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Paper text:
hashimoto encephalopathy ( he ) is defined as a steroid responsive encephalopathy associated with autoimmune thyroiditis ( 1 ) .
symptoms are non - specific and include a wide spectrum of neurologic deficits . among all neurologic symptoms ,
herein , we report a case of he with an unusual presentation of drug resistant status epilepticus . a 33 yr old man , previously diagnosed with hypothyroidism , presented with decreased level of consciousness and generalized tonic - clonic ( gtc ) seizure to namazi hospital , shiraz , iran , during april , 2015 .
the seizure lasted for about 12 min and the patient recovered completely after that episode , so the patient did not refer for any medical care .
after a few days , the patient experienced another seizure - like attack and developed urinary and defecation incontinency , aphasia , total body weakness and disorientation . at that time
, the patient was admitted to a hospital and in 7 days of his admission , his signs and symptoms improved and he was discharged with phenytoin , carbamazepine and levothyroxine .
after these events , the patient had three more hospital admissions due to similar symptoms .
after a one - month symptom free period , he experienced another gtc seizure attack .
the patient experienced four episodes of gtc seizures during the 24 h prior to his last admission , during which the patient did not regain consciousness after the second episode of gtc seizure . on admission ,
the gtc seizures were controlled by administering intra venous phenytoin . during the hospital admission ,
the patient developed myoclonic jerks , followed by mental disorders including aphasia , ataxia , loss of verbal communication , eye contact and complete loss of obedience .
the patient also developed urinary tract infection for which he received ceftriaxone and was resolved .
his laboratory tests for viral markers including the human immunodeficiency and hepatitis c viral antibodies and hepatitis b virus antigen were all negative .
his serum anti - thyroid peroxidase antibody ( anti - tpo ab ) and blood ammonia levels were high ( 1095.8 iu / ml and 189.3 mol / l , respectively ) .
his cpk , lactate dehydrogenase ( ldh ) and crp levels were 137 mg / l , 437 u / l and 58 mg / l , respectively .
thyroid function tests ( tft ) showed low levels of t3 and t4 with high levels of thyroid stimulating hormone ( tsh ) .
other blood tests , including complete blood count , liver function test , kidney function tests and electrolytes , erythrocyte sedimentation rate , homocysteine levels and the peripheral blood smear were all normal .
brain magnetic resonance imaging ( mri ) and brain computed tomography ( ct ) scan were normal .
various anticonvulsant and antipsychotic medications were prescribed for the patient after he developed myoclonic jerk movements and other mental disorders .
the combined treatment controlled the myoclonic jerks but did not improve the mental problems . with no significant improvement in his condition , he underwent two lumbar punctures ( lp ) .
the first lp was to rule out herpes encephalopathy and the second was done to check the possibility of he .
the first cerebrospinal fluid ( csf ) analysis showed an elevated cell count ( 19500/mm ) with two wbcs ( two segments ) , elevated protein levels ( 56 mg / dl ) and normal glucose and ldh levels .
the second csf analysis had a total cell count of 1600/mm with a total wbc count of 10 ( five lymphocytes , 5 segments ) and a high titer of anti - tpo antibodies . with high suspicion of he ,
as the first line of treatment , pulsed methyl prednisolone ( 10 mg ) was administered , however the patient showed little improvement .
a 33 yr old man , previously diagnosed with hypothyroidism , presented with decreased level of consciousness and generalized tonic - clonic ( gtc ) seizure to namazi hospital , shiraz , iran , during april , 2015 .
the seizure lasted for about 12 min and the patient recovered completely after that episode , so the patient did not refer for any medical care .
after a few days , the patient experienced another seizure - like attack and developed urinary and defecation incontinency , aphasia , total body weakness and disorientation . at that time
, the patient was admitted to a hospital and in 7 days of his admission , his signs and symptoms improved and he was discharged with phenytoin , carbamazepine and levothyroxine .
after these events , the patient had three more hospital admissions due to similar symptoms .
after a one - month symptom free period , he experienced another gtc seizure attack .
