Psychosis Secondary to Recurrent Spinal Meningioma
This case report discusses a rare instance of psychosis secondary to recurrent spinal meningioma, highlighting the association between anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis and spinal meningioma. The patient, a 27-year-old man with no prior psychiatric history, presented with a 2-month history of apathy, decreased responsiveness, and malaise. These symptoms were preceded by fatigue and insomnia. The patient had undergone spinal meningioma resection four years earlier, after which similar symptoms had resolved. However, four days after the onset of the current symptoms, the patient developed mutism and irresponsiveness, leading to a diagnosis of organic psychosis and admission to a psychiatric unit.
Initial treatment with antipsychotic medications, including olanzapine, escitalopram, sertraline, and oxazepam, failed to improve the patient’s condition. Cerebrospinal fluid (CSF) analysis revealed a leukocyte count of 56 with 90% lymphocytosis, normal glucose and protein levels, and negative results for serum tumor markers and serologic tests for viral and communicable infections. However, antibodies against NMDAR were detected in both serum and CSF, confirming the diagnosis of anti-NMDAR encephalitis. Despite treatment with human immunoglobulin at 0.4 mg/kg per day for five days, there was no clinical improvement.
Further investigations included computed tomography (CT) scans of the chest, abdomen, and pelvis, which showed no signs of occult neoplasms. A repeat lumbar puncture two weeks later revealed increased nucleated cells (160 × 10^6/L, reference range 0–5 × 10^6/L) with 80% lymphocytes, elevated protein levels (1196.91 mg/L, reference range 150–450 mg/L), and a mild decrease in chloride levels (118.4 mmol/L, reference range 120–132 mmol/L). NMDAR antibodies were again detected in both CSF and serum, with titers of 1:32 and 1:100, respectively. Brain CT and magnetic resonance imaging (MRI) revealed multiple hypodense abnormalities in the cerebral cortex and nodular intensity enhancement in the cervical cord, indicating a recurrence of spinal meningioma.
The patient underwent emergency surgery, and pathological examination confirmed recurrent benign spinal meningioma on the cervical cord. Postoperatively, the patient’s clinical symptoms improved significantly.
Anti-NMDAR encephalitis is the most common cause of autoimmune encephalitis, characterized by psychiatric or cognitive symptoms and typically associated with antibodies against neuronal cell-surface receptors. The first symptoms of anti-NMDAR encephalitis are usually psychoses or cognitive disturbances. Spinal meningioma, a benign neoplasm most commonly found in the thoracic spines of middle-aged women, presents with pain, weakness, and sensory loss. Recurrence rates after surgical resection range from 1.3% to 14.7%. While tumors underlying anti-NMDAR encephalitis are usually teratomas, thymomas, or small cell lung carcinomas, this case suggests that spinal meningioma should also be considered as a potential etiology.
The pathogenesis of spinal meningioma remains unclear, but it has a tendency to relapse and can manifest with complications such as psychosis or acute behavioral changes post-surgery. Therefore, vigilance for tumor recurrence is crucial if surgical resection does not induce clinical improvement. The decrease in antibody titer after tumor resection may explain symptom remission, though this requires further study.
In conclusion, anti-NMDAR encephalitis accompanied by isolated behavioral changes is a rare phenomenon, and its association with recurrent spinal meningioma has not been previously reported. This case underscores the importance of considering central nervous system neoplasms in the differential diagnosis of anti-NMDAR encephalitis, especially in patients with a history of such neoplasms. Psychiatrists and neurosurgeons should be aware of the clinical characteristics and potential etiologies of this syndrome. Further research is needed to explore whether benign tumors are more likely to be associated with NMDAR encephalitis and the mechanisms underlying symptom remission after surgery.
doi.org/10.1097/CM9.0000000000000435
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