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We report, for the first time, the case of amyotrophic lateral sclerosis (ALS) onset in a 54-year-old woman not following a strict gluten-free diet (GFD) for six years after the diagnosis of celiac disease (CD). The patient did not display any sign of gluten toxicity, malabsorption, or CD-related comorbidity. Previous cases reported in medical literature showed ALS-like forms secondary to gluten intolerance, with symptom remission after a strict GFD. Even after correcting dietary flaws, the clinical manifestation of ALS did not improve in our patient. The main neurological involvement was a muscular weakness of upper and lower limbs, and dysphagia causing a weight loss for reduced food intake without any signs of malabsorption. Only vitamin D and folate levels were observed below normal; therefore, nutritional management required an oral supplementation of nutrients and vitamins. Muscle wasting was confirmed by an electrophysiological study using electromyography (EMG); body composition was assessed using bioimpedentiometry. Even after implementing a stricter GFD, clinical manifestations of the ALS did not improve. Therefore, the diagnosis of secondary neurological involvement in gluten ingestion was ruled out with ALS as an exclusionary diagnosis, at the end of the routine diagnostic pathway. The patient is currently under neurological and nutritional follow up for both ALS and CD as per standard of care to ensure her nutritional support and maintain the best possible quality of life during the progressive prognosis of the neurological diagnosis.