the cause of non - herpetic limbic encephalitis is often uncertain , but it has recently been suggested that anti - glur antibody - positive encephalitis may contribute to development of the condition.13 symptoms such as parkinsonism and autonomic failure have been reported in non - herpetic limbic encephalitis.4 the lesions present in non - herpetic limbic encephalitis are typically detected by magnetic resonance imaging ( mri ) during the early phase . there are several case reports regarding mri patterns of anti - n - methyl - d - aspartate receptor ( nmdar ) and anti - voltage gated potassium channel ( vgkc ) antibody encephalitis.5,6 however , case reports regarding anti - glur antibody - positive encephalitis mri are uncommon , and the relationship between anti - glur and anti - nmdar remains unclear . here , we report two unique cases in males with parkinsonism and autonomic failure , as well as atypical mri legions during a later phase . forty - eight days later , he experienced hearing loss and tinnitus in the left ear ; by day 58 , his walking had become unsteady . his cognition gradually became impaired and he was transferred to our hospital 77 days after symptom onset . his consciousness was stupor with glasgow coma scale ( gcs ) at 11 ( e3v3m5 ) . cogwheel rigidity was observed in his bilateral upper limbs , although he was not taking any medication known to cause parkinsonism . his gait was wide - based and unstable , and nuchal rigidity and kernig sign were evident . laboratory examination showed a white cell count of 11,610/l and a serum reactive c - protein of 0.49 mg / dl . cartinoembryonic antigen ( cea ) and carbohydrate antigen 19 - 9 ( ca19 - 9 ) were normal ( 3.68 ng / ml and 1.2 u / ml , respectively ) . an mri during the early clinical phase ( day 78 and 95 ) was unremarkable ( fig . single photon emission computed tomography ( spect ) with tc - ethylcysteinate dimer ( tc - ecd ) showed a mild decrease in bilateral frontal and left parietal cerebrocortical blood flow . a lumbar puncture on the first hospital day revealed a cell count of 63/l ( monocyte 48/l , polynuclear cell 15/l ) , an elevated protein level of 72 mg / dl , and a sugar level of 50 mg / dl compared with a serum level of 86 mg / dl . interleukin-6 in serum was elevated to 519 pg / ml and in csf to 911 pg / ml . autoimmune encephalitis was suspected , and methylprednisolone pulse therapy ( 1,000 mg / day ) was performed twice : at 7880 days and 8587 days from onset . oral prednisolone 60 mg / day ( 1 mg / kg / day ) was initiated , but his consciousness worsened with gcs , dropping to 8 ( e2v1m5 ) at 98 days . although the deep tendon reflex was normal on the day of admission , it became hyperreflexia , and bilateral babinski and chaddock reflexes became positive at 98 days . in an attempt to improve his encephalitis , plasma exchange therapy was performed at 99 , 101 , 103 , and 105 days . this was followed by two doses of methylprednisolone pulse therapy , which gradually improved his symptoms . during the course of symptom improvement , mri signal abnormalities became evident in the bilateral claustrum , medial of the frontal lobe , with periventricular lesions , at 120 days ( fig . when he finally returned home after 167 days , his consciousness was alert with gcs of 15 ( e4v5m6 ) and mmse of 24 . anti - glur2 and 2 antibodies were positive in the csf on the first hospital day , but not in serum after his illness improved ( table 1 ) . anti - vgkc antibody was negative . a 52-year - old previously healthy man developed a fever of 39.5 c and a headache , and four days later gait disturbance appeared accompanied by pollakiuria ( approximately 16 times per day ) and impaired cognition . his consciousness was stupor with gcs at 12 ( e4v4m4 ) , and nuchal rigidity and kernig sign were evident . serum osmotic pressure was 243 mosm / kg  h2o , serum sodium 118 meq / l , urinary osmotic pressure 799 mosm / kg  h2o , and urinary sodium 164 meq / l . cea and ca19 - 9 were normal ( 3.12 ng / ml and 0.8 u / ml , respectively ) . abdominal x - ray showed colon expansion and the patient was diagnosed with paralytic ileus . 1b ) . a cranial mri during the early clinical phase ( day 8) showed relatively high intensity of the right posterior cortex ( fig . the spect with tc - ecd showed a severe decrease in diffuse cerebrocortical blood flow . a lumbar puncture on the first hospital day revealed a cell count of 68/l ( monocyte 68/l , polynuclear 0/l ) , an elevated protein level of 279 mg / dl , and a sugar level of 50 mg / dl compared with a serum level of 78 mg / dl . because autoimmune encephalitis was also suspected , methylprednisolone pulse therapy ( 1,000 mg / day ) was performed for 3 days from days 9 to 11 . his cognition began to improve from day 12 , and cranial mri showed abnormal intensities in the bilateral basal ganglia at day 19 and in the pons at day 57 ( fig . he was able to care for himself at home with his wife and he resumed working . anti - glur2 and 2 antibodies were positive in csf on the first hospital day , but not in the serum after his illness improved ( table 1 ) . forty - eight days later , he experienced hearing loss and tinnitus in the left ear ; by day 58 , his walking had become unsteady . his cognition gradually became impaired and he was transferred to our hospital 77 days after symptom onset . his consciousness was stupor with glasgow coma scale ( gcs ) at 11 ( e3v3m5 ) . cogwheel rigidity was observed in his bilateral upper limbs , although he