Figure 1: A hyperdense central area as seen on the anteroposterior chest X-ray.
Figure 4: Selective pulmonary angiography after deployment of the vascular plug.
The diagnosis of PAVFs is usually established via contrast echocardiography, CT, and angiography. Clinical symptoms, for example cyanosis, may recur due to the redistribution of blood in the pulmonary vascular bed after the closure of PAVFs.[7] In addition, increasing age and pregnancy may be predisposing events when such conditions occur. In the current case, our three-year-old patient was reconsulted for cyanosis. A hyperdense area was seen on the chest X-ray, and the contrast echocardiography showed a premature filling of the left heart chambers. The diagnosis of multiple PAVFs was confirmed by a 3D CT scan and pulmonary angiography; however, no genetic abnormality was noted in this patient.
The first choice of treatment for childhood PAVFs is percutaneous transcatheter embolization via placement of a coil or balloon devices. Surgery is reserved for cases in which this type of intervention is unsuccessful or impossible.[8] Angiographic therapy has become the principal treatment for childhood fistulas in recent years. The drawbacks of the coil are essentially the need to use multiple coils, even for a single vessel, incomplete occlusion, coil reflux, and the high proportion of recanalization.[9] The AVP, which is constructed of nitinol wire mesh, represents a new alternative for PAVF treatment, and it has become increasingly popular in recent years due to its reduced frequency of early and late complications, such as embolic events, thanks to its short occlusion time.
Several published case reports have found the AVP to be safe and efficacious for the treatment of childhood PAVF. Çil et al.[3] reported success with nine applications of the AVP in three PAVF patients. No complications were identified, and successful results were noted. The mean follow-up period for that study was six and a half months. Letourneau- Guillon et al.[10] achieved successful occlusion of 35 feeder arteries using 37 AVPs (a 97% success rate) in 35 PAVF patients between the ages of 11 and 86, and they reported no early complications. However, the recanalization of two vessels (7%) was necessary during the 322-day mean follow-up period.
A cost analysis[3,5] has shown that the AVP is cheaper than the Amplatzer™ Duct Occluder (ADO) or the Gianturco-Grifka vascular occlusion device. In addition, it is also less expensive that the vascular plug coil because of its use of multiple coils. To our knowledge, no literature exists which has compared the economic efficiency of the various interventions for PAVF treatment, but the AVP method seems to rank first thanks to its small introducer system and low cost. The relatively high frequency of post-intervention pleuritis, longer intervention duration, and increased risk of air embolization are the main drawbacks when coils are utilized.
In our case, in the first-stage closure using the AVP II, the patient’s OS rate increased to 95% after closing the two PAVFs, but it dropped to 70% at the one-month follow-up visit. This was explained by the redistribution of the pulmonary blood flow because the third AVF did not increase in size, and no new feeder artery was seen on the control angiogram. The last feeder artery was closed using the AVP I, and no complications were encountered during the 180-day follow-up period.
In conclusion, the use of the AVP I and II for the percutaneous transcatheter occlusion of PAVFs during childhood is dependable and effective.
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