Facial palsy in a 2-month-old infant with Kawasaki disease
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Rheumatol Int (2010) 30:1407–1408 DOI 10.1007/s00296-009-1174-2 123 LETTER TO THE EDITOR Facial palsy in a 2-month-old infant with Kawasaki disease Satvinder Kaur · Ketan Prasad Kulkarni · Prem Narayan Dubey Received: 7 August 2009 / Accepted: 20 September 2009 / Published online: 11 November 2009 © Springer-Verlag 2009 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 devel- oped irritability, refusal to feed and facial deviation towards right side 2 days prior to referral to our hospital. On exami- nation, 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 9 /L, 70% neutrophils), thrombocytosis (7.86 £ 10 9 /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% poly- morphs), 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 immuno- globulin (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 S. Kaur · K. P. Kulkarni · P. N. Dubey (&) Apollo Centre for Advanced Pediatrics, Indraprastha Apollo Hospital, New Delhi 110076, India e-mail: [email protected] S. Kaur e-mail: [email protected] Fig. 1 2-month-old infant with left facial nerve palsy
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Transcript of Facial palsy in a 2-month-old infant with Kawasaki disease
Rheumatol Int (2010) 30:1407–1408
DOI 10.1007/s00296-009-1174-2LETTER TO THE EDITOR
Facial palsy in a 2-month-old infant with Kawasaki disease
Satvinder Kaur · Ketan Prasad Kulkarni · Prem Narayan Dubey
Received: 7 August 2009 / Accepted: 20 September 2009 / Published online: 11 November 2009© Springer-Verlag 2009
An 8-week-old breast-fed female infant had high-gradefever for 2 weeks. Despite multiple oral and intravenousantibiotics, there was no fever defervescence. She devel-oped irritability, refusal to feed and facial deviation towardsright side 2 days prior to referral to our hospital. On exami-nation, she was febrile, extremely irritable, tachypneic andhad pallor, left sided infranuclear facial nerve palsy (FNP)with hepatosplenomegaly. The ear, ocular and the rest ofthe systemic examination were normal. Investigationsrevealed normocytic-normochromic anemia, leucocytosis(25.1 £ 109/L, 70% neutrophils), thrombocytosis (7.86 £109/L), hypoalbuminemia (2.8 gm/dl), positive C-reactiveprotein (249.9 mg/dl), normal urine examination and chestX-ray. Lumbar puncture showed 30 cells (65% poly-morphs), glucose of 63 mg/dl, protein of 71 mg/dl. Blood,urine, CSF and stool cultures were sterile. Computerizedtomography of head was normal. Child was commenced onbroad spectrum intravenous antibiotics with supportivetreatment. She developed erythematous maculopapular rashinvolving the face, trunk, and extremities that was initiallyconsidered ‘drug-rash’. Thereafter, the infant developednonpurulent conjunctivitis. In view of these, a possibility ofKawasaki disease (KD) was entertained. A 2D-Echo showeddilatation of right (8 mm) and left (6 mm) coronary arteriesand left anterior descending artery (6 mm) with minimalpericardial eVusion. Treatment with intravenous immuno-globulin (IVIg) (2 g/kg) and aspirin (75 mg/kg per day)
was initiated. Within 24 h, there was resolution of feverand irritability. The facial nerve palsy resolved over next2 days (Fig. 1).
FNP is a rare manifestation of KD with less than 35cases reported so far [1]. The age of reported patientsranges from 3 to 25 months. We report the youngest suchinfant till date. Atypical KD analogous to our case is morecommon in young infants and is associated with worsecoronary outcomes [2]. The above case emphasizes thatpossibility of KD must be considered in a patient of any agewith unexplained prolonged fever, even with incompletediagnostic features and unusual manifestations like FNP fortimely diagnosis and apt treatment. Appearance of rashshould further alert the clinician.
FNP in KD shows a female preponderance (1.4:1) withpredominant left sided involvement [3, 4]. The inXammatory
S. Kaur · K. P. Kulkarni · P. N. Dubey (&)Apollo Centre for Advanced Pediatrics, Indraprastha Apollo Hospital, New Delhi 110076, Indiae-mail: [email protected]
S. Kaure-mail: [email protected] Fig. 1 2-month-old infant with left facial nerve palsy
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vasculitis of the arteries supplying the facial nerve andimmunologic mechanisms are implicated [3, 4]. AlthoughFNP resolves in all patients surviving the acute phase ofKD, treatment with IVIG seems to shorten the time to fullrecovery. Coronary abnormalities are common (>50%) inKD with FNP.
In conclusion, unexplained FNP in young children witha prolonged febrile illness should prompt consideration ofechocardiography to exclude KD.
ConXict of interest statement None.
References
1. Wright H, Waddington C, Geddes J, Newburger JW, Burgner D(2008) Facial nerve palsy complicating Kawasaki disease. Pediat-rics 122:783–785
2. Sundel RP, Petty RE (2005) Kawasaki disease. In: Cassidy JT, PettyRE, Laxer RM, Lindsley CB (eds) Textbook of pediatric rheumatol-ogy, 5th edn. Elsevier Saunders, Philadelphia, pp 521–538
3. Bushara K, Wilson A, Rust RS (1997) Facial palsy in Kawasakisyndrome. Pediatr Neurol 17:362–364
4. Poon LK, Lun KS, Ng YM (2000) Facial nerve palsy and Kawasakidisease. Hong Kong Med J 6:224–226
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