was not taking any medication known to cause parkinsonism . his gait was wide - based and unstable , and nuchal rigidity and kernig sign were evident . laboratory examination showed a white cell count of 11,610/l and a serum reactive c - protein of 0.49 mg / dl . cartinoembryonic antigen ( cea ) and carbohydrate antigen 19 - 9 ( ca19 - 9 ) were normal ( 3.68 ng / ml and 1.2 u / ml , respectively ) . an mri during the early clinical phase ( day 78 and 95 ) was unremarkable ( fig . single photon emission computed tomography ( spect ) with tc - ethylcysteinate dimer ( tc - ecd ) showed a mild decrease in bilateral frontal and left parietal cerebrocortical blood flow . a lumbar puncture on the first hospital day revealed a cell count of 63/l ( monocyte 48/l , polynuclear cell 15/l ) , an elevated protein level of 72 mg / dl , and a sugar level of 50 mg / dl compared with a serum level of 86 mg / dl . interleukin-6 in serum was elevated to 519 pg / ml and in csf to 911 pg / ml . autoimmune encephalitis was suspected , and methylprednisolone pulse therapy ( 1,000 mg / day ) was performed twice : at 7880 days and 8587 days from onset . oral prednisolone 60 mg / day ( 1 mg / kg / day ) was initiated , but his consciousness worsened with gcs , dropping to 8 ( e2v1m5 ) at 98 days . although the deep tendon reflex was normal on the day of admission , it became hyperreflexia , and bilateral babinski and chaddock reflexes became positive at 98 days . in an attempt to improve his encephalitis , plasma exchange therapy was performed at 99 , 101 , 103 , and 105 days . this was followed by two doses of methylprednisolone pulse therapy , which gradually improved his symptoms . during the course of symptom improvement , mri signal abnormalities became evident in the bilateral claustrum , medial of the frontal lobe , with periventricular lesions , at 120 days ( fig . when he finally returned home after 167 days , his consciousness was alert with gcs of 15 ( e4v5m6 ) and mmse of 24 . anti - glur2 and 2 antibodies were positive in the csf on the first hospital day , but not in serum after his illness improved ( table 1 ) . a 52-year - old previously healthy man developed a fever of 39.5 c and a headache , and four days later gait disturbance appeared accompanied by pollakiuria ( approximately 16 times per day ) and impaired cognition . his consciousness was stupor with gcs at 12 ( e4v4m4 ) , and nuchal rigidity and kernig sign were evident . serum osmotic pressure was 243 mosm / kg  h2o , serum sodium 118 meq / l , urinary osmotic pressure 799 mosm / kg  h2o , and urinary sodium 164 meq / l . cea and ca19 - 9 were normal ( 3.12 ng / ml and 0.8 u / ml , respectively ) . abdominal x - ray showed colon expansion and the patient was diagnosed with paralytic ileus . 1b ) . a cranial mri during the early clinical phase ( day 8) showed relatively high intensity of the right posterior cortex ( fig . the spect with tc - ecd showed a severe decrease in diffuse cerebrocortical blood flow . a lumbar puncture on the first hospital day revealed a cell count of 68/l ( monocyte 68/l , polynuclear 0/l ) , an elevated protein level of 279 mg / dl , and a sugar level of 50 mg / dl compared with a serum level of 78 mg / dl . because autoimmune encephalitis was also suspected , methylprednisolone pulse therapy ( 1,000 mg / day ) was performed for 3 days from days 9 to 11 . his cognition began to improve from day 12 , and cranial mri showed abnormal intensities in the bilateral basal ganglia at day 19 and in the pons at day 57 ( fig . he was able to care for himself at home with his wife and he resumed working . anti - glur2 and 2 antibodies were positive in csf on the first hospital day , but not in the serum after his illness improved ( table 1 ) . in addition to disturbance of recognition , parkinsonism , and myoclonus appeared in case 1 and urinary retention , paralytic ileus , and siadh appeared in case 2 . in case 2 , whether pons inflammation involved hyponatremia remains unknown since the abnormal intensity of the mri appeared after hyponatremia had diminished . . therefore , there may be several clinical types of anti - glur antibody - positive encephalitis . both cases showed slightly increased mri intensity during the early phase , which later became evident at 120 days ( case 1 ) and 57 days ( case 2 ) . non - herpetic limbic encephalitis frequently shows mri abnormalities in the limbic system , but the present cases showed later appearance of abnormal intensities in bilateral claustrum and the pons . the relationship between the symptom and mri abnormal area of anti - glur antibody - positive encephalitis may clear the mechanism of encephalitis . these cases demonstrate the importance of considering anti - glur antibody - positive encephalitis even when there is no abnormal intensity in the cranial mri in the early phase .
we report two cases of anti - glutamic acid receptor ( anti - glur ) antibody - positive encephalitis in males with symptoms such as parkinsonism , urinary retention , and paralytic ileus . 
 although non - herpetic encephalitis typically shows magnetic resonance imaging ( mri ) lesions in the limbic system during early stages , the present cases showed mri lesions during later stages in the bilateral claustrum and pons . in both cases , 
 anti - glur2 and 2 antibodies were later shown to be positive in the cerebrospinal fluid but negative in the serum . 
 although early detection of anti - glur antibodies is essential , early treatment may be significantly more important .