Paragonimiasis is a zoonosis that is caused by Paragonimus westermani. Humans infected with the Paragonimus species have been reported worldwide and about 293.8 million people are at risk [1]. Patients infected with Paragonimus westermani may experience fever, cough, abdominal pain, or seizures. Most cases present with symptoms related to the lungs. This study reports a rare case of an individual infected with cerebral paragonimiasis and presented as cerebral infarction and cranial venous sinus thrombosis.A 30-year-old man was admitted to our hospital because of sudden slow response and slurred speech for 3 days in July 2007. He also felt episodic numbness at right limbs. Neurologic examination showed alert, oriented, Wernicke aphasia. Right nasolabial fold was narrow and mouth deviated to left. Muscle strength of right upper limb was II/V, and other limbs were V/V. Bilateral Babinski sign was negative. Most laboratory tests were normal. Cranial MRI showed cerebral infarction of left temporal lobe and basal ganglia ( Figure 1A-C). MRA showed occlusion of left cerebral middle artery ( Figure 1D). Cerebrospinal fluid (CSF) analysis showed normal pressure, white cell counts, and total protein level. RPR and TPPA tests were negative. Patient was diagnosed with cerebral infarction and received aspirin 100 mg, qd. Several months later, the strength of right limbs returned to near normal.In October 2010, this patient was admitted to our hospital again because of recurrent flexion of upper limbs, reflexion of lower limbs, and loss of consciousness for 3 days. After treatment with diazepam, spasms were controlled gradually except the frequency of spasm in the right upper limb became higher. Neurologic examination showed lethargy, increased muscle tone, and a positive Babinski sign on the right limbs. Most laboratory tests were normal except D-dimer, which was elevated (1071 ug/L, rang: <200 ug/L). Cranial CEMRI suggested right frontal lobe lesion and gyrus enhancement, and left temporal lobe and basal ganglia cerebral infarction (Figure 2A-E). Cranial CTA showed significant stenosis of the left middle cerebral artery with a blockage after the M2 segment and significant stenosis of right middle cerebral artery ( Figure 2F). Administration of anti-epilepsy drugs (Levetiracetam tablets 0.5, po, qd; Sodium valproate tablets 0.2, po, tid; Phenobarbital injection, im, q8 h) showed limited effect. Limb spasms became more frequent, and the patient went into a lasting coma. The round nodule in the frontal lobe hinted at the possibility of parasitic infection. The CSF analysis showed mild increase in protein (58.2 mg/dL) and positive paragonimiasis antibody reaction. Considering the clinical findings and the patient's history of eating raw freshwater crabs, cerebral paragonimiasis was diagnosed and 2 g of praziquantel was administered. Low molecular weight heparins calcium injection, warevan tablet, and carbamazepine were also administered because cranial CEMRV identified sinus thrombosis in the superior sagittal sinus, sinuses sig...