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| CASE REPORT |
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| Year : 2017 | Volume
: 14
| Issue : 2 | Page : 112-114 |
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Coronary artery aneurism in Kawasaki disease: Case series and literature review
Josephat M Chinawa1, Swati Garekar2, Bhadra Trivedi2
1 Department of Pediatrics, College of Medicine, University of Nigeria, University of Nigeria Teaching Hospital, Ituku, Ozalla, Enugu, Nigeria
2 Department of Pediatric Cardiology, Fortis Hospital Limited, Bhandup (West), Mumbai, Maharashtra, India
| Date of Web Publication | 26-Oct-2017 |
Correspondence Address:
Josephat M Chinawa
Department of Paediatrics, University of Nigeria, University of Nigeria Teaching Hospital, Ituku, Ozalla, Enugu
Nigeria
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/njc.njc_32_16
Kawasaki disease (KD) is associated with coronary artery lesion, especially in the form of aneurysm and ectasia. The first case is 1-year, 9-month-old female; the second is a 13-month-old male, the third is a 14-month-old female, and the fourth is a 5-year, 6-month-old male who all satisfied the criteria of KD. Echocardiography done for all the series showed dilatation of the left anterior descending coronary artery measuring 2.15 mm with Z score of 2.53 and focal dilatation of left main coronary artery with maximum diameter of 2.9 mm with Z score of 2.44 for the first case, prominent left coronary artery measuring 2.19 mm with Z score of 1.28 with borderline dilatation of the circumflex coronary artery measuring 2 mm with a Z score of 2.13 for the second case, and left coronary dilatation measuring 3.5 mm with Z score of 3.61 and borderline left coronary artery dilatation measuring 3.5 mm with a Z score of 2.24, respectively, for the last two cases. We documented four cases of KD as much as we know, and all the four cases presented with coronary artery aneurism with a good response to chemotherapy. Keywords: Child, coronary artery, echocardiogram, Kawasaki
How to cite this article:
Chinawa JM, Garekar S, Trivedi B. Coronary artery aneurism in Kawasaki disease: Case series and literature review. Nig J Cardiol 2017;14:112-4 |
| Introduction | |  |
Kawasaki disease (KD), also called mucocutaneous syndrome of unknown etiology, usually occurs between 6 months and 5 years of age.[1]
The criteria for the diagnosis of KD are satisfied in the presence of fever for at least 5 days and at least four of the following five criteria: polymorphous rash; oral mucous membrane involvement, including red fissured lips, strawberry tongue, or injected pharynx, bilateral nonexudative conjunctivitis; changes with extremities including erythema or edema of palms and soles; or periungal desquamation as a late sign in the subacute phase (~2 or more weeks after illness onset); and cervical lymphadenopathy.[2]
However, incomplete KD should be considered in children who have an unexplained prolonged febrile illness and have not met the diagnostic criteria.[2]
KD is associated with coronary artery lesion with 31% as ectasia and 13% as aneurysm on echocardiogram.[2] KD is the most common cause of coronary artery aneurysm in the pediatric age group in developed countries with a subset of patients at risk for ischemia.[3]
Management of coronary artery aneurysm in KD involves the use of immunoglobulin. The efficacy of intravenous immunoglobulin administered in the acute phase of KD in reducing the occurrence of coronary artery abnormalities is well established.[4]
In Africa, there were few cases of KD reported and none of them had coronary artery aneurysm. For instance, Sotimehin et al. reported a case in Nigeria without any coronary artery involvement, but at autopsy, coronary artery aneurysm was deciphered.[5] Sanin and Ahmed [6] in Sokoto recorded a case of KD without coronary artery affectation. It is interesting to note that in the 1 year of my program in Mumbai, we recorded four cases of KD, of which three had coronary artery aneurysm. This high frequency necessitated the presentation of these case series.
| Case Report | | |
The first case is a 1-year, 9-month-old female; the second is a 13-month-old male, the third is a 14-month-old female, and the fourth is a 5-year, 6-month-old male who all satisfied the criteria of KD. Echocardiography (echo) done for all the series showed dilatation of the left anterior descending coronary artery measuring 2.15 mm with Z score of 2.53 and focal dilatation of left main coronary artery with maximum diameter of 2.9 mm with Z score of 2.44 for the first case, prominent left coronary artery measuring 2.19 mm with Z score of 1.28 with borderline dilatation of the circumflex coronary artery measuring 2 mm with a Z score of 2.13 for the second case, and left coronary dilatation measuring 3.5 mm with Z score of 3.61 and borderline left coronary artery dilatation measuring 3.5 mm with a Z score of 2.24, respectively, for the last two cases.
NB: The case series was commenced on intravenous immunoglobulin.
