Surgery for childhood appendicitis is considered to have good prognosis. Critical illness polyneuropathy (CIP) is a rare devastating neuromuscular disorder characterised by profound axonal motor dysfunction of unknown aetiology. We report a unique case of CIP in a young adolescent female at a regional pediatric surgery centre following operation for appendicitis. Prognosis with CIP is highly unpredictable as illustrated by the devastating outcome in this report. Health care professionals need to be proactive in triaging early referral of children with suspected appendicitis.