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Neonatal extremity compartment syndrome is an extremely rare diagnosis. Risk factors that predispose infants to a hypercoagulable state or trauma have been implicated, but the exact mechanisms remain poorly understood. The hallmark of the condition is extremity swelling with sentinel skin changes. We report a case of upper extremity compartment syndrome from initial presentation until 3 months after discharge and discuss the importance of prompt diagnosis and timely surgical evaluation., Competing Interests: Conflict of Interest All authors have no conflicts of interest to disclose., (The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. ( https://creativecommons.org/licenses/by-nc-nd/4.0/ ).)