Int J Clin Exp Pathol 2015;8(8):9468-9470 www.ijcep.com /ISSN:1936-2625/IJCEP0007286
Original Article Giant right coronary artery aneurysm secondary to Kawasaki disease in child: a case report Shanshan Zhang1, Geli Liu1, Tielian Yu2, Guiming Zhou3, Rongxiu Zheng1 Department of Pediatrics, General Hospital, Tianjin Medical University, Tianjin 300052, China; 2Department of Ultrasound, General Hospital, Tianjin Medical University, Tianjin 300052, China; 3Department of Medical Imaging, General Hospital, Tianjin Medical University, Tianjin 300052, China 1
Received February 24, 2015; Accepted April 14, 2015; Epub August 1, 2015; Published August 15, 2015 Abstract: Coronary artery aneurysm or ectasia was reported in approximately 15% to 25% of the affected children, particularly in the proximal end of the main blood vessel and the left anterior descending part. Rare patients have been reported with aneurysm in the distal end of the right coronary artery. In this case report, we present a rare case with aneurysm in the distal end of the right coronary artery. Multi-slice computed tomography was performed for the coronary angiography. Aspirin (10 mg/kg body weight per day) and gamma globulin (2 kg/kg body weight) was administrated via intravenous injection. The patient is currently in a healthy status with a 12-month follow up. Keywords: Giant right coronary artery aneurysm, Kawasaki disease, child, ultrasonic echocardiogram
Introduction Kawasaki disease refers to an acute systemic inflammatory disorder frequently reported in young children [1, 2]. Coronary artery aneurysms or ectasia was noticed in approximately 15% to 25% of the children with Kawasaki disease, especially in the proximal end of the main blood vessel and the left anterior descending part [3]. In this report, we present a rare patient with Kawasaki disease combined with aneurysm in the distal end of the right coronary artery. Case report A 7-year-old male patient was admitted to our hospital due to fever lasting for 6 days combined with conjunctival hyperemia for 1 day and strawberry tongue. To decrease the body temperature, antibiotics (penicillin and Cephalosporin) was administrated in another hospital previously, however, the body temperature was still in a range of 38-40°C. No abnormality was identified in the laboratory test. Ultrasonic echocardiogram indicated the internal diameter of left coronary artery main stem was 3.1 mm, while that of the right coronary
artery main stem was 3.4 mm. The boy was diagnosed with Kawasaki disease. For the treatment, aspirin (10 mg/kg body weight per day) and gamma globulin (2 g/kg body weight) was administrated via intravenous injection. On the fourth day, the body temperature was reduced to a normal range, the conjunctiva hyperemia was eliminated, and desquamation was noticed at the distal end of the fingers and the balanus. To monitor the progress of the disease, ultrasonic echocardiogram was performed on day 6, which revealed the internal diameters of left and right coronary artery main stem were 3.1 mm and 4.7 mm (Figure 1), respectively. Tumor-like lesion was noticed in local sites, with the maximal width of 5.5 mm. Subsequently, coronary angiography was performed using multi-slice computed tomography followed by value rendering and curved planar reconstruction, which revealed the uneven width in the lumen of the right coronary artery. Meanwhile, uneven aneurysm-like lesion was noticed in local part with a maximal diameter of 6.4 mm (Figure 2). Further, tumorlike lesion was noticed at the distal end. The patient is currently in a healthy status with a 12-month follow up.
Giant right coronary artery aneurysm secondary to Kawasaki disease in child ed tomography with an internal diameter of 6.4 mm.
Figure 1. Ectasia of right coronary artery was revealed together with enhanced echo of the tunica intima. RCA, right coronary artery; AO, aorta.
Ultrasonic echocardiogram has been acknowledged as the basic procedure for the imaging of coronary artery in patients with Kawasaki disease [7]. Nevertheless, its accuracy in the identification of obstructive lesion and the imaging of the distal end of the coronary artery is still limited. Currently, coronary CT angiography is considered as the golden standard for the diagnosis of Kawasaki disease [8], but its disadvantages such as invasiveness and high cost have posed great obstacles for its further application in clinical practice. In our study, multi-slice computed tomography was used in the evaluation of coronary artery lesions in patients with Kawasaki disease. Compared with conventional coronary angiography, it showed various advantages such as noninvasive, less radiation damage and low cost [9].
