Pediatric Interventions

Successful Coronary Stenting in a 4-Year-Old Child

John W. Moore, 1* MD, MPH, and Maurice Buchbinder, 2 MD

This report describes a 4-yr-old with critical coronary artery stenosis acquired aftersurgery for congenital heart disease. The patient was treated successfully with coronarystenting after unsuccessful angioplasty. Cathet. Cardiovasc. Diagn. 44:202–205, 1998.r 1998 Wiley-Liss, Inc.

Key words: pediatric coronary stenosis; stent; angioplasty

INTRODUCTION

Among adults, acquired coronary artery stenoses arecommon and usually treated by angioplasty, often withstent placement. Bypass surgery, once first-line therapy, iscurrently reserved for more complex cases. In children,isolated congenital coronary artery stenosis is extremelyrare. Congenital coronary artery disease has also beenassociated with pulmonary atresia and intact ventricularseptum and with William’s syndrome [1]. Pediatriccoronary artery stenosis is most often acquired in Ka-wasaki disease [2], after surgery for congenital heartdisease [3,4], after cardiac transplant [5,6], and in homo-zygous lipid disorders [7,8]. As in adults, children withsignificant coronary stenosis may have angina or isch-emic cardiomyopathy or may die from arrhythmias.

Technical barriers related to the small size of children’scoronary arteries have limited intervention in very youngpatients with coronary artery stenosis. To date, the goldstandard has been surgery to bypass coronary obstruction.Most surgical experience with coronary artery stenosishas been gained among the relatively large number ofpatients with stenoses related to Kawasaki disease. Saphe-nous vein, internal thoracic artery, and gastroepiploicartery grafts have been employed to bypass criticalstenoses in children as young as 12 mo [9]. In patientshaving surgery before 7 yr of age, the largest follow-upstudy demonstrated patency rates of 70% in arterial graftsand of only 27% in vein grafts 5–7 yr after the operation[10]. Thus, the surgical treatment of coronary stenosis inchildren has evolved toward the use of arterial grafts.

Reported experience with transcatheter treatment ofpediatric coronary stenosis is anecdotal. Case reportshave described angioplasties of stenosis related to Ka-wasaki disease [11–13], Takayasu’s arteritis [14], postop-

erative congenital heart disease [15,16], and familialhypercholesterolemia [17]. In addition, angioplasty hasbeen used to correct stenosis of bypass grafts [18,19].This report describes the first successful use of stentplacement with angioplasty to correct coronary stenosisin a young child.

CASE REPORT

The patient presented at the age of 9 mo because of aheart murmur and mild cyanosis. Evaluation revealedtricuspid atresia associated with transposition of the greatarteries, severe subpulmonic stenosis, and a restrictiveventricular septal defect. A common origin of the rightand left coronary arteries in the right coronary sinus wasalso identified. At 13 mo of age, the patient had surgery tocreate hemi-Fontan physiology, enlarge the ventricularseptal defect, and divide the main pulmonary artery.Subsequently, during catheterization, an aneurysm of theleft ventricle (apparently related to ventricular septaldefect enlargement) was identified, and left pulmonaryartery stenosis was corrected with angioplasty and stentplacement. The ventricular aneurysm was repaired at 22mo of age, and follow-up selective coronary angiographyidentified a discrete critical (95%) left main coronaryartery stenosis. At 24 mo of age, a left-internal-thoracic-

1Children’s Heart Institute, Children’s Hospital, San Diego, Cali-fornia2Sharp Memorial Hospital, San Diego, California

*Correspondence to: John W. Moore, M.D., M.P.H., The NemoursCardiac Center, Alfred I. duPont Hospital for Children, Wilmington,DE 19899.

