Lhermitte Sign Associated with Postdural Puncture Headache in a Parturient

POSTDURAL puncture headache (PDPH) is a potential complication of neuraxial anesthesia. A variety of associated symptoms have been reported. ¹ We present a unique case of PDPH associated with Lhermitte sign in a parturient.

Case Report

A healthy, 19-yr-old parturient (gravida 1 para 0) had an uneventful spontaneous vaginal delivery with epidural analgesia at another hospital. Approximately 2 h after delivery, she developed a severe postural headache, for which head computed tomography scan and lumbar puncture were performed. The results of both studies were negative, thereby supporting the diagnosis of PDPH. Accordingly, an epidural blood patch was performed the following day, which resulted in prompt resolution of her headache. However, her headache recurred and worsened over the next 48 h. In addition to recrudescence of her headache, the patient experienced severe, sharp pain associated with tingling that originated in her neck and radiated to her arms and chest. Both the pain and tingling worsened with neck flexion. Concerned about the possibility of an impending or evolving neurologic catastrophe, she was emergently transferred to our institution for further neurologic evaluation and treatment.

Neurologic and neurosurgical consultations corroborated the aforementioned neurologic physical examination findings reported by the referring physician. Magnetic resonance (MR) imaging and MR angiography of the head and spine demonstrated a substantial fluid collection extending from C4 to L4. The fluid was located in both the subdural and epidural spaces, depending on the level (figs. 1 and 2). Interestingly, the fluid collection resulted in anterior displacement of the spinal cord against the lower cervical and thoracic vertebral bodies (figs. 1 and 2). Based on the constellation of the patient's history, physical examination findings, and diagnostic study results, the final diagnosis was low cerebrospinal fluid pressure headache associated with Lhermitte sign.

Fig. 1

Fig. 2

The patient was treated with bedrest, intravenous fluids, caffeine, and morphine patient-controlled analgesia. Her symptoms resolved within 7 days without adverse sequelae. Repeat MR imaging performed 5 days after the initial scan demonstrated resolution of the epidural and subdural fluid collection (fig. 3).

Fig. 3

Discussion

Lhermitte sign, a sensory phenomenon that occurs during flexion of the neck, was first described by Marie and Chatelin in 1917 and later by Lhermitte in 1924. ¹ PDPH with associated neurologic symptoms is a well-documented potential complication of neuraxial anesthesia. ² However, we were unable to find any previously reported cases of PDPH with concurrent Lhermitte sign. Kelly et al.³ reported a case of a 27-yr-old man who developed postural, sharp, shooting pain radiating down both arms following diagnostic lumbar puncture that completely resolved with an epidural blood patch. Similarly, Page ⁴ reported a case in which a patient developed headache associated with unilateral upper extremity paresthesia in the C8 distribution. The patient experienced immediate relief from the headache following an epidural blood patch; however, the paresthesia persisted for 2 days. In both of these cases, it is unclear whether the sensory symptoms were exacerbated during neck flexion, and, thus, neither can be described as Lhermitte sign.

Lhermitte sign is a sensory phenomenon that occurs during flexion of the neck. ^5-7 Clinically, it is characterized as an electric shock-like or tingling sensation that originates in the neck or upper thorax and radiates down the spine into the extremities. ^5-7 Although this sign is not pathognomonic for a particular disease, it has been reported to occur in patients diagnosed with multiple sclerosis, cervical spondylotic myelopathy, neck trauma, spinal cord tumor, Chiari malformation, pseudotumor cerebri, systemic lupus erythematosus, herpes zoster, and vitamin B12 deficiency. ^5-7 It also has been observed following cervical radiation or cisplatin chemotherapy. ^5,6 However, Lhermitte sign has not been previously described following dural puncture.

Although the etiology of Lhermitte sign has yet to be fully elucidated, its pathogenesis is thought to be related to injury (e.g., compression, distortion, inflammation, radiation, or metabolic or toxic aberrations) of the cervical dorsal columns. ^5-7 After injury, the damaged dorsal column axons or cell bodies have increased mechanosensitivity, producing ectopic action potential impulses that occur with greatest frequency during cervical flexion (i.e., a maneuver that can alter spinal cord length by 1 to 2 cm). ^5,6

We suspect that accidental dural puncture during labor epidural placement (compounded by intentional dural puncture during diagnostic work-up and epidural blood patch) was responsible for our patient's signs and symptoms. That is, the dural rent allowed a sufficient volume of cerebrospinal fluid to escape into the subdural and epidural spaces, resulting in anterior displacement of the spinal cord and compression of the dorsal columns (figs. 1 and 2). In support of our hypothesis, resolution of the patient's clinical findings coincided with resorption of the fluid collections (as demonstrated on repeat MR image;fig. 3).

Conclusions

Neurologic complications associated with PDPH occur and may require neurologic consultation to elucidate the etiology, to facilitate treatment, and to anticipate the patient's prognosis. Our patient's neurologic findings invoked trepidation of a catastrophic neurologic event. However, with conservative management, she recovered completely without adverse sequelae.

The authors thank Charlotte H. Rydberg, M.D. (Assistant Professor, Department of Radiology, Mayo Clinic and Mayo Foundation, Rochester, Minnesota), and her colleagues for providing their neuroradiologic expertise to interpret the MR images provided in this case report.