Isolated abducens palsy in adolescent girl with Kawasaki disease

Abstract

Listen

Translate article

Kawasaki disease (KD) represents one of the most common types of pediatric vasculitis. To date, there has been one case that is associated with abducens palsy.1 Here, we present a case of classic KD with right abducens palsy that developed during the subacute phase of the illness. A 12-year-old Caucasian girl with an unremarkable medical history was referred to our hospital with a 4-day history of fever, weakness and a rash. On the second day of fever, she developed rash in the lower extremities, vomiting, headache and complete blood count revealed leukocytosis (19 000/mm3). Physical examination revealed a maculopapular rash that blanched with central clearing over her trunk and lower extremities, unilateral lymphadenopathy, bilateral conjunctival injection, oropharynx hyperemia and strawberry tongue. There was no organomegaly, costovertebral tenderness or meningeal findings. Complete blood count and biochemical parameters were non-specific. Diagnosis of KD was made and 2D-echocardiography was performed, which showed a dilated proximal left coronary artery measuring 5.6–5.8 mm. Intravenous immunoglobulin (IVIG) and high-dose acetylsalicylate (80 mg/kg) were given. By her 4th hospital day, 2 days after IVIG therapy, she complained of double vision and lateral rectus palsy. There were no characteristic findings on computed cranial tomography. Lumbar puncture was not abnormal. Her cranial magnetic resonance imaging study was normal. Neurological exam and laboratory findings were consistent with right abducens palsy (Fig. 1). Oral dexamethasone 0.5 mg/kg/dose was started twice a day on her 5th hospital day. On the seventh hospital day the patient was afebrile and her general status was improved, but cranial nerve palsy persisted. Her erythrocyte sedimentation rate was relatively decreased to 40 mm/h. After her 13th hospital day, a dose of steroid treatment was decreased to a half-dose and after another week steroid treatment was ceased. The patient was discharged on the 20th day of hospitalization with acetylsalicylate therapy. A follow-up examination 4 weeks later presented resolution of right abducens palsy and disappearance of coronary involvement. Loss of abduction of the right eye is seen, demonstrating isolated sixth-nerve palsy. The diagnosis of KD can be made when there are fewer than four clinical features if coronary artery involvement is documented.2 Our case had four of the five criteria at the time of presentation (conjunctivitis, mucosa changes, lymphadenopathy and rash). Although the coronary artery involvement of KD is the most important cause of morbidity and mortality, neurological findings can be expected as KD has a multisystemic nature. Irritability and lethargy are common features during the clinical course of KD due to aseptic meningitis.3, 4 Nevertheless, cranial nerve palsy associated with KD has rarely been seen and the mortality rate is higher in KD associated with facial nerve palsy, as compared with KD without facial nerve palsy.1 Although this single case is not enough to be evidence, we suggest that recovery of the neurological finding may be related with acetylsalicylate and steroid treatment. This report reinforces the need to be aware of various manifestations of KD beside coronary artery involvement. It is also helpful to keep in mind the diagnosis of KD in any child with high fever and without cranial palsy.