Int J Clin Pediatr

International Journal of Clinical Pediatrics, ISSN 1927-1255 print, 1927-1263 online, Open Access

Article copyright, the authors; Journal compilation copyright, Int J Clin Pediatr and Elmer Press Inc

Journal website http://www.theijcp.org

Case Report

Volume 8, Number 2, October 2019, pages 45-50

A 2-Year-Old Boy With Refractory Kawasaki Disease Who Developed a Giant Aneurysm After Successful Treatment With Infliximab

Figure 1. Clinical course after admission. Upper panel shows changes in RCA diameter and CRP levels and lower panel shows time course of applied medicine. ASA: aspirin; PSL: prednisolone; IVIG: intravenous immunoglobulin; UTI: ulinastatin; IFX: infliximab; WF: warfarin; RCA: right coronary artery; CRP: C-reactive protein.

Figure 2. (a) Chest X-ray showing the CTR was 50% without cardiomegaly, and (b) transthoracic echocardiogram, showing the RCA was 7.3 mm in diameter and a thrombus was not detected inside the aneurysm. RCA: right coronary artery; Ao: ascending aorta; CTR: cardiothoracic ratio.

Figure 3. (a, b) Transthoracic echocardiogram and (c, d) coronary angiogram. (a) The RCA was 7.3 mm in diameter and a thrombus (4.5 × 15.7 mm) was detected inside the aneurysm (day 10). (b) The size of the RCA was 14.3 × 31.4 mm in diameter without a thrombus (day 61). RCA: right coronary artery; Ao: ascending aorta; RV: right ventricle.

**Table 1.** Laboratory Data
Alb: albumin; ALT: alanine aminotransferase; APTT: activated partial thromboplastin time; AST: aspartate aminotransferase; CK: creatine kinase; Cr: creatinine; CRP: C-reactive protein; FDP: fibrin and fibrinogen degradation product; Hb: hemoglobin; Ht: hematocrit; LDH: lactate dehydrogenase; NT-pro BNP: N-terminal pro brain natriuretic peptide; Plt: platelet; PT: prothrombin time; RBC: red blood cell; TB: total bilirubin; TP: total protein; UN: urea nitrogen; WBC: white blood cell; RSV: respiratory syncytial virus; ASO: antistreptolysin O.
WBC	16.4 × 10³/µL	TB	1.2 mg/dL	RSV	(-)
Neu	88.3%	AST	160 IU/L	Flu (A/B)	(-/-)
Eo	0.1%	ALT	93 IU/L	Mycoplasma	< 40
Baso	0.1%	LDH	429 IU/L	ASO	< 20
Mono	3.5%	CK	35 IU/L	Blood culture	Negative
Lym	8.0%	UN	13 mg/dL
RBC	4.43 × 10⁶/µL	Cr	0.33 mg/dL	Urianalysis
Hb	11.4 g/dL	TP	6.7 g/dL	Protein	(-)
Ht	34.3%	Alb	4.0 g/dL	Blood	(-)
Plt	450 × 10³/µL	CRP	16.0 mg/dL	Sugar	(-)
PT	57%	Na	129 mEq/dL	Cre	171.2 mg/dL
APTT	36.2 s	K	4.6 mEq/dL	N	27 mEq/L
Fib	762 mg/dL	Cl	93 mEq/dL	Cl	15 mEq/L
FDP	14 µg/mL	Ca	9.5 mg/dL	β2-MG	13,520 µg/L
D-dimer	3.6 µg/mL	P	3.2 mg/dL
NT-pro BNP	969 pg/mL	IgG	821 mg/dL
		IgA	117 mg/dL
		IgM	107 mg/dL

Table 1. Laboratory Data

Alb: albumin; ALT: alanine aminotransferase; APTT: activated partial thromboplastin time; AST: aspartate aminotransferase; CK: creatine kinase; Cr: creatinine; CRP: C-reactive protein; FDP: fibrin and fibrinogen degradation product; Hb: hemoglobin; Ht: hematocrit; LDH: lactate dehydrogenase; NT-pro BNP: N-terminal pro brain natriuretic peptide; Plt: platelet; PT: prothrombin time; RBC: red blood cell; TB: total bilirubin; TP: total protein; UN: urea nitrogen; WBC: white blood cell; RSV: respiratory syncytial virus; ASO: antistreptolysin O.

WBC

16.4 × 10³/µL

1.2 mg/dL

RSV

(-)

Neu

88.3%

AST

160 IU/L

Flu (A/B)

(-/-)

0.1%

ALT

93 IU/L

Mycoplasma

< 40

Baso

0.1%

LDH

429 IU/L

ASO

< 20

Mono

3.5%

35 IU/L

Blood culture

Negative

Lym

8.0%

13 mg/dL

RBC

4.43 × 10⁶/µL

0.33 mg/dL

Urianalysis

11.4 g/dL

6.7 g/dL

Protein

(-)

34.3%

Alb

4.0 g/dL

Blood

(-)

Plt

450 × 10³/µL

CRP

16.0 mg/dL

Sugar

(-)

57%

129 mEq/dL

Cre

171.2 mg/dL

APTT

36.2 s

4.6 mEq/dL

27 mEq/L

Fib

762 mg/dL

93 mEq/dL

15 mEq/L

FDP

14 µg/mL

9.5 mg/dL

β2-MG

13,520 µg/L

D-dimer

3.6 µg/mL

3.2 mg/dL

NT-pro BNP

969 pg/mL

IgG

821 mg/dL

IgA

117 mg/dL

IgM

107 mg/dL

**Table 2.** Summary of Previous Reports on IFX Treatment in Kawasaki Disease
Author (reference)	Number of patients	Age (M/F)	Effective rate (%)	Illness day at treatment	Developed CA after IFX (%)	Study design
IFX: infliximab; CA: coronary aneurysm.
Burns et al [3]	16	2.7 (0.1 - 13.1) 11/5	81	19 (8 - 53)	0 (0/5)	Retrospective
Song et al [4]	16	2.8 (0.2 - 5.8) 13/3	69	17 (6 - 29)	0 (0/16)	Retrospective
Mori et al [5]	20	4.6 (1.9 - 10.5) 10/10	90	-	5 (1/20)	Open-label
Sonoda et al [6]	76	3.4 (1 - 10) 51/25	92	8.5 (6 - 11)	12 (9/76)	Retrospective
Masuda et al [7]	434	2.8 (0.1 - 11.5)	73	9	7 (32/434)	Retrospective
Our series	11	1.9 (1.0 - 12.6) 7/4	73	9 (6 - 15)	9 (1/11)	Retrospective

Table 2. Summary of Previous Reports on IFX Treatment in Kawasaki Disease

Author (reference)

Number of patients

Age (M/F)

Effective rate (%)

Illness day at treatment

Developed CA after IFX (%)

Study design

IFX: infliximab; CA: coronary aneurysm.

Burns et al [3]

2.7 (0.1 - 13.1) 11/5

19 (8 - 53)

0 (0/5)

Retrospective

Song et al [4]

2.8 (0.2 - 5.8) 13/3

17 (6 - 29)

0 (0/16)

Retrospective

Mori et al [5]

4.6 (1.9 - 10.5) 10/10

5 (1/20)

Open-label

Sonoda et al [6]

3.4 (1 - 10) 51/25

8.5 (6 - 11)

12 (9/76)

Retrospective

Masuda et al [7]

434

2.8 (0.1 - 11.5)

7 (32/434)

Retrospective

Our series

1.9 (1.0 - 12.6) 7/4

9 (6 - 15)

9 (1/11)

Retrospective