IntroductionLangerhans cell histiocytosis (LCH) occurs rarely in the spine of adults. The radiological findings usually resemble vertebral tumors. Etiology of LCH has not been clearly established yet. Therapeutic approaches are still controversial. We describe a case of LCH in an adult spine.Case descriptionA patient who presented with low back pain had an osteolytic lesion in the L1 vertebral body without neurological deficits, and fluoroscopy-guided needle biopsy of the L1 vertebral body was performed. The immunohistochemical diagnosis confirmed LCH. The patient was successfully treated with conservative methods.DiscussionThe choice of appropriate therapy is very important, with treatment options varying from watch-and to aggressive treatment.ConclusionLCH is considered as a pediatric disease that is extremely rare in the spine of adults and should be include in the differential diagnosis of osteolytic vertebral lesions. Conservative treatment is best choice for a patient with LCH without neurological deficit or spinal instability.