AbstractBackgroundAntimicrobial resistance and therapy‐related adverse effects make Mycobacterium abscessus treatment challenging. Omadacycline is a novel, bioavailable aminomethylcycline with favourable in vitro activity against M. abscessus.AimsTo describe a case report and review the published literature describing outcomes for M. abscessus infections treated with omadacycline.MethodsSystematic literature review.ResultsWe identified three articles that, in addition to our case report, describe 18 patients. Pulmonary infections were most frequent. Minimum inhibitory concentrations were reported for two isolates (0.25 and 0.5 mg/L). Despite half the patients starting omadacycline because of failure of prior therapy, 15 (83%) had a favourable outcome, defined as ‘cure’, ‘improvement’ or ‘clinical success’ as determined by the primary study authors. One patient (6%) discontinued omadacycline because of gastrointestinal intolerance.ConclusionsAlthough the limited observational data and in vitro susceptibility results are encouraging, randomised control trials are required to determine the role of omadacycline as part of combination therapy for this most difficult‐to‐treat pathogen.