(Circulation. 1999;99:2621-2625.)
© 1999 American Heart Association, Inc.
Brief Rapid Communications |
Fate of the Stented Arterial Duct
From the Departments of Paediatric Cardiology, Yorkshire Heart Centre (J.L.G., O.U., M.E.C.B., K.G.W.), Leeds, and Freeman Hospital (C.W., J.R.L.H.), Newcastle on Tyne, UK.
Correspondence to Dr John Gibbs, Department of Paediatric Cardiology, Yorkshire Heart Centre, Leeds General Infirmary, West Yorkshire LS1 3EX, UK.
Abstract
Background—The technical aspects of ductal stenting have been reported, but little is known of the fate of the duct after stent implantation.
Methods and Results—Nineteen patients underwent stent implantation to maintain ductal patency. Eight had hypoplastic left heart (HLH) syndrome, 10 had pulmonary atresia, and 1 had tricuspid atresia. Median survival with HLH was 57 (12 to 907) days. Stent implantation was successful in all cases of HLH, but there were no long-term survivors. Two well-palliated infants died at transplantation. Median survival with duct-dependant pulmonary flow was 183 (0 to 1687) days, with 3 patients well at latest follow-up (56, 55, and 9 months, respectively). There were 2 operative deaths due to ductal spasm and 4 late deaths, 1 due to duct thrombosis, 1 due to chronic lung disease, and 2 of unknown cause. Stent implantation failed in 4 of the 11 cases. Assessment of endothelialization was possible in 13 cases; the stent was partially covered in 3 and fully endothelialized in all 10 cases assessed >8 weeks after implantation. In patients stented for inadequate pulmonary flow, ductal intimal hyperplasia occurred by 9 months in all 3 survivors but responded to repeated dilation.
Conclusions—Ductal stenting cannot be recommended. In patients with HLH, it provides only short-term palliation even when combined with pulmonary artery banding. With duct-dependent pulmonary blood flow, the procedure carries high risk, and duration of palliation is poor. In patients with bilateral ducts and absent central pulmonary arteries, good palliation may be achieved, but repeated angioplasty is necessary to counteract intimal hyperplasia.
Key Words: stents • arteries • pulmonary heart disease • pediatrics
Animal studies have suggested that ductal patency may be maintained by stent implantation,1 2 but only preliminary human studies with short-term results have been reported.3 4 5 This report describes our experience with ductal stenting over 6 years, with maximum patient follow-up of 53 months.
Methods
Nineteen infants aged 4 to 78 days, with weight ranging from 2 to 4.1 kg, were included. Eight had hypoplastic left heart syndrome (HLH), 10 had pulmonary atresia, and 1 had tricuspid atresia with a restrictive ventricular septal defect. Johnson & Johnson stainless steel stents were used in all cases. The technical details of stent implantation have been described previously.3 4 Ethics committee approval was obtained for the studies.
Results
Duct-Dependent Systemic Blood Flow
In all 8 patients with HLH, stent implantation was successful.
Seven also had pulmonary artery banding and atrial septostomy.
There was no procedure-related death in this group. Survival ranged
from 12 days to 30 months (median, 51 days). Two patients survived in
good health until transplantation at age 2 and 6 months, respectively,
but both died during surgery. The single patient who did not have
pulmonary artery banding died during surgical reconstruction of
the aortic arch at age 12 days. Four patients aged between 2 and 10
weeks died of right ventricular failure due to excessive
pulmonary blood flow despite bilateral pulmonary artery
banding. One died at the age of 30 months of abrupt right
ventricular failure despite redilation of the stent and
effective banding.
Assessment of duct morphology was possible at surgery or autopsy in 7
of the 8 patients. All the stents appeared widely patent, but in 1 case
there was a minor degree of constriction at the aortic end of the duct,
the end of the stent being 
2 mm proximal to the junction of the
duct and aorta. As early as 2 weeks, there was partial
endothelialization (Figure 1
). All stents examined >3 months
after implantation were fully endothelialized. In 1
patient, the duct became mildly restrictive (withdrawal gradient of
20 mm Hg) owing to growth and intimal hyperplasia. This responded
to repeat dilation at the age of 8 months.
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Duct-Dependent Pulmonary Blood Flow
Stent implantation was successful in 7 of the 11 patients with
duct-dependent pulmonary blood flow. In 2 patients, death
occurred during catheterization owing to duct spasm,
and in 2 others, implantation failed because of duct tortuosity.
Duct patency was maintained after a single procedure in 3 patients, but 2 procedures were required in 1 case, 3 procedures in 2, and 5 procedures in 1. Of the 9 ducts stented in 7 patients, a single stent was required in 5, 2 stents were required in 1, 3 stents in 3 ducts, and 4 (with some overlap) in 1.
That the duct was not initially fully stented only became apparent between 4 and 10 days after prostaglandin E was stopped. Constriction occurred at the aortic end in 1 case and at the pulmonary artery end in the other 4 cases. Accurate stent positioning was difficult because of high flow in the duct and anatomic distortion caused by the guidewire and catheter.
