Axillary cellulitis as a manifestation of Kawasaki disease

Kawasaki disease (KD) is an acute, self-limiting, systemic vasculitis of childhood. The diagnosis is based on six clinical criteria: fever, bilateral nonexudative conjunctivitis, erythema of the lips and oral mucosa, changes in the extremities, rash, and cervical lymphadenopathy. The inflammatory process involves the coronary arteries and aneurysms or dilatations may lead to ischemic heart disease or sudden death. There is a 25% chance of serious cardiovascular damage if treatment is not given early in the course of the disease.1 However, diagnosis depends on the six clinical signs and no specific laboratory test for definitive diagnosis exists. We report an unusual case of KD whose presenting symptom was axillary cellulitis. We were unable to diagnose KD initially because the patient had few clinical signs of KD. A 4-year-old girl was referred to our hospital with a 3-day history of fever and right axillary pain. On admission, physical examination showed a slight swelling of her right axilla but no redness and she was unable to raise her right arm. Left cervical lymphadenopathy was noted, but other signs of KD were absent. Laboratory findings included a white blood cell count of 14 600 cells per mm3, with 81.0% neutrophils, 11.0% lymphocytes, 7.5% monocytes and 0.5% basophils. Her erythrocyte sedimentation rate was 76 mm/h and the C-reactive protein level (CRP) was 14.4 mg/dL. Antibody tests for cytomegalovirus, Epstein–Barr virus, Coxiella burnetii, and Bartonella henselae were negative, the antistreptolysin-O was under 5 IU/mL, and a pharyngeal culture was negative on her admission. Magnetic resonance imaging (MRI) of the axilla showed widespread inflammatory changes in the subcutaneous tissues that were compatible with a diagnosis of cellulitis, although there was no necrotizing fasciitis or suppurative change in the subcutaneous tissue (Fig. 1). No coronary artery lesions were detected on echocardiography. Magnetic resonance imaging reveals inflammation in right axillary soft tissues. The MRI findings and laboratory data seemed consistent with the diagnosis of bacterial cellulitis, so she was treated with cefotaxime (100 mg/kg/day). Over the next three days, her high fever continued, ranging between 39 and 41°C and the local pain and swelling persisted. The CRP increased to 22.8 mg/dL. Blood and axillary swab (surface and postincision) cultures were negative. Polymerase chain reaction for mycobacterium was negative. On hospital day 3, cefotaxime was changed to meropenem (100 mg/kg/day) and ampicillin (200 mg/kg/day) was added on hospital day 5. On hospital day 3, bilateral nonexudative conjunctivitis was observed. On hospital day 7, her lips became red and a rash gradually appeared on her trunk. At this time, a small pericardial effusion and right coronary arterial dilatation were identified by echocardiogram. KD was considered to be the probable diagnosis because of the lack of positive bacterial cultures and the development of five of the six diagnostic criteria of KD with right coronary arterial dilatation. She was treated with intravenous immunoglobulin (IVIG) (2 g/kg) over 24 h and aspirin (30 mg/kg/day). With treatment, her fever resolved within 72 h. Her axillary pain and swelling disappeared gradually and the high-intensity axillary lesions on MRI improved within 40 days. The CRP also fell rapidly and a repeat echocardiogram showed regression of the coronary lesion. She presented membranous desquamation of the fingers on hospital day 13. Finally, she fulfilled all six diagnostic criteria of KD. Her platelet count increased to 614 000 per mm3 on hospital day 14. She was clinically well and discharged on hospital day 17. On review of the literature, presentation with cellulitis as a complication of KD appears to be rare.2–9 There have been a few previous reports describing presentation with orbital (two cases), pharyngeal (three cases), cervical (one case), axillary (one case), and inguinal cellulitis (one case). The head and neck areas seem to be the favored sites for cellulitis associated with KD, which also appears to occur especially in older children with severe cervical lymphadenopathy.7 From the lack of an infectious etiology and no response to antibiotics, the cellulitis was thought to be immunoreactive not bacterial. Treatment with IVIG and/or a non-steroidal anti-inflammatory agent was effective in all eight patients previously reported. Although presentation with cellulitis in KD cases appears to be rare, it may be the initial symptom as in the present case. We performed an MRI study for KD-associated cellulitis for the first time.2–9 While not useful for distinguishing bacterial cellulitis from KD-associated cellulitis, it helped exclude severe necrotizing fasciitis and suppurative changes in the subcutaneous tissues, which are possible developments in cases of bacterial cellulitis. Diagnosis of KD in the absence of its typical clinical manifestations is difficult and in our patient, a clinical diagnosis of KD could only be made after she fulfilled sufficient diagnostic criteria. In the eight cited patients, it took 1–9 days after the onset of cellulitis to diagnose KD. KD should be included in the differential diagnosis of patients with cellulitis of unknown etiology, especially in the head and neck regions and also the axilla. Careful blood and imaging studies including MRI and additional echocardiograms should be requested for these patients. The prognosis for KD is generally good; however, cardiac lesions may lead to sudden death. Early diagnosis and treatment are important for preventing the serious complications of KD. We thank Dr Peter M. Olley (Emeritus Professor, University of Alberta) for language advice.

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