Adult-onset Sacral Meningocele Causing a Specific Headache Triggered by Compression or Adoption of a Sitting or Supine Posture

Abstract

We report a rare case of adult-onset sacral meningocele where compression triggered a specific headache. A 46-year-old woman presented with a headache, which worsened when she was in a sitting or supine position. A subcutaneous mass was observed on her left buttock, the compression of which also induced headache. No neurological deficits were evident. Lumbar and sacral magnetic resonance imaging demonstrated a meningocele in the left dorsal buttock, connecting to the sacral cerebrospinal fluid (CSF) space, and spinal computed tomography revealed sacral dysplasia. Initial meningocele resection improved the patient's headache, but the cyst recurred 2 years later. Following repeated surgery to reinforce the meningocele orifice, the headache was relieved and has been absent for more than 6 years. The headache was due to intracranial pressure fluctuations due to CSF influx into and drainage from the meningocele. Meningocele development in adulthood can be owing to a spinal bone defect and pressure load on the spinal dura. Surgical resection can improve symptoms resulting from meningocele, and reinforcement of the orifice using an artificial surgical membrane effectively prevents recurrence.

Keywords: adult-onset; headache; meningocele; supine posture.

Fig. 1

A: Sagittal lumbosacral magnetic resonance imaging T2-weighted image showing a homogeneous, high-signal subcutaneous mass of 4 cm in diameter in the left upper gluteal cleft. The anteroposterior diameter of the mass appeared smaller than that measured with palpation, probably due to the patient being supine. B: The mass is contiguous from the dural sac of the sacral spine without any internal neural tissues, suggesting a diagnosis of meningocele. C: Three-dimensional computed tomography scan of the pelvis showing a defect of the left vertebral arch at the S4 and S5 levels and slight rightward deviation of the vertebral body.

Fig. 2

A: Lumbosacral magnetic resonance imaging (MRI) T2-weighted sagittal image after the first surgery. The meningocele was completely resected. B: Sagittal lumbosacral MRI T2-weighted image 2 years after the first surgery showing recurrence of the meningocele protruding from the sacral spine.