Abstract

An 8-week-old breast-fed female infant had high-grade fever for 2 weeks. Despite multiple oral and intravenous antibiotics, there was no fever defervescence. She developed irritability, refusal to feed and facial deviation towards right side 2 days prior to referral to our hospital. On examination, she was febrile, extremely irritable, tachypneic and had pallor, left sided infranuclear facial nerve palsy (FNP) with hepatosplenomegaly. The ear, ocular and the rest of the systemic examination were normal. Investigations revealed normocytic-normochromic anemia, leucocytosis (25.1 £ 10/L, 70% neutrophils), thrombocytosis (7.86 £ 10/L), hypoalbuminemia (2.8 gm/dl), positive C-reactive protein (249.9 mg/dl), normal urine examination and chest X-ray. Lumbar puncture showed 30 cells (65% polymorphs), glucose of 63 mg/dl, protein of 71 mg/dl. Blood, urine, CSF and stool cultures were sterile. Computerized tomography of head was normal. Child was commenced on broad spectrum intravenous antibiotics with supportive treatment. She developed erythematous maculopapular rash involving the face, trunk, and extremities that was initially considered ‘drug-rash’. Thereafter, the infant developed nonpurulent conjunctivitis. In view of these, a possibility of Kawasaki disease (KD) was entertained. A 2D-Echo showed dilatation of right (8 mm) and left (6 mm) coronary arteries and left anterior descending artery (6 mm) with minimal pericardial eVusion. Treatment with intravenous immunoglobulin (IVIg) (2 g/kg) and aspirin (75 mg/kg per day) was initiated. Within 24 h, there was resolution of fever and irritability. The facial nerve palsy resolved over next 2 days (Fig. 1). FNP is a rare manifestation of KD with less than 35 cases reported so far [1]. The age of reported patients ranges from 3 to 25 months. We report the youngest such infant till date. Atypical KD analogous to our case is more common in young infants and is associated with worse coronary outcomes [2]. The above case emphasizes that possibility of KD must be considered in a patient of any age with unexplained prolonged fever, even with incomplete diagnostic features and unusual manifestations like FNP for timely diagnosis and apt treatment. Appearance of rash should further alert the clinician. FNP in KD shows a female preponderance (1.4:1) with predominant left sided involvement [3, 4]. The inXammatory

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