In adults, besides atherosclerotic causes, Kawasaki disFigure 2. Curved planar reconstruction images of the right coronary artery ease could lead to coronary indicated uneven width of the right coronary artery, together with local aneuartery aneurysm [10, 11]. In rysm-like lesion in local parts. The maximal diameter was 6.4 mm. Aneurysm 2006, Manghat et al reported was noticed at the distal end of the coronary artery. an adult with Kawasaki disease, and the multi-detector row CT coronary angiography indicated giant Discussion coronary artery aneurysm in the left anterior Few cases with giant right coronary artery descending artery [12]. Nevertheless, no child aneurysm have been reported in adults [4]. For with Kawasaki disease concurrent with giant most patients with Kawasaki disease, the right coronary artery aneurysm has been major parts affected by coronary artery disease reported previously after literature research in were proximal end of the left anterior descendthe PubMed, Medline, and Embase database. ing branch, proximal end of the right coronary Currently, the treatment of Kawasaki disease is artery, left coronary artery main stem, distal mainly depended on the intravenous gamma end of the right coronary artery and the adjaglobulin and high-dose aspirin to halt inflammacent area of the right coronary artery to the tion and to reduce the likelihood of developing posterior descending branch [5, 6]. In this case, coronary disorders [13]. In most patients, the patient was identified with giant right cororegression of coronary ectasia or aneurysm nary artery aneurysm using multi-slice comput-
9469
Int J Clin Exp Pathol 2015;8(8):9468-9470
Giant right coronary artery aneurysm secondary to Kawasaki disease in child was noted about 12 months to 24 months. However, in less than 2% of the patients, systemic aneurysm may occur. In this case, the patient received intravenous injection of gamma globulin and aspirin, and he is in a healthy status during the 12-month follow-up. Coronary artery aneurysms or ectasia was commonly noticed in children with Kawasaki disease, especially in the proximal end of the main blood vessel and the left anterior descending part. We present a rare case with Kawasaki disease combined with aneurysm in the distal end of the right coronary artery. After treatment, a satisfactory outcome was obtained during the 12-month follow-up. Disclosure of conflict of interest
[4]
[5]
[6]
[7]
[8]
None. Address correspondence to: Drs. Rongxiu Zheng and Geli Liu, Department of Pediatrics, General Hospital, Tianjin Medical University, Tianjin 300052, China. Tel: +86-22-60361673; E-mail: rzheng2015@ 126.com (RXZ); [email protected] (GLL)
[9]
References [1]
[2]
[3]
Adsuar-Gomez A, Gonzalez-Calle A and Coserria-Sanchez JF. Giant coronary aneurysms in Kawasaki disease. Rev Esp Cardiol (Engl Ed) 2014; 67: 489. Bratincsak A, Reddy VD, Purohit PJ, Tremoulet AH, Molkara DP, Frazer JR, Dyar D, Bush RA, Sim JY, Sang N, Burns JC and Melish MA. Coronary artery dilation in acute Kawasaki disease and acute illnesses associated with Fever. Pediatr Infect Dis J 2012; 31: 924-926. Newburger JW, Takahashi M, Gerber MA, Gewitz MH, Tani LY, Burns JC, Shulman ST, Bolger AF, Ferrieri P, Baltimore RS, Wilson WR, Baddour LM, Levison ME, Pallasch TJ, Falace DA and Taubert KA. Committee on Rheumatic Fever, Endocarditis and Kawasaki Disease; Council on Cardiovascular Disease in theYoung; American Heart Association; American Academy of Pediatrics. Diagnosis, treatment, and long-term management of Kawasaki disease: a statement for health professionals from the Committee on Rheumatic Fever, Endocarditis and Kawasaki Disease, Council on Cardiovascular Disease in the Young, American Heart Association. Circulation 2004; 110: 2747-2771.
