Ann Thorac Surg 2003;76:1923-1928
© 2003 The Society of Thoracic Surgeons
Original article: cardiovascular
Clipped tube fenestration after extracardiac Fontan allows for simple transcatheter coil occlusion
Timothy J. Bradley, MB, ChBa,
Derek G. Human, BM, BCha,
J.A. Gordon Culham, MDa,
Walter J. Duncan, MDa,
Michael W. H. Patterson, MB, ChBa,
Jacques G. LeBlanc, MDa,
Suvro S. Sett, MD*a
a Cardiac Sciences and Department of Radiology, British Columbia's Children's Hospital, Vancouver, British Columbia, Canada
Accepted for publication June 30, 2003.
* Address reprint requests to Dr Sett, Cardiac Sciences, British Columbia's Children's Hospital, 4480 Oak St, Vancouver BC V6H 3V4, Canada
e-mail: ssett{at}cw.bc.ca
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Abstract
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BACKGROUND: Expensive devices are increasingly used to close a patent fenestration after a modified Fontan operation. We report our 5-year institutional experience of clipped tube fenestration after extracardiac Fontan operation, which allows for simple transcatheter coil occlusion.
METHODS: We retrospectively reviewed 30 children, median age of 4.0 years (range, 2.4 to 8.8 years) who underwent extracardiac Fontan operation between May 1996 and May 2001, and were fenestrated using a 4- to 8-mm diameter clipped tube graft.
RESULTS: Ten children had a patent fenestration occluded by transcatheter placement of 15 detachable coils (5- to 8-mm diameter). Aortic oxygen saturations increased on average by 5.5% (2% to 14%) and mean pressures in the Fontan circuit by 2.5 mm Hg (0 to 3 mm Hg). Four had immediate complete occlusion angiographically and 6 had trivial residual shunt, but complete occlusion by echocardiography at follow-up. There have been no immediate complications, late coil embolizations, thromboembolic events, or documented hemolysis within a follow-up after coil implantation of 1.7 years (0.4 to 4.5 years). Spontaneous fenestration closure was documented in 8 patients at cardiac catheterization and 9 patients by echocardiography with consistent improvement in resting transcutaneous oxygen saturation. Two children with a patent fenestration have been considered inappropriate for closure, and there was one early surgical death. There have been no complications related to the tube fenestration modification within a follow-up postoperation of 2.6 years (0.1 to 5.5 years).
CONCLUSIONS: Clipped tube fenestration after extracardiac Fontan operation is a useful surgical modification that allows for simple transcatheter coil occlusion.
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Introduction
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Many modifications to the Fontan procedure have been made since its original description in 1971 [1]. The concept of fenestration was introduced in an attempt to reduce the mortality and morbidity in high-risk patients by allowing a small residual right-to-left shunt [2, 3]. However, in the longer term, a patent fenestration will produce persistent desaturation and leave the patient at risk for a paradoxical embolism [4, 5]. There are increasing reports in the literature describing transcatheter closure of Fontan fenestrations using expensive devices [6–11].
We now routinely perform the extracardiac Fontan operation and have developed a useful surgical modification for fenestrating the circuit using a small tube graft between the extracardiac conduit and the pulmonary venous atrium. Surgical clips can then be used to mark or narrow the midportion of the tube graft, which facilitate location of the fenestration on fluoroscopy and provide stability for anchoring transcatheter coil(s) when closure of a patent fenestration is considered appropriate. Whereas technically this is not a true fenestration, as there is length to the communication, surgical clipping of the tube graft does actually create a window at the narrowest point between the two circulations. To distinguish this from fenestration by direct anastomosis, we have coined the term "clipped tube fenestration," but from a physiologic standpoint there is no difference. The first child in this series has been previously described in detail in a case report [12]. In this report, we present our 5-year experience of using a clipped tube fenestration after the extracardiac Fontan operation to facilitate subsequent transcatheter coil occlusion of persistently patent fenestrations.
