BACKGROUND: Corneal nerves can be examined using in vivo confocal microscopy (IVCM). This new technique permits sequential observation of the corneal subbasal nerve plexus and detects early signs of diabetic peripheral neuropathy. OBJECTIVE: To describe a patient with autoimmune peripheral neuropathy followed up using corneal IVCM. DESIGN: Case report. SETTING: Clinic of neurology, Geneva, Switzerland. Patient A 56-year-old man with peripheral neuropathy diagnosed as anti-myelin-associated glycoprotein neuropathy. His symptoms initially worsened despite the administration of intravenous immunoglobulins and plasma exchange. Evolution was eventually favorable after rituximab and corticosteroids were given. At 1-year follow-up, clinical recovery was almost complete, and the patient was stable according to the results of clinical and electrophysiologic assessments. Main Outcome Measure Corneal nerve measurement by IVCM. RESULTS: Examination of corneal nerves using IVCM at 2 different times during the patient's clinical evolution (peak disease and recovery phase) demonstrated histologic signs that correlated with the results of clinical and electrophysiologic assessments. CONCLUSION: This observation supports the hypothesis that corneal IVCM could also be helpful for the early detection or follow-up of autoimmune peripheral neuropathy.