to validate these preliminary findings in two cases of ALS.