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Congenital muscular torticollis (CMT) is the most common form of torticollis in children, significantly outnumbering orthopedic, neurologic, and ocular causes. CMT may present as a palpable sternomastoid tumor (SMT) or a simple tightness of the sternocleidomastoid muscle (SCM), designated as idiopathic muscular torticollis (IMT). Muscular torticollis has been associated with positional plagiocephaly in neonates who slept in the supine position. We have had difficulty in treating some of these combined cases by traditional methods such as physiotherapy, stretching exercises, and molding helmets. In November 2000, we began injecting botulinum toxin type A in cases in which there was persistent IMT, despite significant physical therapy input. The 15 patients included in this retrospective study all presented with IMT and positional plagiocephaly; all had responded poorly to conservative treatment, including physiotherapy, stretching exercises, or use of a helmet. In the attempt to avoid progression to surgical release, these patients were treated with botulinum toxin injected into the affected SCM and subsequent additional physiotherapy. All appeared to respond well, and a retrospective analysis of this treatment strategy was undertaken. Information gathered included a questionnaire, skull-shape tracings, and photographs. Independent outcome assessment data were then obtained from the regional child development teams and community physiotherapists. These results show that 14 of 15 children with recalcitrant IMT and positional plagiocephaly treated with botulinum toxin obtained sufficient improvement in neck range of motion and head position as to make surgical release of the muscle unnecessary. Our conclusion is that the use of botulinum toxin is a safe and effective adjunct to physical therapy in treating recalcitrant IMT; in selected cases, it may obviate the need for surgical release of a tight but nonfibrotic SCM.