Biopsies of multifidus muscles were procured from patients with idiopathic scoliosis prior to Harrington rod instrumentation. Specimens from both convex and concave sides at the apex of the curve were examined by light and electron microscopy and compared with normal muscle samples. Abnormalities were detected on the concave aspect of the curve and the most dramatic morphological changes were noted at the myotendinous junction. Here a structural defect in the form of discontinuities in the sarcolemmal membranes of some muscle fibres was accompanied by large numbers of intimately-adhering connective tissue cells. Structural disorganization of the associated tendon occurred in conjunction with increased vascularization and with fatty cell and leukocyte infiltration. Further from the myotendon junction, structural lesions appeared more chronic and non-specific subsequent to the incipient sarcolemmal break in the affected muscle fibres. Hypertrophy, atrophy, centralization of nuclei, and disruption of sarcotubular and myofibrillar elements were noted in some muscle cells. While the aetiology of this disorder is unknown, a supposition is made that the primary change is an inherent weakness and subsequent break in the sarcolemma at the myotendon junction. This site is an important clue to the pathogenesis of this disease since it reflects morphological change in rapidly growing tissue occurring at the time of the rapid adolescent growth spurt leading to progression of the scoliotic curve.