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Patients and methods
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Since 1996, at British Columbia's Children's Hospital, we have electively performed a fenestrated extracardiac Fontan operation in all cases. Between May 1996 and May 2001, 30 children underwent the extracardiac Fontan operation with tube fenestration. This excludes an additional 5 children who had the extracardiac Fontan operation, but were fenestrated by direct anastomosis early in this experience according to individual surgeon preference at the time. The median age at surgery was 4.0 years (range, 2.4 to 8.8 years). Initially, all children were maintained postoperatively on Warfarin. Then, from April 2000 onwards, they were included in a multicenter study regarding anticoagulation after the Fontan operation and were randomized to either aspirin or warfarin.
Surgical technique
The extracardiac conduit was constructed by placing a 16- to 20-mm diameter Gore-Tex graft (WL Gore and Associates, Flagstaff, AZ) between the inferior vena cava at the level of the diaphragm and the underside of the mediastinal pulmonary artery for either right- (n = 27) or left- (n = 3) sided grafts. Extracardiac conduits were placed with the child on cardiopulmonary bypass and with the heart beating.
The tube fenestration (Fig 1)
was then constructed using another 4- to 8-mm diameter Gore-Tex graft. This smaller graft was first lined up between the common atrium and extracardiac conduit to get a measure of the length required. The distal end of the fenestration graft was then closed with a 5-0 Gore-Tex suture (for hemostasis), and a circle corresponding to the graft diameter was excised in the side adjacent to the atrial wall. The midportion of the extracardiac conduit was then partially clamped and a small ellipse corresponding to the cross-sectional area of the fenestration graft was removed. The proximal end of the fenestration graft was then sewn end-to-side to this opening with a 5-0 Gore-Tex suture. The atrial wall was then partially clamped and a horizontal incision was made. It was considered an important point to see the endothelium when the atrium was incised, as some atria may be very thick-walled and an incomplete incision would prevent an adequate anastomosis. The circular hole in the distal end of the fenestration graft was then sewn side-to-side to the atrial opening with a 5-0 Gore-Tex suture. This side-to-side anastomosis was done in order to prevent kinking. The fenestration graft was then de-aired and the partial clamp on the atrial wall was removed.
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Fig 1. Clipped tube fenestration. The atrial anastomosis was performed in a side-to-side manner to avoid kinking. The surgical clips were used to facilitate later fluoroscopic localization of the fenestration. The clips were applied in a staggered fashion in an attempt to taper the internal lumen and promote stability of coil placement should later occlusion be necessary.
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According to the intraoperative hemodynamics and oxygen saturation data after separation from cardiopulmonary bypass, the fenestration graft was then marked or narrowed with surgical clip(s) to facilitate later fluoroscopic localization and subsequent transcatheter coil occlusion of persistently patent fenestrations. To reduce the size of the fenestration, a medium-large Weck clip(s) (Ethicon, Johnson and Johnson, Arlington, TX) was placed across the midportion of the fenestration graft. If two or more Weck clips were required, their depth of occlusion was staggered in an attempt to taper the internal lumen and promote stability of coil placement should later occlusion be necessary. The Weck clips had the additional benefit that they could be easily removed by squeezing on the shoulder and gently pulling, and then reapplied as was necessary. The patency of the fenestration was then checked by intraoperative transoesophageal echocardiography. Over the duration of this surgical experience, the larger 8-mm diameter Gore-Tex graft independent of patient size was found to be the best size of tube graft to use to allow the creation of larger anastomoses to the extracardiac conduit and the pulmonary venous atrium and to facilitate the staggered placement of medium-large Weck clips.
Coil implantation technique
Transesophageal echocardiography was performed immediately before cardiac catheterization to detect thrombus, to evaluate flow within the Fontan circuit, and to determine if the fenestration was patent. Percutaneous arterial and venous accesses were obtained. Aortic oxygen saturation (Ao SaO2) and mean pressure within the Fontan circuit (PF) were measured initially. Then, the tube fenestration with the attached Weck clip(s) was easily located on fluoroscopy (Fig 2)
and an angiogram was performed adjacent to this in the extracardiac conduit. For patent fenestrations, a test balloon occlusion was performed with hemodynamic monitoring. The fenestration was then crossed with a nontapered end-hole catheter (Cook [Canada], Toronto, Canada), and a suitably sized Jackson detachable coil (Cook [Canada]) was implanted. Coils were placed astride the surgical clips whenever they had been applied across the midportion of the fenestration (Fig 3).