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[10]
[11] [12]
[13]
Jha NK, Ouda HZ, Khan JA, Eising GP and Augustin N. Giant right coronary artery aneurysm- case report and literature review. J Cardiothorac Surg 2009; 4: 18. Xu QQ, Ding YY, Lv HT, Zhou WP, Sun L, Huang J and Yan WH. Evaluation of Left Ventricular Systolic Strain in Children With Kawasaki Disease. Pediatr Cardiol 2014; 35: 1191-7. Miyamoto T, Ikeda K, Ishii Y and Kobayashi T. Rupture of a coronary artery aneurysm in Kawasaki disease: A rare case and review of the literature for the past 15 years. J Thorac Cardiovasc Surg 2014; 147: e67-69. Kanamaru H, Sato Y, Takayama T, Ayusawa M, Karasawa K, Sumitomo N and Harada K. Assessment of coronary artery abnormalities by multislice spiral computed tomography in adolescents and young adults with Kawasaki disease. Am J Cardiol 2005; 95: 522-525. Carbone I, Cannata D, Algeri E, Galea N, Napoli A, De Zorzi A, Bosco G, D’Agostino R, Menezes L, Catalano C, Passariello R and Francone M. Adolescent Kawasaki disease: usefulness of 64-slice CT coronary angiography for follow-up investigation. Pediatr Radiol 2011; 41: 11651173. Wu MT, Hsieh KS, Lin CC, Yang CF and Pan HB. Images in cardiovascular medicine. Evaluation of coronary artery aneurysms in Kawasaki disease by multislice computed tomographic coronary angiography. Circulation 2004; 110: e339. Nichols L, Lagana S and Parwani A. Coronary artery aneurysm: a review and hypothesis regarding etiology. Arch Pathol Lab Med 2008; 132: 823-828. Syed M and Lesch M. Coronary artery aneurysm: a review. Prog Cardiovasc Dis 1997; 40: 77-84 Manghat NE, Morgan-Hughes GJ, Cox ID and Roobottom CA. Giant coronary artery aneurysm secondary to Kawasaki disease: diagnosis in an adult by multi-detector row CT coronary angiography. Br J Radiol 2006; 79: e133136. Dimitriades VR, Brown AG and Gedalia A. Kawasaki disease: pathophysiology, clinical manifestations, and management. Curr Rheumatol Rep 2014; 16: 423.
Int J Clin Exp Pathol 2015;8(8):9468-9470
Original Article Giant right coronary artery aneurysm secondary to Kawasaki disease in child: a case report Shanshan Zhang1, Geli Liu1, Tielian Yu2, Guiming Zhou3, Rongxiu Zheng1 Department of Pediatrics, General Hospital, Tianjin Medical University, Tianjin 300052, China; 2Department of Ultrasound, General Hospital, Tianjin Medical University, Tianjin 300052, China; 3Department of Medical Imaging, General Hospital, Tianjin Medical University, Tianjin 300052, China 1
Received February 24, 2015; Accepted April 14, 2015; Epub August 1, 2015; Published August 15, 2015 Abstract: Coronary artery aneurysm or ectasia was reported in approximately 15% to 25% of the affected children, particularly in the proximal end of the main blood vessel and the left anterior descending part. Rare patients have been reported with aneurysm in the distal end of the right coronary artery. In this case report, we present a rare case with aneurysm in the distal end of the right coronary artery. Multi-slice computed tomography was performed for the coronary angiography. Aspirin (10 mg/kg body weight per day) and gamma globulin (2 kg/kg body weight) was administrated via intravenous injection. The patient is currently in a healthy status with a 12-month follow up. Keywords: Giant right coronary artery aneurysm, Kawasaki disease, child, ultrasonic echocardiogram
Introduction Kawasaki disease refers to an acute systemic inflammatory disorder frequently reported in young children [1, 2]. Coronary artery aneurysms or ectasia was noticed in approximately 15% to 25% of the children with Kawasaki disease, especially in the proximal end of the main blood vessel and the left anterior descending part [3]. In this report, we present a rare patient with Kawasaki disease combined with aneurysm in the distal end of the right coronary artery. Case report A 7-year-old male patient was admitted to our hospital due to fever lasting for 6 days combined with conjunctival hyperemia for 1 day and strawberry tongue. To decrease the body temperature, antibiotics (penicillin and Cephalosporin) was administrated in another hospital previously, however, the body temperature was still in a range of 38-40°C. No abnormality was identified in the laboratory test. Ultrasonic echocardiogram indicated the internal diameter of left coronary artery main stem was 3.1 mm, while that of the right coronary
artery main stem was 3.4 mm. The boy was diagnosed with Kawasaki disease. For the treatment, aspirin (10 mg/kg body weight per day) and gamma globulin (2 g/kg body weight) was administrated via intravenous injection. On the fourth day, the body temperature was reduced to a normal range, the conjunctiva hyperemia was eliminated, and desquamation was noticed at the distal end of the fingers and the balanus. To monitor the progress of the disease, ultrasonic echocardiogram was performed on day 6, which revealed the internal diameters of left and right coronary artery main stem were 3.1 mm and 4.7 mm (Figure 1), respectively. Tumor-like lesion was noticed in local sites, with the maximal width of 5.5 mm. Subsequently, coronary angiography was performed using multi-slice computed tomography followed by value rendering and curved planar reconstruction, which revealed the uneven width in the lumen of the right coronary artery. Meanwhile, uneven aneurysm-like lesion was noticed in local part with a maximal diameter of 6.4 mm (Figure 2). Further, tumorlike lesion was noticed at the distal end. The patient is currently in a healthy status with a 12-month follow up.