Received 22 September 1997; Revision accepted 19 December 1997

Catheterization and Cardiovascular Diagnosis 44:202–205 (1998)

r 1998 Wiley-Liss, Inc.

artery-to-distal-left coronary bypass operation was per-formed. Thereafter, the patient was well and by clinicalcriteria was a suitable candidate for Fontan completion.Preoperative diagnostic catheterization performed at 48mo of age showed acceptable ventricular function (single-plane left ventricular ejection fraction of 63%, ventricularend diastolic pressure of 6 mmHg), with abnormal wallmotion at the site of the repaired ventricular aneurysm.However, there was persistent severe left coronary arterystenosis, and the bypass artery had become tortuous andsmall (,1 mm in diameter). No collaterals were apparent.The decision to perform angioplasty and, if necessary,stent placement was made to secure the patient’s leftcoronary blood supply, which appeared to be unreliable.

Therapeutic catheterization was performed at 49 mo ofage (weight5 14.0 kg) with general anesthesia. An8-French sheath was placed in the femoral artery, and

heparin (100 units/kg) and cefazolin (25 mg/kg) wereadministered. An 8-French Judkins right coronary guidecatheter engaged the common orifice of the coronaryarteries but selectively intubated the right coronary artery.This catheter was exchanged for an 8 French multipur-pose guide catheter, which engaged the left coronaryartery. A selective left coronary angiogram demonstratedthe stenosis (Fig. 1). Subsequently a 0.014-in. torqueablecoronary guide wire was passed through the guidecatheter, across the stenosis into the distal left coronaryartery. A prepared 3.0-mm coronary angioplasty catheterwas advanced to the lesion and inflated to 8 atm. At thisinflation pressure, a persisting ‘‘waist’’ was apparent atthe site of stenosis (Fig. 2). This catheter was deflated andremoved. A prepared high-pressure 3.25-mm coronaryangioplasty catheter with a Palmaz-Schatz coronary stentmounted was advanced to the lesion, and the proximalarticulation of the stent was placed across the stenosis.

Fig. 1. Left anterior oblique angiogram showing critical leftcoronary artery stenosis (arrow).

Fig. 2. Left anterior oblique view of low-pressure angioplastyballoon with persisting ‘‘waist’’ at peak inflation pressure (ar-row).

Fig. 3. Left anterior oblique view of implanted, fully expandedPalmaz-Schatz coronary stent (arrow).

Fig. 4. Left anterior oblique angiogram showing widely patentleft coronary artery after stent implantation (arrow).

Successful Coronary Stenting in a 4-Year-Old Child 203

The stent was implanted at 20 atm pressure, and the‘‘waist’’ was completely abolished at full inflation pres-sure (Fig. 3). After deflation and removal of the angio-plasty catheter and guide wire, a selective left coronaryangiogram showed excellent relief of the stenosis (Fig.4). The patient recovered uneventfully. There were nocomplications.

The patient was discharged from the hospital onwarfarin and aspirin. She was well and returned forFontan completion 6 mo later. Selective coronary angio-plasty, performed prior to surgery, demonstrated excellentpatency of the left coronary artery, with a mild tubularnarrowing but no restenosis at the stent (Fig. 5). Ventricu-lar function and wall motion were unchanged from priorto angioplasty and stent placement. Fenestrated Fontanwas performed uneventfully, and the patient is well 9 moafter Fontan completion. She takes warfarin because ofthe Fontan fenestration and the coronary stent.

This case demonstrates that coronary stent implanta-tion can be employed safely with angioplasty to correctcoronary stenosis in young children. Excellent patencywas observed after 6 mo and presumably may continuefor the longer term. Given the sophistication and wideavailability of transcatheter treatment of adult coronarydisease, it is appropriate to apply this experience inpediatric settings. As in this case, a team approach withdual operators (pediatric and adult interventional cardiolo-gists) should be undertaken.