If repeat angiography detected residual shunting, a second coil implant was attempted. In some cases, additional venous or systemic to pulmonary collaterals were also coil occluded. Finally, Ao SaO2 and PF were remeasured.
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Fig 2. Frontal projection shows the end-hole catheter engaged in the systemic venous side of the tube fenestration. Contrast can be seen entering the pulmonary venous atrium. (Reproduced from Ann Thorac Surg, Sanatani S, et al, Extracardiac Fontan operation with tube fenestration allowing transcatheter coil occlusion, 66, 933–4, Copyright 1998, with permission from The Society of Thoracic Surgeons.)
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Fig 3. Lateral projection shows the first coil astride the narrowing demarcated by the surgical clips. A second coil is being placed on the systemic venous side of the tube fenestration. (Reproduced from Ann Thorac Surg, Sanatani S, et al, Extracardiac Fontan operation with tube fenestration allowing transcatheter coil occlusion, 66, 933–4, Copyright 1998, with permission from The Society of Thoracic Surgeons.)
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Data collection
All medical records and echocardiographic, hemodynamic, and angiographic data were reviewed retrospectively. Age at operation and the diameters of the grafts used to construct the extracardiac conduit and the tube fenestration were recorded. Resting transcutaneous oxygen saturations in room air immediately before operation and the best postoperatively were recorded. Documented spontaneous fenestration closures by transthoracic echocardiography and at cardiac catheterization were noted. For children who underwent transcatheter coil occlusion of the tube fenestration, Ao SaO2 and PF pre- and postcoil implantation were recorded. Number of coils used and presence of residual shunting on final angiography and follow-up echocardiography were noted. Duration of follow-up postoperation and after coil implantation was calculated, and any complications that could be attributed to the clipped tube fenestration surgical modification or transcatheter coil occlusion were sought.
Statistical analysis
Numerical results are given as median (range). Pre– and post–coil implantation measurements of Ao SaO2 and PF were compared with paired Student's t test.
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Results
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An overview of the outcomes is provided (Fig 4).
There have been no complications related to the tube fenestration modification within a follow-up postoperation of up to 5.5 years (median, 2.6 years; range, 0.1 to 5.5 years). No child has had premature closure of the tube fenestration with associated adverse hemodynamics that required additional management or thrombosis related to narrowing of the tube fenestration with the clips in the early postoperative period. All children have had at least a 1-year follow-up, except for a single child (with documented spontaneous fenestration closure), who returned to live overseas.
Ten children had transcatheter coil occlusion of a patent fenestration (Table 1)
by implantation of 15 detachable coils (diameters, 5 to 8 mm). Aortic oxygen saturation increased from 92% (85 to 96%) to 97.5% (94 to 99%), with an average increase of 5.5% (2% to 14%, p < 0.0004). Mean pressure in the Fontan circuit increased from 10.5 mm Hg (7 to 13 mm Hg) to 13 mm Hg (7 to 16 mm Hg), with an average increase of 2.5 mm Hg (0 to 3 mm Hg, p < 0.0004). On final angiography, 4 had complete occlusion immediately and 6 had trivial residual shunting, but all had complete occlusion by echocardiography at follow-up. There have been no immediate complications, late coil embolizations, thromboembolic events, or documented hemolysis within a follow-up post–coil implantation of up to 4.5 years (median, 1.7 years; range, 0.4 to 4.5 years).
Spontaneous fenestration closure was documented in 8 patients at cardiac catheterization (Table 2)
and in 9 patients by echocardiography with consistent improvement in resting transcutaneous oxygen saturation (Table 3).
Two children with a patent fenestration have been considered inappropriate for closure, and there was one early surgical death (Table 4).
The first child had a very difficult recovery after the Fontan operation and has pulmonary arteries and ventricular function considered too marginal for fenestration closure. The other child was found at cardiac catheterization to have a small native left pulmonary artery, which is isolated from the Fontan circuit, so again, fenestration closure has not been considered appropriate. The early surgical mortality was a child who immediately after the Fontan operation remained very unstable with severe metabolic acidosis and low output syndrome. The decision was made to take down the Fontan operation and create a central systemic-t-pulmonary artery shunt, but the child did not survive the second procedure.