the patient experienced four episodes of gtc seizures during the 24 h prior to his last admission , during which the patient did not regain consciousness after the second episode of gtc seizure . on admission ,
the gtc seizures were controlled by administering intra venous phenytoin . during the hospital admission ,
the patient developed myoclonic jerks , followed by mental disorders including aphasia , ataxia , loss of verbal communication , eye contact and complete loss of obedience .
the patient also developed urinary tract infection for which he received ceftriaxone and was resolved .
his laboratory tests for viral markers including the human immunodeficiency and hepatitis c viral antibodies and hepatitis b virus antigen were all negative .
his serum anti - thyroid peroxidase antibody ( anti - tpo ab ) and blood ammonia levels were high ( 1095.8 iu / ml and 189.3 mol / l , respectively ) .
his cpk , lactate dehydrogenase ( ldh ) and crp levels were 137 mg / l , 437 u / l and 58 mg / l , respectively .
thyroid function tests ( tft ) showed low levels of t3 and t4 with high levels of thyroid stimulating hormone ( tsh ) .
other blood tests , including complete blood count , liver function test , kidney function tests and electrolytes , erythrocyte sedimentation rate , homocysteine levels and the peripheral blood smear were all normal .
brain magnetic resonance imaging ( mri ) and brain computed tomography ( ct ) scan were normal .
various anticonvulsant and antipsychotic medications were prescribed for the patient after he developed myoclonic jerk movements and other mental disorders .
the combined treatment controlled the myoclonic jerks but did not improve the mental problems . with no significant improvement in his condition , he underwent two lumbar punctures ( lp ) .
the first lp was to rule out herpes encephalopathy and the second was done to check the possibility of he .
the first cerebrospinal fluid ( csf ) analysis showed an elevated cell count ( 19500/mm ) with two wbcs ( two segments ) , elevated protein levels ( 56 mg / dl ) and normal glucose and ldh levels .
the second csf analysis had a total cell count of 1600/mm with a total wbc count of 10 ( five lymphocytes , 5 segments ) and a high titer of anti - tpo antibodies . with high suspicion of he ,
as the first line of treatment , pulsed methyl prednisolone ( 10 mg ) was administered , however the patient showed little improvement .
with a prevalence of 2.1 in 100000 ( 3 ) , he is considered a rare condition . due to its vague clinical manifestations ( 2 ) and uncertain diagnostic tests ,
the diagnosis is made when high anti - thyroid antibodies accompany encephalopathy symptoms ( 4 ) , and no other infectious , metabolic or paraneoplastic processes are responsible for a patient s condition ( 3 ) .
first the vasculitic type and second the indolent progressive type , with the former including acute stroke - like episodes ( 27% ) , seizures ( 66% ) and psychosis ( 38% ) and the latter including dementia , myoclonus and altered consciousness ( 3 ) . recently a third set of symptoms has been introduced , as the relapsingremitting type , with patients showing cognitive dysfunction and psychiatric symptoms ( 5 ) .
our patient developed status epilepticus along with other non - specific neurological symptoms such as aphasia , incontinency and disorientation . in hashimoto encephalopathy ,
patients most commonly present with seizures , but status epilepticus is a rare symptom to present , manifesting in an estimated 12% of patients ( 2 ) . to the best of our knowledge , very few cases of he have been reported in literature with such a presentation .
as stated earlier , he presents with a wide variety of symptoms and serological tests and imaging studies usually reveal non - specific results .
however , they are necessary to rule out other possible causes of encephalopathy ( 2 , 3 ) . at the time of the disease onset ,
thyroid function may vary . as reported , patients are mainly hypothyroid or euthyroid , as in our patient who was a known case of hypothyroidism and presented with hypothyroidism on admission and during his disease course .
brain imaging findings are non - specific , showing ischemia , granuloma or degenerative process ( 5 ) .
although the changes are non - specific , more than 50% of patients have an abnormal brain imaging from the onset of their disease course , with the most common finding being cerebral atrophy ( 1 ) . in our case
eeg is abnormal in almost 100% of the patients , mostly showing transient epileptic activity ( 1 , 3 ) .
csf findings mostly document increased cerebral fluid protein and pleocytosis ( 3 , 5 ) .
