The report of two patients with SOD1 mutations in an apparent sporadic context (families 5 and 7) highlighted that in the current era of emerging disease-modifying drugs to treat SOD1 mutated patients, and with the upcoming urgent need to screen all families, only systematic screening of all ALS patients, including apparent sporadic cases, can capture all patients harboring these mutations. The gene discussed is SOD1; the disease is amyotrophic lateral sclerosis.