Adjunct Targeted Biologic Inhibition Agents to Treat Aggressive Multivessel Intraluminal Pediatric Pulmonary Vein Stenosis

Callahan, Ryan; Kieran, Mark W.; Baird, Christopher W.; Colan, Steven D.; Gauvreau, Kimberlee; Ireland, Christina; Marshall, Audrey C.; Sena, Laureen; Vargas, Sara O.; Jenkins, Kathy J.

doi:10.1016/j.jpeds.2018.01.029

Cited by 76 publications

(57 citation statements)

References 31 publications

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“…(2,14) Callahan, et al evaluated the use of imatinib mesylate with or without bevacizumab targeting neoproliferative myofibroblast-like cells with tyrosine kinase receptor expression, as adjuncts to modern interventional therapies for the treatment of multivessel intraluminal pulmonary vein stenosis. (13) The results of the study were inconclusive with a major confounding factor being the use of other conventional management of the PVS during the study period. The role of these agents needs further evaluation.…”

Section: Resultsmentioning

confidence: 92%

“…(2,10,12) A recent study by Callahan, et al explored the use of adjunct targeted biologic inhibition agents to treat paediatric PVS. (13) Histologic series have identified fibroblasts and myofibroblastlike cells as significant contributors to the mesenchymal proliferation that is a hallmark of pulmonary vein stenosis pathophysiology. (2,14) Callahan, et al evaluated the use of imatinib mesylate with or without bevacizumab targeting neoproliferative myofibroblast-like cells with tyrosine kinase receptor expression, as adjuncts to modern interventional therapies for the treatment of multivessel intraluminal pulmonary vein stenosis.…”

Section: Resultsmentioning

confidence: 99%

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Congenital pulmonary vein stenosis at an African tertiary care centre over a 25-year period

Lebea¹,

Cilliers²,

Ntsinjana³

2019

SAHJ

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INTRODUCTIONCongenital pulmonary vein stenosis (PVS) is a rare condition, resulting from abnormal incorporation of the common pulmonary vein into the left atrium. (1)(2)(3) One or more of the 4 pulmonary veins may be involved and it is commonly associated with other congenital heart defects (CHD), but can occur in isolation. (2)(3)(4) Commonly, patients present with respiratory symptoms with varying degrees of pulmonary hypertension. (5,6) Surgical results are generally poor with a high risk of recurrence and high mortality.(3) Reports are limited to small case series with little data on outcomes. (7) The aim of the study was to describe a series of cases with congenital pulmonary vein stenosis seen at the Chris Hani Baragwanath Academic Hospital, a large Southern African tertiary institution, over a 25-year period. METHODS AND SUBJECTSPatients were retrospectively identified using an electronic paediatric cardiology database system dating from January 1990 to January 2015. Clinical notes, catheterisation and imaging data of patients were reviewed. Data extracted for analysis included patient demographics, site of the lesion and the number of veins affected, associated congenital heart defects (CHD), and management strategies and outcomes. Patients with anomalous venous connection were excluded from this analysis. Background: Congenital pulmonary vein stenosis (PVS) is a rare condition, which results from abnormal embryological incorporation of the common pulmonary vein into the left atrium. Methods: A retrospective descriptive case series study was conducted with the aim of describing the characteristics and outcome of children with congenital PVS at an African tertiary care centre over a 25-year period. A computerised paediatric cardiology database initiated in the early 1990s was sourced to identify patients, following which clinical records were retrieved and reviewed. Results: Five cases of congenital PVS were identifi ed between January 1990 and January 2016 and accounted for 0.0007% of all congenital heart defects seen at the centre during the study period. The age at diagnosis ranged from 22 months -13 years. Most patients presented with respiratory symptoms, with 2 patients presenting with recurrent haemoptysis. The diagnosis of PVS was confi rmed by cardiac catheterisation and pulmonary angiography in all patients. All cases were right-sided unilateral PVS and all were associated with one or more congenital heart defects. Mild pulmonary hypertension and elevated capillary wedge pressures were found in all patients. Only one patient underwent specifi c surgery to relieve the PVS, which subsequently recurred. Another patient underwent a lobectomy following recurrent haemoptysis, but subsequently died of sepsis. Conclusion: Congenital PVS is a rare condition often associated with other congenital cardiac defects. Respiratory symptoms are common at presentation, with haemoptysis forming part of the presenting clinical spectrum, which is in keeping with the published literature. Elevated pulmonary artery pre...

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Section: Resultsmentioning

confidence: 92%

Section: Resultsmentioning

confidence: 99%

Congenital pulmonary vein stenosis at an African tertiary care centre over a 25-year period

Lebea¹,

Cilliers²,

Ntsinjana³

2019

SAHJ

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“…Histological studies of PVS in dogs have shown changes in cell type expression with intimal thickening and a transition from endothelial cell markers to mesenchymal cell markers . Several studies have examined the role of myofibroblast inhibition which has shown limited promise in preventing restenosis in congenital PVS patients . This finding is supported by a translational study by DeSimone et al which found prophylactic PV dilation with a drug‐coated balloon (DCB) immediately after ablation could lower the risk of developing PVS .…”

Section: Discussionmentioning

confidence: 97%

Recurrent pulmonary vein stenosis after successful intervention: Prognosis and management of restenosis

