Case Reports in Neurological Medicine

The case presented is that of a young male with postanoxic brain injury secondary to cocaine overdose who began to exhibit choreiform movements of the left upper extremity. Traditional treatment options for chorea were unsuccessful, leading to the administration of fentanyl, which rapidly resolved the patient’s choreiform movements. There is a limited research involving the treatment of chorea in anoxic brain injury as well as fentanyl’s role in the movement pathway. We hypothesize that chorea can be caused or exacerbated by opioid withdrawal in a patient with chronic opioid use through modulation of dopamine transmission.

Neurologic manifestations of sarcoidosis are rare, and even rarer still are cases of isolated neurosarcoidosis. The clinical presentation of isolated neurosarcoidosis can be highly variable, and diagnosis is particularly challenging, the gold standard being tissue biopsy. We describe a patient with a history of atypical parkinsonian syndrome and chronic right frontal lobe infarct who developed weakness, imbalance, and gait disequilibrium in 2008, with magnetic resonance imaging at that time showing leptomeningeal and nodular enhancements in the bilateral frontal and parietal lobes. The patient had an extensive negative workup in 2010 but ultimately did not receive a definitive diagnosis with a tissue biopsy until 2020. The patient also notably failed a 3-month course of steroids after his biopsy due to a lack of symptomatic improvement. This case highlights the clinical variability and diagnostic difficulties of isolated neurosarcoidosis. We also highlight that our patient did not have any symptomatic improvement on steroids, which do typically provide some relief for patients.

Although Bell’s palsy is a common diagnosis of acute isolated peripheral facial palsy (PFP), acute isolated PFP can be the first presentation of various illnesses, including COVID-19 disease. A female with a known history of well-controlled diabetes mellitus presented initially with acute isolated PFP mimicking Bell’s palsy. A course of oral prednisolone was given to treat acute PFP. Severe fifth cervical radicular pain, which is unusual for Bell’s palsy followed 3 days later. The COVID-19 infection was finally diagnosed by a real-time polymerase chain reaction (RT-PCR) test 15 days after facial paralysis when typical pulmonary infection symptoms developed. Oral favipiravir was given for the treatment of COVID-19 infection, to which the symptoms completely responded. The COVID-19 infection as a cause of acute isolated PFP should be added to the differential diagnosis of acute isolated PFP, albeit without typical pulmonary infection symptoms, particularly during the global pandemic of the infection.

Moyamoya disease is often diagnosed after intracranial hemorrhage in adult patients. Here, we report a case of unilateral moyamoya disease treated with indirect revascularization combined with cranioplasty after treatment for acute subdural hematoma and subcortical hemorrhage. A middle-aged woman with disturbed consciousness was transferred to our hospital. Computed tomography (CT) revealed an acute subdural hematoma with left temporoparietal subcortical hemorrhage. Three-dimensional CT angiography indicated a scarcely enhanced left middle cerebral artery (MCA) that was suspected to be delayed or nonfilling due to increased intracranial pressure. Subsequently, hematoma evacuation and external decompression were performed. Postoperative digital subtraction angiography (DSA) revealed stenosis of the left MCA and moyamoya vessels, indicating unilateral moyamoya disease. Forty-five days after the initial procedure, we performed encephalo-arterio-synangiosis (EAS) using the superficial temporal artery simultaneously with cranioplasty for the skull defect. The modified Rankin Scale score of the patient one year after discharge was 1, and the repeat DSA showed good patency of the EAS. Revascularization using EAS in the second step can be an option for revascularization for hemorrhagic moyamoya disease if the patient required cranioplasty for postoperative skull defect after decompressive craniotomy.

Diabetic striatopathy is a very rare neurological complication of diabetes. We report the case of an 86-year-old woman with poorly controlled type 2 diabetes admitted to the internal medicine ward for sudden onset of altered sensorium and severe bilateral choreiform and ballistic movements. The precise pathophysiology of this condition is not well understood. Our communication aims to remind clinicians to consider the possibility of diabetic striatopathy when poor-controlled diabetic patients have sudden-onset choreiform and ballistic movements. Moreover, this case suggests the possibility that oxidative and endoplasmic reticulum stress may be involved in this process.

We report a very rare case of referred pain associated with entrapment of the greater occipital nerve (GON) occurring not only in the ipsilateral hemiface but also in the ipsilateral limb. There is an extensive convergence of cutaneous, tooth pulp, visceral, neck, and muscle afferents onto nociceptive and nonnociceptive neurons in the trigeminal nucleus caudalis (medullary dorsal horn). In addition, nociceptive input from trigeminal, meningeal afferents projects into trigeminal nucleus caudalis and dorsal horn of C1 and C2. Together, they form a functional unit, the trigeminocervical complex (TCC). The nociceptive inflow from suboccipital and high cervical structures is mediated with small-diameter afferent fibers in the upper cervical roots terminating in the dorsal horn of the cervical cord extending from the C2 segment up to the medullary dorsal horn. The major afferent contribution is mediated by the spinal root C2 that is peripherally represented by the greater occipital nerve (GON). Convergence of afferent signals from the trigeminal nerve and the GON onto the TCC is regarded as an anatomical basis of pain referral in craniofacial pain and primary headache syndrome. Ipsilateral limb pain occurs long before the onset of the referred facial pain. The subsequent severe hemifacial pain suggested GON entrapment. The occipital nerve block provided temporary relief from facial and extremity pain. Imaging studies found a benign osteoma in the ipsilateral suboccipital bone, but no direct contact with GON was identified. During GON decompression, severe entrapment of the GON was observed by the tendinous aponeurotic edge of the trapezius muscle, but the osteoma had no contact with the nerve. Following GON decompression, the referred trigeminal and extremity pain completely disappeared. The pain referral from GON entrapment seems to be attributed to the sensitization and hypersensitivity of the trigeminocervical complex (TCC). The clinical manifestations of TCC hypersensitivity induced by chronic entrapment of GONs are diverse when considering the occurrence of extremity pain as well as facial pain.