[Figure 1] shows still echo image of some of the reported series.
| Discussion | | |
We documented four cases of KD within 1 year of the study, and three cases presented with coronary artery aneurysm. They had a good response to chemotherapy. A study in Japan has documented a prevalence of coronary artery aneurysm in KD as 31% on echo.[2] Contrarily, Senzakiet al.[7] noted that though the most common complication of this disease includes coronary artery dilation and aneurysm formation, it occurs in 10–15% of patients during the acute stage. As we noted three cases with KD to have had coronary artery aneurysm, these cases were seen just in 1 year. A longer period of study will make the prevalence of KD clearer.
Although coronary artery aneurysms generally are not seen during the first 10 days of illness, it is stipulated that any patient in whom KD is strongly suspected should have echo since these abnormalities that aid in diagnosis may appear within the first 10 days of fever.[8] In the acute phase of illness, coronary artery abnormalities include lack of tapering, perivascular brightness, and ectasia.[9] These echo findings were all noted in our series.
The common sites of coronary involvement (in ascending other of importance) include the proximal left anterior descending coronary artery, proximal right coronary artery, left main coronary artery, left circumflex branch, distal right coronary artery, and the junction of the right and posterior descending coronary arteries.[10] We reported the involvement of left circumflex and left anterior descending coronary artery in our series.
We noted no valvular involvement in our series. Aortic root dilatation may occur as part of the overall vasculitis, but it is usually mild.[11]
Recent studies showed that children with KD have elevated expression, activity, or protein levels of matrix metalloproteinases (MMPs), which suggested that imbalances in MMPs or MMP/tissue inhibitor of MMP play important pathophysiological roles in the development of coronary artery lesions in this disease.[12] Drugs that will decrease or create antagonistic effect of protein levels of MMPs may help curb the incidence of coronary artery aneurysm in KD.
We commenced intravenous immunoglobulin in our series and they are being followed up. The percentage of patients developing coronary aneurysms is reduced with early administration of intravenous immunoglobulin. Nearly 50% of those coronary artery aneurysms will show angiographic regression within 1–2 years following the illness, with smaller lesions having a greater chance of resolution.[12] We did not do any coronary angiogram in our cases since clinical and echo findings suffice for diagnosis and follow-up.
| Conclusion | | |
We documented four cases of KD as much as we know, and all the four cases presented with coronary artery aneurysm with a good response to chemotherapy.
Acknowledgments
We acknowledge everyone who contributed in the analysis and interpretation of data.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Gordon JB, Kahn AM, Burns JC. When children with Kawasaki disease grow up: Myocardial and vascular complications in adulthood. J Am Coll Cardiol 2009;54:1911-20.  [ PUBMED] |
| 2. | Bratincsak A, Reddy VD, Purohit PJ, Tremoulet AH, Molkara DP, Frazer JR, et al. Coronary artery dilation in acute Kawasaki disease and acute illnesses associated with fever. Pediatr Infect Dis J 2012;31:924-6. [ PUBMED] |
| 3. | Nakamura Y, Yashiro M, Uehara R, Oki I, Kayaba K, Yanagawa H. Increasing incidence of Kawasaki disease in Japan: Nationwide survey. Pediatr Int 2008;50:287-90. [ PUBMED] |
| 4. | Cimaz R, Sundel R. Atypical and incomplete Kawasaki disease. Best Pract Res Clin Rheumatol 2009;23:689-97. [ PUBMED] |
| 5. | Sotimehin SA, Ogunlesi TA, Adekanmbi AF, Fetuga MB, Odumuyiwa EA, Olowu OA. Kawasaki disease in a Nigerian child a case report. Niger Med Pract 2010;57:1 |
| 6. | Sani UM, Ahmed H. Kawasaki disease: An unusual presentation in a 14-year old boy in Sokoto, North Western Nigeria. Niger J Paediatr 2013;40:422-5. |
| 7. | Senzaki H, Chen CH, Ishido H, Masutani S, Matsunaga T, Taketazu M, et al. Arterial hemodynamics in patients after Kawasaki disease. Circulation 2005;111:2119-25. |
| 8. | Newburger JW, Takahashi M, Gerber MA, Gewitz MH, Tani LY, Burns JC, et al. 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. Pediatrics 2004;114:1708-33. |
| 9. | Mueller F, Knirsch W, Harpes P, Prêtre R, Valsangiacomo Buechel E, Kretschmar O. Long-term follow-up of acute changes in coronary artery diameter caused by Kawasaki disease: Risk factors for development of stenotic lesions. Clin Res Cardiol 2009;98:501-7. |
| 10. | Ravekes WJ, Colan SD, Gauvreau K, Baker AL, Sundel RP, van der Velde ME, et al. Aortic root dilation in Kawasaki disease. Am J Cardiol 2001;87:919-22. |
| 11. | Senzaki H. The pathophysiology of coronary artery aneurysms in Kawasaki disease: Role of matrix metalloproteinases. Arch Dis Child 2006;91:847-51. |
| 12. | Newburger J, Takahashi M, Gerber M, Gerwitz M, Tani L. Diagnosis, treatment, and long term management of Kawasaki disease. Circulation 2004;110:2747-2471. |
[Figure 1]
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