Because of the rarity and diversity of suitable lesions,case selection should involve careful planning based on

diagnostic coronary angiography and the specifics of theclinical setting. The significance, location, and morphol-ogy of the lesion and the nature of the disease processshould be considered carefully prior to selecting transcath-eter treatment. Most guidelines for adult angioplasty andstent implantation should apply in selection and planningof pediatric cases [20,21]. Lesions caused by scarring orfatty accumulation are amenable to transcatheter therapy.However, lesions associated with marked intimal prolif-eration, abnormal wall composition, or multiple aneu-rysms should be approached advisedly. In this patient,angioplasty was selected because the lesion was signifi-cant, discrete, and caused by scar formation. Angioplastyof this left main coronary stenosis had acceptable riskbecause a bypass graft provided an alternative bloodsupply to the distal left coronary system. Because angio-plasty at 8 atm failed to eliminate the stenosis (asdemonstrated by the persisting balloon ‘‘waist’’), high-pressure angioplasty was required. The stent was em-ployed to support the arterial wall during the high-pressure inflation. In this case, the stent was indicatedbecause of the suboptimal results of angioplasty.

The small size of pediatric coronary arteries ultimatelylimits the applicability of angioplasty and stents. How-ever, this 14-kg patient allowed use of a standard 8French guide catheter with conventional wires, angio-plasty balloon catheters, and coronary stents. Smallerpatients may be approached with 5 or 6 French guidecatheters for angioplasty and 6 French guide catheterswhen stent placement is also needed. At present, thesmallest coronary balloon angioplasty diameter is 1.5 mmand the smallest available coronary stent diameter is 2.0mm (Cook Cardiology, Bloomington, IN). These factorslimit angioplasty and stent placement to coronary diseasein arteries of normal lumen diameter greater than 1.5 mm.Nevertheless, by employing currently available technol-ogy, most pediatric cases of coronary stenosis may betreated with angioplasty and stents.

The decision to employ angioplasty and stent place-ment should also encompass consideration of potentialearly closure and restenosis, which have been observed inadult patients after stent placement. Smaller diametervessels and stents may be risk factors. Clearly, thelong-term outcome of stenting small vessels in smallchildren is unknown.

Surgical bypass grafts are also limited by the diameterof the graft and the diameter of the normal coronaryartery distal to an obstruction. Both graft and coronaryarteries must have minimum diameters of at least 1.5 mmfor bypass surgery to be feasible. Existing experiencewith surgery is too limited to define long-term outcomes.

We suggest that cases of pediatric coronary obstructionbe evaluated for less invasive transcatheter treatment by

Fig. 5. Six-month follow-up left anterior oblique angiogramshowing mild tubular narrowing of the left coronary artery andno restenosis (arrow).

204 Moore and Buchbinder

using angioplasty and stents before considering bypasssurgery.

REFERENCES

1. Bird LM, Billman GF, Lacro RV, Spicer RL, Jariwala LK, HoymeE, Zamora-Salinas R, Morris C, Viskochil D, Frikke MJ, JonesMC: Sudden death in Williams syndrome: Report of ten cases. JPediatr 129:926–931, 1996.

2. Suzuki A, Kamiya T, Kuwahara N, Ono Y, Kohata T, Takahashi O,Kimura K, Takamiya M: Coronary arterial lesions of Kawasakidisease: Cardiac catheterization findings of 1100 cases. PediatrCardiol 7:3–9, 1986.

3. Tanel RE, Wernovsky G, Landzeberg MJ, Perry SB, Burke RP:Coronary artery abnormalities detected at cardiac catheterizationfollowing the arterial switch operation for transposition of thegreat arteries. Am J Cardiol 76:153–157, 1995.

4. Bonhoeffer P, Gonnet D, Pechaud JF, Stu¨mper O, Aggoun Y,Villain E, Kachaner J, Sidi D: Coronary artery obstruction after thearterial switch operation for transposition of the great arteries innewborns. J Am Coll Cardiol 29:202–206, 1997.

5. Addonizio LJ, Hsu DT, Smith CR, Gersony WM, Rose EA: Latecomplications in pediatric cardiac transplant recipients. Circula-tion, suppl IV 80:II–219, 1989.

6. Pahl E, Zales VR, Fricker FJ, Addonizio LJ: Post transplantcoronary artery disease in children. A multicenter national survey.Circulation 90:II56–II60, 1994.

