Introduction: This case report describes perhaps the youngest case of neuropathic arthropathy (NA) in an upper extremity documented in a patient with myelomeningocele. Methods: Case report. Results: This case report describes presenting symptoms, results of radiologic work-up, treatment approach and functional outcome thus far. Included is a clinical picture of the patient as well as a radiograph of the affected elbow. Discussion: NA, also known as Charcot joint, is a rare occurrence in the upper extremity. NA is well documented in the literature; however, the majority of reports are of adults with affected lower extremities. This report describes the very rare occurrence of upper extremity involvement of a 5 year old child with myleomeningocele. As the literature does not guide treatment for this age of patient, this study reports the treatment plan and the child's outcome thus far. Due to the rarity of this situation, future research will be quite difficult. Reports of cases such as this including treatment and outcome will be helpful to guide clinical decision-making for future cases. © 2007 Informa UK Ltd.