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Comment
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Although fenestration during the Fontan operation has been reported to reduce morbidity and mortality [2, 3, 13–16], whether it should be performed routinely remains controversial [17–21]. Whereas initially it was considered more indicated in high-risk patients having a modified Fontan operation [20], more recently it has also been shown to improve short-term outcomes in standard-risk patients [21]. Spontaneous fenestration closure is well recognized, but persistent patency warrants closure to improve arterial oxygen saturations and reduce the risks of right to left shunting [4, 5]. Transcatheter fenestration device occlusion has been reported using the Amplatzer Septal Occluder [6, 9–11], Clamshell [7], CardioSEAL [7, 8], and Starflex devices [8]. Transcatheter coil occlusion of patent fenestrations created surgically by the direct anastomosis technique has been reported in two other small series [22, 23].
The initial report of a tube Fontan fenestration was of a snare-controlled Gore-Tex tube that could be adjusted to control the size of the fenestration allowing for closure in the late postoperative period [24]. Subsequently, there have been two cases reported of tube Fontan fenestrations, including our initial description [12, 25]. In the current series, an 8-mm tube was usually selected to construct a large anastomosis between the extracardiac conduit and the pulmonary venous atrium, including three instances where the underlying surgical anatomy necessitated the conduit to be placed to the left of the heart. Surgical clips were used to narrow the midportion of the fenestration tube according to the hemodynamic data once the patient was weaned from cardiopulmonary bypass, or simply used to mark the fenestration.
The spontaneous fenestration closure rate of 59% (17/29 survivors) compares favorably with those that can be extracted from other series. Only 13% (2/15) were found to have spontaneously closed at cardiac catherization in one series of 17 high-risk patients with univentricular hearts who had the lateral tunnel Fontan operation and baffle fenestration [14]. A larger series reported a 26% (30/117) spontaneous closure rate after successful Fontan operation with baffle fenestration, as demonstrated by lack of any right-to-left shunt by contrast echocardiography [17]. Finally, another series reported a 19% (16/85) spontaneous closure rate after successful lateral tunnel with a baffle fenestration [20]. We envisage that "spontaneous" closure of the tube fenestration is a gradual process of narrowing to a critical point and then occlusion. The timing of this event is likely dependent on pre- and postoperative hemodynamics and the presence of coexisting additional venous or systemic to pulmonary collaterals. Whereas frequent follow-up with serial echocardiography and transcutaneous oxygen saturation monitoring may have allowed an estimate of the timing of "spontaneous" closure, this was not undertaken in this series, as it was not clinically indicated. Furthermore, the duration of beneficial effect from a fenestration in the Fontan circuit will always be individual dependent. More importantly, in this series, there was no instance in this series of premature closure of the tube fenestration that necessitated additional management.
For those children with persistent desaturation in whom the fenestration was found to be patent at cardiac catheterization, the surgical clips facilitated localization of the fenestration on fluoroscopy. For those where the clips had been used to narrow the midportion, this provided stability for anchoring the coil(s) at transcatheter implantation. Although trivial residual shunting was often present initially, complete occlusion was subsequently documented in all cases.
The Jackson detachable coils are easy to use and have the added benefit of being retrievable before release if there are concerns regarding placement. We have no local experience of using devices for fenestration closure, and therefore have no basis for direct cost comparisons. The fluoroscopy screening times were comparable with those for device closure of atrial secundum defects in our lab. The procedural expenses would be otherwise similar, which leaves the cost of the device versus the coil. The cost of any currently available device and deployment system would be in the range of at least 10 to 20 times the cost of a coil and suitable catheter, even if two coils were needed, as in 50% of our cases. A final consideration is that the long-term safety of coils is now well established in the setting of patent ductus arteriosus occlusion, but the outcome data for devices is still evolving.
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Conclusions
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Tube fenestration is a simple and safe surgical modification to the extracardiac Fontan operation. Surgical clipping facilitates localization of the fenestration at cardiac catheterization and allows transcatheter coil occlusion of persistently patent fenestrations as a safe and less expensive alternative to device closure.
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Acknowledgments
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The authors thank Dr Jim Potts and Merrilee Sett for assisting in the preparation of this manuscript.
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References
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Abstract
Introduction