7. Sprecher DL, Schaefer EJ, Kent KM, Gregg RE, Zech LA, HoegJM, McManus B, Roberts WC, Brewer HB: Cardiovascularfeatures of homozygous familial hypercholesterolemin: Analysisof 16 patients. Am J Cardiol 54:20–30, 1984.

8. Berenson GS, Wattigney WA, Tracy RE, Newman WP, SrinivasanSR, Webber LS, Dalferes ER, Strong JP: Artherosclerosis of theaorta and coronary arteries and cardiovascular risk factors inpersons aged 6 to 30 years and studied at necropsy (the BogalusaHeart Study). Am J Cardiol 70:851–858, 1992.

9. Kitamura S, Kameda Y, Seki T, Kawachi K, Endo M, Takeuchi Y,Kawasaki T, Kawashima Y: Long-term outcome of myocardialrevascularization in patients with Kawasaki coronary artery dis-ease. J Thorac Cardiovasc Surg 107:663–674, 1994.

10. Grabitz RG, Messmer BJ, Seghaye MC, Englehardt W, Muhler E,von Bernuth G: Internal mammary artery bypass graft for impaired

coronary perfusion after neonatal arterial switch operation. Eur JCardiothorac Surg 6:388–390, 1992.

11. Ino T, Nishimoto K, Akimoto K, Park I, Shimazaki S, Yabuta K,Yamaguchi H: Percutaneous transluminal coronary angioplasty forKawasaki disease: a case report and literature review. PediatrCardiol 12:33–35, 1991.

12. Satler LF, Leon MB, Kent KM, Pichard AD, Martin GR: Angio-plasty in a child with Kawasaki disease. Am Heart J 124:216–219,1992.

13. Ino T, Akimoto K, Ohkubo M, Nishimoto K, Yabuta K, Takaya J,Yamaguchi H: Application of percutaneous transluminal coronaryangioplasty to coronary arterial stenosis in Kawasaki disease.Circulation 93:1709–1715, 1996.

14. Lee HY, Rao PS: Percutaneous transluminal coronary angioplastyin Takayasu’s arteritis. Am Heart J 132:1084–1086, 1996.

15. Allada V, Jarmakani JM, Yeatman L: Percutaneous transluminalcoronary angioplasty in an infant with coronary artery stenosisafter arterial switch operation. Am Heart J 122:1464–1465, 1991.

16. Hausdorf G, Kampmann C, Schneider M: Coronary angioplastyfor coronary stenosis after the arterial switch procedure. Am JCardiol 76:621–623, 1995.

17. Mehan VK, Salzmann C, Pfammatter JP, Stocker FP, Meier B: Leftmain coronary angioplasty in a 10-year-old boy with homozygousfamilial hypercholesterolemia. Cathet Cardiovasc Diagn 29:24–27, 1993.

18. Kawata T, Hasegawa J, Yoshida Y, Yoshikawa Y, Kawachi K,Kitmaura S: Percutaneous transluminal coronary angioplasty ofthe left internal thoracic artery graft: A case report in a child.Cathet Cardovasc Diagn 32:340–342, 1994.

19. Carey D, Vincent RN, Brames GP Willimas WH, Douglas JS:Balloon angioplasty of a tissue conduit stenosis after surgicalrepair of anomalous left coronary artery. Cathet Cardiovasc Diagn30:339–340, 1993.

20. Ryan TJ, Bauman WB, Kennedy JW, Kereiakes DJ, King SB,McCallister BD, Smith SC, Ullyot DJ: Guidelines for percutane-ous transluminal coronary angioplasty a report of the AmericanCollege of Cardiology/American Heart Association Task Force onassessment of diagnostic and therapeutic cardiovascular proce-dures (Committeee on Percutaneous Transluminal Coronary angio-plasty). J Am Coll Cardiol 22:2033–2054, 1993.

21. Pepine CJ, Holmes DR, Block PC, Brinker JA, Mark OB, MullinsCE, Nissen SE, Topol EJ, Williams DO: Coronary artery stents. JAm Coll Cardiol 28:782–794, 1996.

Successful Coronary Stenting in a 4-Year-Old Child 205