after our patient displayed resistance to antiepileptic medication , lp was performed and csf analysis showed elevated protein and pleocytosis along with high anti - tpo ab titer ( 6 ) . initially to control the symptoms , administration of antiepileptic and thyroid modulatory medications is common ( 7 ) . then to control the disease from its basic pathogenesis ,
first line immunosuppressive medications are corticosteroids , to which most patients show dramatic response , for which the disease is also termed
only 10% of the patients do not respond to steroids , for whom alternative treatments such as ivig , plasma exchange should be started ( 3 ) . informed and written consent was obtained from the patient before reporting the case .
first the vasculitic type and second the indolent progressive type , with the former including acute stroke - like episodes ( 27% ) , seizures ( 66% ) and psychosis ( 38% ) and the latter including dementia , myoclonus and altered consciousness ( 3 ) . recently a third set of symptoms has been introduced , as the relapsingremitting type , with patients showing cognitive dysfunction and psychiatric symptoms ( 5 ) .
our patient developed status epilepticus along with other non - specific neurological symptoms such as aphasia , incontinency and disorientation . in hashimoto encephalopathy ,
patients most commonly present with seizures , but status epilepticus is a rare symptom to present , manifesting in an estimated 12% of patients ( 2 ) . to the best of our knowledge , very few cases of he have been reported in literature with such a presentation .
as stated earlier , he presents with a wide variety of symptoms and serological tests and imaging studies usually reveal non - specific results .
however , they are necessary to rule out other possible causes of encephalopathy ( 2 , 3 ) . at the time of the disease onset , thyroid function may vary . as reported , patients are mainly hypothyroid or euthyroid , as in our patient who was a known case of hypothyroidism and presented with hypothyroidism on admission and during his disease course .
brain imaging findings are non - specific , showing ischemia , granuloma or degenerative process ( 5 ) .
although the changes are non - specific , more than 50% of patients have an abnormal brain imaging from the onset of their disease course , with the most common finding being cerebral atrophy ( 1 ) . in our case both brain mri and ct scan were normal .
eeg is abnormal in almost 100% of the patients , mostly showing transient epileptic activity ( 1 , 3 ) .
csf findings mostly document increased cerebral fluid protein and pleocytosis ( 3 , 5 ) .
after our patient displayed resistance to antiepileptic medication , lp was performed and csf analysis showed elevated protein and pleocytosis along with high anti - tpo ab titer ( 6 ) .
initially to control the symptoms , administration of antiepileptic and thyroid modulatory medications is common ( 7 ) . then to control the disease from its basic pathogenesis ,
first line immunosuppressive medications are corticosteroids , to which most patients show dramatic response , for which the disease is also termed
only 10% of the patients do not respond to steroids , for whom alternative treatments such as ivig , plasma exchange should be started ( 3 ) .
if diagnosed and treated in time , he patients recover with minimal complications . on the other hand , delay in correct treatment
due to the non - specific symptoms and laboratory findings , he is easily missed .
based on our patient we recommend thyroid function tests , serum , and csf anti - thyroid antibodies be evaluated in all patients with encephalopathy or anti - epileptic - resistant seizures including status epilepticus .
a 33 yr old man , previously diagnosed with hypothyroidism , presented with decreased level of consciousness and generalized tonic - clonic ( gtc ) seizure to namazi hospital , shiraz , iran , during april 2015 . 
 the patient later referred with another episode of seizure like attack for which he received phenytoin , carbamazepine and levothyroxine and was discharged . during his last admission , the patient was admitted with chief complaints of decreased consciousness and four gtc attacks . on admission , 
 the patients had aphasia , ataxia , loss of verbal communication , eye contact and complete loss of obedience . 
 thyroid function tests showed low levels of t3 and t4 with high levels of thyroid stimulating hormone . 
 other blood tests were all either normal or slightly abnormal . 
 lumbar puncture and csf analysis had a high titer of anti - tpo antibodies . with high suspicion of hashimoto encephalopathy , pulsed methyl prednisolone ( 10 mg ) 
 was administered , however the patient showed little improvement . 
 therefore , plasmaphresis was started , to which the patient showed dramatic response .