Giant right coronary artery aneurysm secondary to Kawasaki disease in child ed tomography with an internal diameter of 6.4 mm.
Figure 1. Ectasia of right coronary artery was revealed together with enhanced echo of the tunica intima. RCA, right coronary artery; AO, aorta.
Ultrasonic echocardiogram has been acknowledged as the basic procedure for the imaging of coronary artery in patients with Kawasaki disease [7]. Nevertheless, its accuracy in the identification of obstructive lesion and the imaging of the distal end of the coronary artery is still limited. Currently, coronary CT angiography is considered as the golden standard for the diagnosis of Kawasaki disease [8], but its disadvantages such as invasiveness and high cost have posed great obstacles for its further application in clinical practice. In our study, multi-slice computed tomography was used in the evaluation of coronary artery lesions in patients with Kawasaki disease. Compared with conventional coronary angiography, it showed various advantages such as noninvasive, less radiation damage and low cost [9].
In adults, besides atherosclerotic causes, Kawasaki disFigure 2. Curved planar reconstruction images of the right coronary artery ease could lead to coronary indicated uneven width of the right coronary artery, together with local aneuartery aneurysm [10, 11]. In rysm-like lesion in local parts. The maximal diameter was 6.4 mm. Aneurysm 2006, Manghat et al reported was noticed at the distal end of the coronary artery. an adult with Kawasaki disease, and the multi-detector row CT coronary angiography indicated giant Discussion coronary artery aneurysm in the left anterior Few cases with giant right coronary artery descending artery [12]. Nevertheless, no child aneurysm have been reported in adults [4]. For with Kawasaki disease concurrent with giant most patients with Kawasaki disease, the right coronary artery aneurysm has been major parts affected by coronary artery disease reported previously after literature research in were proximal end of the left anterior descendthe PubMed, Medline, and Embase database. ing branch, proximal end of the right coronary Currently, the treatment of Kawasaki disease is artery, left coronary artery main stem, distal mainly depended on the intravenous gamma end of the right coronary artery and the adjaglobulin and high-dose aspirin to halt inflammacent area of the right coronary artery to the tion and to reduce the likelihood of developing posterior descending branch [5, 6]. In this case, coronary disorders [13]. In most patients, the patient was identified with giant right cororegression of coronary ectasia or aneurysm nary artery aneurysm using multi-slice comput-
9469
Int J Clin Exp Pathol 2015;8(8):9468-9470
Giant right coronary artery aneurysm secondary to Kawasaki disease in child was noted about 12 months to 24 months. However, in less than 2% of the patients, systemic aneurysm may occur. In this case, the patient received intravenous injection of gamma globulin and aspirin, and he is in a healthy status during the 12-month follow-up. Coronary artery aneurysms or ectasia was commonly noticed in children with Kawasaki disease, especially in the proximal end of the main blood vessel and the left anterior descending part. We present a rare case with Kawasaki disease combined with aneurysm in the distal end of the right coronary artery. After treatment, a satisfactory outcome was obtained during the 12-month follow-up. Disclosure of conflict of interest
