Hemifacial spasm and trigeminal neuralgia in Chiari’s I malformation with hydrocephalus: Case report and literature review

•Hemifacial spasm (HFS) is one of the hyperactive cranial neuropathy that causes benign, chronic, involuntary paroxysmal contractions in the muscles on one side of the face.•The etiology of HFS has been generally accepted as the result of vascular compression of the intracranial portion of facial nerve, usually near the brainstem, namely the root exit zone (REZ). Trigeminal neuralgia (TN) is a unilateral paroxysmal facial pain in the distribution of the fifth cranial nerve (CN).•The etiology of TN is highly identical to HFS. There are small proportions of patients with different pathology except for neurovascular compression (NVC).•We report a patient with ipsilateral TN and HFS secondary to Chiari’s I malformation with hydrocephalus.•All of his clinical symptoms were resolved by a programmable ventriculoperitoneal shunt (VPS). This is the first report of one case only presenting as coexistent ipsilateral TN and HFS secondary to Chiari’s I malformation with hydrocephalus.

Chiari’s I malformation with hydrocephalus is commonly seen in clinical experience. Trigeminal neuralgia (TN) and hemifacial spasm (HFS) are most commonly related to vascular compression of the root entry/enter zone (REZ). Until now, TN and HFS associated with hydrocephalus caused by Chiari’s malformation have not been reported. The patient was a 24-year old male with left HFS and ipsilateral TN. Arnold–Chiari’s I malformation with hydrocephalus and platybasia were found in magnetic resonance imaging (MRI) of brain. We underwent a programmable ventriculoperitoneal shunt with complete resolution of all symptoms. This is the first report of one case only presenting as coexistent ipsilateral TN and HFS secondary to Chiari’s I malformation with hydrocephalus.