Discussion
Atypical initial presentation in KD is very important, because it can lead to serious complications, especially cardiovascular adverse events. Atypical or incomplete KD diagnosis is challenging. Uncommon manifestations of KD in children have been reported in previous case reports including pyuria, meningitis, ventricular arrhythmia, or shock (9, 10). Introducing such atypical presentations of KD will be beneficial in increasing physicians’ attention in suspicious to KD and prevent delayed diagnosis. In the case, we mentioned here, a seven-year-old girl presented with initial isolated torticollis that subsequently other classic symptoms were added and finally giant aneurysm was diagnosed.
Torticollis in this seemed to be due to painful cervical lymphadenitis (11). The rate of cervical lymphadenopathy occurring as the initial presenting symptoms is only approximately 12% (12). Despite of uncommon manifestation, some cases are reported previously KD presented by torticollis. Dyer et al. reported a 6-year-old male who presented with torticollis. In their case, a series of investigations for elevated inflammatory markers revealed dilated coronary artery aneurysms on echocardiogram and treated with high-dose IVIG and low-dose aspirin (11). Runel-Belliard et al. reported a case of KD with arthritis in a 4-year-old girl whose initial presentation was a febrile torticollis (13). In another case, a nine-year-old boy presented with fever, lymphadenopathy, parotitis, and torticollis with initial diagnosis of infective parotitis and finally KD was diagnosed (14). In our case, she was underwent CABG because of LV dysfunction and after that, she was followed and was healthy until 2 years later.
Dilatation of the coronary arteries can occur in approximately 10-20% of KD patients. In half of them, the aneurysms can regress within 1 or 2 years, and no abnormalities are found by angiography in the coronary-arterial system. In the remaining, in contrast, the aneurysms can persist with obviously irregular lumens of the coronary arteries. In 3% of the patients initially having aneurysms, coronary-arterial obstruction progresses. The time-span between the onset of the disease and development of the coronary-arterial stenosis leading to CABG varies from several months to 20 years. The indications for CABG are to be determined not only by findings derived from angiography, but also on the basis of other clinical factors, such as severity of myocardial ischemia, history of myocardial infarction and ventricular performance (15-19). It is very difficult to determine the indications for catheter intervention and CABG in children, based on the diagnosis and localization of the affected coronary artery, using standard methods (20).