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Paper text:
here , we are presenting two very rare cases we came across in a span of 1 year .
one of the patients was a 9-year - old boy born of nonconsanguineous marriage , presented with fever for 5 days along with the acute onset of dizziness followed by unsteady gait and jerkiness of both eyes and abnormal jerks of limbs .
his past medical , immunization , family , and social history were unremarkable . on examination , he was conscious and alert .
blood examination revealed mild pallor with falciparum tropozoite ; plasmodium falciparum antigen ( plasmodium lactate dehydrogenase [ pldh ] ) was positive .
urinary catecholamine testing and cerebrospinal fluid ( csf ) analysis including herpes simplex polymerase chain reaction and antibody for mycoplasma , antibody ( igm ) viral capsid antigen of epstein - barr virus ( ebv ) were normal .
magnetic resonance imaging ( mri ) of the brain with and without contrast was normal .
computed tomography ( ct ) scans of chest , pelvis , and abdomen ( done immediately and 3 months later ) did not reveal neuroblastoma .
we started acth at a dose 75 iu / m / day on the 4 day with continuation of artesunate , clindamycin , and other supportive measures .
fever subsided within 48 h. neurologic symptom did not improve significantly over 2 weeks , so we doubled the dose .
another well - built boy of 6 years presented with a similar history of fever which was diagnosed to have p. falciparum malaria ( pldh antigen positive ) referred to us for ataxia .
all general and neurological examination showed that eyes were normal except cerebellar signs  truncal ataxia , abnormal coordination tests , and mild hypotonia .
after 3 weeks , abnormal chaotic multidirectional rapid saccadic movement without intersaccadic interval suggesting opsoclonus developed .
repeat mri of the brain , neuroblastoma screening with ct thorax and abdomen , and urine catecholamine analysis were unremarkable .
we started high - dose acth ( 75 u / m /dose twice daily ) with our prior experience .
he responded in 2 weeks . at present , he is also fine with a tapering dose of acth and a monthly dose of ivig
the first child is now without medication . during follow - up , no relapse occurred .
side effect was monitored closely for cushingoid feature , hypertension , headache , obesity , and infection .
the pathophysiology of oma has been speculated to involve igg and igm autoantibodies directed against neural antigens in cerebellar purkinje cells , cerebral cortical neurons , and axons .
the immune mechanism may be a type ii or iv hypersensitivity . up to 50% of children with oma harbor neuroblastoma ; rest are associated with flu vaccine , infection with streptococcus ebv , mycoplasma pneumoniae , st .
treatment for oms from whatever the cause has not been uniformly successful . in children , this syndrome usually resolves with acth , or corticosteroid , but in most of the patients , symptoms recur after withdrawal .
most authors believe that in adults this syndrome needs no treatment as symptoms are self - limiting and resolve within 68 weeks .
there are other reports indicating that clonazepam , baclofen , valproate , and 5-hydroxytryptophan had provided much needed symptomatic relief to these patients . in children , as per the guideline of the national organization for rare disorders , acth , ivig , and rituximab are the best options .
some authority shows beneficial effect of steroid in the form of pulse methyl prednisolone and oral prednisolone , but the national organization for rare disorders did not demonstrate any benefit .
we have shown that high - dose acth and ivig are safe and there was no relapse .
minimal side effects such as cushingoid feature and systolic blood pressure just above 90 percentile were documented which responded to salt restriction and disappeared with stoppage of therapy in the first child .
the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s )
has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal .
the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed .
the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s )
has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal .
the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed .
opsoclonus - myoclonus ataxia ( oma ) syndrome is rare in children , mostly caused by neuroblastoma . here 
 , we present two very rare cases presenting with oma due to falciparum malaria . both of them responded to a high dose of adrenocorticotrophin hormone and intravenous immunoglobulin without recurrence and complication .