Background Duplication of the spine is very rare, and this malformation is generally considered as a severe form of type I split cord malformations. To the best of our knowledge, this is the first reported case of spine duplication associated with lipomyelomeningocele. Case We report an exceptional case of 14-year-old, asymptomatic and neurologically intact girl with duplication of the spine and marked separation of bony elements at thoraco-lumbar region. One of the split thecal sacs includes a tethered spinal cord whereas other thecal sac has no visible neural content, and there is a neighbor lipomyelomeningocele located in the midline. Conclusion A surgical operation was planned to release the tethered cord and instrumentation and fusion for scoliosis; however, the operation was declined by the patient.
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