[4]
[5]
[6]
[7]
[8]
None. Address correspondence to: Drs. Rongxiu Zheng and Geli Liu, Department of Pediatrics, General Hospital, Tianjin Medical University, Tianjin 300052, China. Tel: +86-22-60361673; E-mail: rzheng2015@ 126.com (RXZ); [email protected] (GLL)
[9]
References [1]
[2]
[3]
Adsuar-Gomez A, Gonzalez-Calle A and Coserria-Sanchez JF. Giant coronary aneurysms in Kawasaki disease. Rev Esp Cardiol (Engl Ed) 2014; 67: 489. Bratincsak A, Reddy VD, Purohit PJ, Tremoulet AH, Molkara DP, Frazer JR, Dyar D, Bush RA, Sim JY, Sang N, Burns JC and Melish MA. Coronary artery dilation in acute Kawasaki disease and acute illnesses associated with Fever. Pediatr Infect Dis J 2012; 31: 924-926. Newburger JW, Takahashi M, Gerber MA, Gewitz MH, Tani LY, Burns JC, Shulman ST, Bolger AF, Ferrieri P, Baltimore RS, Wilson WR, Baddour LM, Levison ME, Pallasch TJ, Falace DA and Taubert KA. Committee on Rheumatic Fever, Endocarditis and Kawasaki Disease; Council on Cardiovascular Disease in theYoung; American Heart Association; American Academy of Pediatrics. Diagnosis, treatment, and long-term management of Kawasaki disease: a statement for health professionals from the Committee on Rheumatic Fever, Endocarditis and Kawasaki Disease, Council on Cardiovascular Disease in the Young, American Heart Association. Circulation 2004; 110: 2747-2771.
9470
[10]
[11] [12]
[13]
Jha NK, Ouda HZ, Khan JA, Eising GP and Augustin N. Giant right coronary artery aneurysm- case report and literature review. J Cardiothorac Surg 2009; 4: 18. Xu QQ, Ding YY, Lv HT, Zhou WP, Sun L, Huang J and Yan WH. Evaluation of Left Ventricular Systolic Strain in Children With Kawasaki Disease. Pediatr Cardiol 2014; 35: 1191-7. Miyamoto T, Ikeda K, Ishii Y and Kobayashi T. Rupture of a coronary artery aneurysm in Kawasaki disease: A rare case and review of the literature for the past 15 years. J Thorac Cardiovasc Surg 2014; 147: e67-69. Kanamaru H, Sato Y, Takayama T, Ayusawa M, Karasawa K, Sumitomo N and Harada K. Assessment of coronary artery abnormalities by multislice spiral computed tomography in adolescents and young adults with Kawasaki disease. Am J Cardiol 2005; 95: 522-525. Carbone I, Cannata D, Algeri E, Galea N, Napoli A, De Zorzi A, Bosco G, D’Agostino R, Menezes L, Catalano C, Passariello R and Francone M. Adolescent Kawasaki disease: usefulness of 64-slice CT coronary angiography for follow-up investigation. Pediatr Radiol 2011; 41: 11651173. Wu MT, Hsieh KS, Lin CC, Yang CF and Pan HB. Images in cardiovascular medicine. Evaluation of coronary artery aneurysms in Kawasaki disease by multislice computed tomographic coronary angiography. Circulation 2004; 110: e339. Nichols L, Lagana S and Parwani A. Coronary artery aneurysm: a review and hypothesis regarding etiology. Arch Pathol Lab Med 2008; 132: 823-828. Syed M and Lesch M. Coronary artery aneurysm: a review. Prog Cardiovasc Dis 1997; 40: 77-84 Manghat NE, Morgan-Hughes GJ, Cox ID and Roobottom CA. Giant coronary artery aneurysm secondary to Kawasaki disease: diagnosis in an adult by multi-detector row CT coronary angiography. Br J Radiol 2006; 79: e133136. Dimitriades VR, Brown AG and Gedalia A. Kawasaki disease: pathophysiology, clinical manifestations, and management. Curr Rheumatol Rep 2014; 16: 423.
Int J Clin Exp Pathol 2015;8(8):9468-9470
