Abstract The interspinous region is an atypical source of low back pain, and it can often be difficult to identify. There are many structural components in the interspinous region that can contribute to interspinous pain, including the interspinous ligament, which plays a role in providing stabilization to the spine. Successful treatments of interspinous pain have not been well characterized in the literature. This case presents the first documentation of the use of extracorporeal shockwave therapy as a noninvasive treatment for refractory interspinous-related low back pain. Extracorporeal shockwave therapy has previously been shown to facilitate regeneration and tissue healing in tendons and ligaments but has not previously been used to treat interspinous pain. A 24-yr-old former collegiate softball player presented with 5 mos of low back pain; the interspinous ligament was clinically suspected as a pain generator, and this was confirmed via an ultrasound-guided injection. She underwent a course of physical therapy that improved function but did not improve pain, and nonsteroidal anti-inflammatory drugs only provided minimal and temporary relief. Three consecutive extracorporeal shockwave therapy treatment sessions provided 90% improvement in pain and function, and she was able to return to exercise and recreational sports. At more than 6 mos after extracorporeal shockwave therapy treatment, she reported no recurrences or functional limitations.