Fender

Widmer

Mahowald

et al. 2019

Cathet Cardio Intervent

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Objectives: The aim of this study was to describe management of recurrent pulmonary vein stenosis (PVS) and determine if stenting is superior to balloon angioplasty (BA) in preventing subsequent restenosis.Background: PVS is a serious complication of atrial fibrillation ablation. BA and stenting are effective therapies; however, restenosis frequently occurs. Here we report management of recurrent stenosis. Methods:This was a prospective observational study performed from 2000 to 2014.Results: One hundred and thirteen patients with severe PVS underwent intervention in 88 veins treated with BA and 81 treated with stenting. Forty-two patients experienced restenosis. Restenosis was more common in veins treated with BA (RRR 53%[95% CI 32-70%, p = .008]). A second intervention was performed in 41 patients. In the 34 vessels treated with initial BA, 24 were treated for restenosis with a stent and 10 were treated with a second BA. The recurrence rate was 46% in those treated with BA followed by stenting and 50% in those treated with two BA procedures. In the 22 veins treated with initial stenting, 9 were treated with another stent and 13 were treated with BA. The recurrence rate was 44% in those treated with a second stent and 46% for those treated with a stent followed by BA. The risk of a third stenosis was the same among all groups (Analysis of variance [ANOVA] p = .99). Limited sample size precluded analysis of outcome by stent size. Conclusions: Restenosis occurred in 44% of patients overall. Management is challenging; stenting does not appear to be superior to BA. K E Y W O R D S pulmonary vein stenosis, vascular patency, stents, balloon angioplasty, catheter ablation 1 | INTRODUCTION Pulmonary vein stenosis (PVS) is estimated to complicate between 0.3 and 3.4% of atrial fibrillation (AF) ablation procedures. 1-4 Severe PVS is frequently symptomatic; over 80% of patients experienced symptoms of shortness of breath, cough, chest pain, hemoptysis, and pulmonary infarction. 5 High grade stenosis can be effectively managed Abbreviations: AF, atrial fibrillation; ANOVA, analysis of variation; BA, balloon angioplasty; BMI, body mass index; BMS, bare metal stent; CHA 2 DS 2 VASc, congestive heart failure, hypertension, age ≥ 75 years, diabetes, stroke or TIA or thromboembolism, vascular disease, age 65-75, sex (female); CI, confidence interval; CT, computed tomography; DCB, drugcoated balloon; DES, drug-eluting stent; HR, hazard ratio; ICE, intracardiac echo; IQR, interquartile range; PV, pulmonary vein; PVS, pulmonary vein stenosis; RR, relative risk; SD, standard deviation; TIA, transient ischemic attack. This work was conducted at the Mayo Clinic, Rochester, MN. ;95:954-958. wileyonlinelibrary.com/journal/ccd All variables were analyzed using software from SAS (version 9.4, Cary, NC). Statistical analysis was carried out on both a per patient and a per vein level. Normally distributed continuous variables are reported as a mean and SD, whereas nonuniformly distributed continuous variables are reported using the median ...

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“…Given these findings, therapy targeting myofibroblast proliferation was attempted in a prospective trial using systemic methotrexate but found significant toxicities and no benefit. Another clinical trial evaluating the use of systemic Imatinib and Bevacizumab began in 2008 has recently been published …”

Section: Discussionmentioning

confidence: 99%

“…Another clinical trial evaluating the use of systemic Imatinib and Bevacizumab began in 2008 has recently been published. 20,21 DES offer the potential benefit of delivering a targeted antiproliferative effect to prevent neo-intimal proliferation as a result of stent implantation without the side effects of systemic therapy. DES have been used extensively in the adult population for coronary artery disease and have been shown to decrease in-stent stenosis.…”

Section: Discussionmentioning

confidence: 99%

Comparison of drug eluting versus bare metal stents for pulmonary vein stenosis in childhood

Khan

Qureshi

Justino

2019

Cathet Cardio Intervent

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Objective Comparison of outcomes using bare metal (BMS) and drug‐eluting (DES) stents in pulmonary vein stenosis (PVS). Background PVS is a serious condition with frequent restenosis after surgical and percutaneous interventions. After experiencing encouraging results with DES, we sought to compare outcomes of BMS and DES in native and post‐surgical PVS. Methods and Results A retrospective review of all patients who underwent stent implantation between 08/93 and 11/17 for PVS at Texas Children's Hospital was performed. BMS were used to treat 58 lesions in 37 patients and 105 DES used to treat 105 lesions in 41 patients. Mean age at first stent implant was 2.9 ± 3.5 years in BMS and 16.2 ± 18.8 months in DES group. Of those with follow‐up catheterization, mean lumen loss rate from stent implant to first follow‐up catheterization was 0.85 ± 1.47 mm/month over 6.4 ± 6.4 months in the BMS group (n = 44 lesions) compared to 0.16 ± 0.31 mm/month over 6.8 ± 7.4 months in the DES group (n = 86 lesions), p < .01. Follow‐up for the BMS group was 14 months (6 days–22.3 years), with 13 mortalities, eight lesions were re‐stented and six complete occlusions were noted. Follow‐up for DES group (including four cross‐overs) was 17.5 months (3 days‐9 years), with 10 mortalities, seven lesions were re‐stented, 11 had complete occlusion, 20 new adjacent lesions in the same vessel underwent stenting and 12 stents were intentionally fractured. Conclusion DES have significantly lowered lumen loss rate when compared to BMS at medium term follow‐up and can be fractured to enable larger diameters. Availability of larger diameter DES would be ideal.

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Adjunct Targeted Biologic Inhibition Agents to Treat Aggressive Multivessel Intraluminal Pediatric Pulmonary Vein Stenosis

Cited by 76 publications

References 31 publications

Congenital pulmonary vein stenosis at an African tertiary care centre over a 25-year period

Congenital pulmonary vein stenosis at an African tertiary care centre over a 25-year period

Recurrent pulmonary vein stenosis after successful intervention: Prognosis and management of restenosis

Comparison of drug eluting versus bare metal stents for pulmonary vein stenosis in childhood

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