Of the 7 early survivors, 2 died suddenly in their cribs at the age of 6 and 11 weeks, respectively. No clear cause was found, and both had patent ducts at autopsy (1 had situs ambiguous and the other an extensive cleft palate). One child died suddenly at the age of 11 months; autopsy showed acute thrombosis of the duct with some constriction at the pulmonary artery, the distal end of the stent stopping 2 mm from the distal end of the duct. One patient with bronchopulmonary dysplasia became increasingly cyanosed at the age of 4 months; a bridged stent had been used, and there was mild stenosis adjacent to the bridge. We were unable to cross the stent with a balloon, so the patient had a modified Blalock Taussig shunt but died postoperatively owing to chronic lung disease despite a patent duct and patent shunt.
Three patients remain alive and well. Two have absent central
pulmonary arteries with bilateral ducts and have needed 3 and 1
elective repeat catheterizations, respectively, to
maintain adequate saturations (around 80%) and adequate
pulmonary artery growth (Figure 2) at their current respective ages of 5
years and 16 months. One patient with tricuspid atresia underwent 2
repeat dilations (1 during treatment for endocarditis), remaining well
at the age of 5, when she had a successful bidirectional Glenn shunt
implanted (the duct was left alone at operation).
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Increasing cyanosis in the 3 long-term survivors appeared to be due in
part to the patients' growth, but angiography showed
endothelial proliferation to be a factor in all (Figure 3). In the 4 patients who died, autopsy
showed full endothelialization of the stent, with a
thin, complete layer of endothelium formed as early as
6 weeks (Figure 4). Microscopic
thickness of the neoendothelium varied from 0.04 to
0.09 mm over the mesh itself and from 0.15 to 0.28 mm between
the mesh.
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Discussion
Duct-Dependent Systemic Blood Flow
The high mortality rate associated with the Norwood operation
after its introduction in the United Kingdom prompted a search for
alternative palliation, particularly with a view to later
transplantation. Ductal stenting as a bridge to transplantation met
with some success in California,5 but the small UK donor
pool suggested that duct patency without control of pulmonary
blood flow would not allow sufficient time to find a donor organ. We
therefore chose to band both pulmonary arteries and decompress
the left atrium as well as to stent the duct. Ductal stenting in HLH
proved to be technically straightforward, with no procedural mortality
in this group and excellent medium-term maintenance of ductal
patency, with little intimal proliferation. Controlling
pulmonary blood flow proved very difficult, with 4 of the 8
babies dying with heart failure and excessive pulmonary flow
despite the bandings. The 1 long-term survivor (30 months) was well
until he abruptly developed fatal right ventricular
failure.
Duct-Dependent Pulmonary Blood Flow
Stenting the duct when it is long and tortuous (as is common with
pulmonary atresia with ventricular septal defect)
is technically demanding, and even gentle passage of a guidewire may
cause fatal ductal spasm. Even after apparent success, duct
constriction may occur after prostaglandin withdrawal if a
short section of the duct is unstented. Unexplained sudden death
occurred in 2 patients who had patent ducts at autopsy. Sudden death
may occur after palliation with an aortopulmonary
shunt,6 and it seems likely that this may be related to
the underlying disease rather than specifically related to the mode of
palliation.
Redilation of the duct was relatively straightforward, only failing in 1 case, and effectively prolongs palliation. However, the need for repeated redilations raises concern about cumulative radiation dosage.
Endothelialization
A thin but complete layer of endothelium forms
over the stent as early as 1 month. The thickest
endothelium occurred between the mesh of the stent,
with a thinner layer covering the mesh itself.
Neoendothelial proliferation played an important role
in duct-dependent pulmonary blood flow. Although redilation was
successful, there was a gradual recurrent fall in oxygen saturation as
the duct again became compromised by neoendothelium.
Future changes in stent design might reduce endothelial
reaction,7 but at present repeat dilation offers
temporary treatment for endothelial proliferation in
the stented duct, as it does in other parts of the
circulation.8 9
Conclusions
Maintenance of duct patency by stent implantation is
theoretically attractive but in practice is disappointing. With HLH,
the duct may be kept open, but quality of palliation is poor because of
difficulty in controlling pulmonary blood flow even after
banding of the pulmonary arteries; the most recent UK results
of the Norwood operation and its modifications are clearly
superior.
With duct-dependent pulmonary blood flow when the duct is tortuous, there is a risk of fatal ductal spasm. Even when stenting initially appears successful, early duct stenosis may occur unless stent positioning has been exact. However, patients with "disconnected" pulmonary arteries and bilateral ducts, who are difficult to palliate surgically, often have less tortuous ducts, allowing good short- and medium-term palliation to be achieved by stenting; it is also possible to enlarge the stent by repeated balloon dilation as the child grows. The technique may be justified in this rare subgroup of patients with pulmonary atresia, but for the majority of infants with duct-dependent pulmonary or systemic blood flow, ductal stenting cannot be recommended.
Acknowledgments
We are grateful to Drs Philip daCosta, Les Davidson, and Cedric Abbott from the Department of Histopathology, Leeds General Infirmary and to Dr A. Davison from the Department of Histopathology, Freeman Hospital, Newcastle for providing autopsy data.
Received January 11, 1999; revision received March 15, 1999; accepted March 25, 1999.
References
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