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Baastrup's disease represents a frequent, primarily radiological phenomenon on imaging studies of the spine. Nevertheless, it can present as a rare, symptomatically relevant pathology that implies a therapeutic consequence. Yet, there is little evidence and agreement on a consistent treatment strategy in the current literature. Here, we present the case of a 46-year-old man who presented with chronic, persistent midline back pain that was relieved by flexion and aggravated by spinal extension. Extensive imaging studies, including computed tomography, magnetic resonance imaging, and single-photon emission computed tomography confirmed the close approximation of the spinous processes at the levels L4-L5 and L5-S1. Clinically symptomatic isolated Baastrup's disease was confirmed by a local anesthetic infiltration test. As conservative treatment options failed, partial resection of the spinous processes was performed. Conservative treatment, including analgesics and physical therapy, represents the initial treatment approach for Baastrup's disease. When clinical features of Baastrup's disease are present, differential diagnoses have been excluded, and conventional therapy has been exhausted surgical decompression with low surgical risk and good prognosis may be indicated after careful consideration of the indications.