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1. Use of Amplatzer Fenestrated Atrial Septal Defect Device in a Child with Familial Pulmonary Hypertension

Author

Zahid Amin, Francis Rouault, Alain Fraisse, Marc Humbert, and Philippe Chetaille

Subjects
Cardiac Catheterization, medicine.medical_specialty, Hypertension, Pulmonary, medicine.medical_treatment, Prosthesis Design, Catheterization, Coronary circulation, Coronary Circulation, Internal medicine, medicine.artery, Heart Septum, medicine, Humans, Vascular Patency, Atrial septostomy, Cardiac catheterization, Echocardiography, Doppler, Pulsed, business.industry, Prostheses and Implants, Vascular surgery, medicine.disease, Pulmonary hypertension, Echocardiography, Doppler, Color, Cardiac surgery, medicine.anatomical_structure, Child, Preschool, Pediatrics, Perinatology and Child Health, Pulmonary artery, Exercise Test, Cardiology, Female, Cardiology and Cardiovascular Medicine, business
Abstract

In a 4.5-year-old child with refractory pulmonary arterial hypertension, we performed atrial septostomy with the application of an Amplatzer fenestrated device designed to maintain patency. Continuous intravenous epoprostenol infusion was started concomitantly. Forty-two months after the procedure, the patient had no recurrent syncope and remained in New York Heart Association functional class II. Fenestration of the atrial septum is feasible in children with pulmonary artery hypertension. No conclusion regarding the patient's need for an atrial septal defect can be drawn since concomitant prostanoid therapy was administered. The long-term patency of the atrial communication needs further confirmation and the optimal timing for its application has to be determined.

Published
2006
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3. Parental electrocardiographic screening identifies a high degree of inheritance for congenital and childhood nonimmune isolated atrioventricular block

Author

Hervé Le Marec, Caroline Bonnet, Jean-René Lusson, Raphaël P. Martins, Jean-Jacques Schott, Francis Rouault, Alban-Elouen Baruteau, Jean-Benoit Thambo, Christian Dina, Elisabeth Villain, François Godart, Philippe Mabo, Vincent Probst, Alain Fraisse, François Marçon, Albin Behaghel, Véronique Gournay, Sophie Guillaumont, Yves Dulac, Jean-Marc Schleich, Stéphanie Chatel, Swanny Fouchard, Alain Chantepie, Béatrice Delasalle, Jean-Claude Daubert, unité de recherche de l'institut du thorax UMR1087 UMR6291 ( ITX ), Université de Nantes ( UN ) -Institut National de la Santé et de la Recherche Médicale ( INSERM ) -Centre National de la Recherche Scientifique ( CNRS ), Department de Chirurgie cardiaque des cardiopathies congénitales, Centre chirurgical Marie Lannelongue, Service de cardiologie et maladies vasculaires, Université de Rennes 1 ( UR1 ), Université de Rennes ( UNIV-RENNES ) -Université de Rennes ( UNIV-RENNES ) -Hôpital Pontchaillou-CHU Pontchaillou [Rennes], CIC-IT Rennes, Hôpital Pontchaillou-Institut National de la Santé et de la Recherche Médicale ( INSERM ), Laboratoire Traitement du Signal et de l'Image ( LTSI ), Université de Rennes ( UNIV-RENNES ) -Université de Rennes ( UNIV-RENNES ) -Institut National de la Santé et de la Recherche Médicale ( INSERM ), Laboratoire de génie des procédés - environnement - agroalimentaire ( GEPEA ), Mines Nantes ( Mines Nantes ) -Université de Nantes ( UN ) -Ecole Nationale Vétérinaire, Agroalimentaire et de l'alimentation Nantes-Atlantique ( ONIRIS ) -Centre National de la Recherche Scientifique ( CNRS ), Service de cardiologie pédiatrique [CHU Necker], Assistance publique - Hôpitaux de Paris (AP-HP)-CHU Necker - Enfants Malades [AP-HP], Service cardiologie pédiatrique [Bordeaux], CHU Bordeaux [Bordeaux], Service de Cardiologie Infantile [CHRU Nancy], Centre Hospitalier Régional Universitaire de Nancy ( CHRU Nancy ), Service de cardiologie pédiatrique [Nantes], Centre hospitalier universitaire de Nantes ( CHU Nantes ), Cabinet de Cardiologie pédiatrique, Service de cardiologie pédiatrique [Tours], CHU Tours, Service de cardiologie Pédiatrique [Montpellier], Centre Hospitalier Régional Universitaire [Montpellier] ( CHRU Montpellier ), Service de cardiologie Pédiatrique [Lille], Centre Hospitalier Régional Universitaire [Lille] ( CHRU Lille ), Service de cardiologie pédiatrique et congénitale adulte [CHU de Dijon], Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand ( CHU Dijon ), Service de cardiologie Pédiatrique [Marseille], Hôpital de la Timone [CHU - APHM] ( TIMONE ), Service de cardiologie [Clermont-Ferrand], CHU Clermont-Ferrand, Service pédiatrie-cardiologie, CHU Toulouse [Toulouse]-Hôpital des Enfants, unité de recherche de l'institut du thorax UMR1087 UMR6291 (ITX), Université de Nantes - UFR de Médecine et des Techniques Médicales (UFR MEDECINE), Université de Nantes (UN)-Université de Nantes (UN)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Service de cardiologie et maladies vasculaires [Rennes] = Cardiac, Thoracic, and Vascular Surgery [Rennes], CHU Pontchaillou [Rennes], Hôpital Pontchaillou-Institut National de la Santé et de la Recherche Médicale (INSERM), Laboratoire Traitement du Signal et de l'Image (LTSI), Université de Rennes 1 (UR1), Université de Rennes (UNIV-RENNES)-Université de Rennes (UNIV-RENNES)-Institut National de la Santé et de la Recherche Médicale (INSERM), Laboratoire de génie des procédés - environnement - agroalimentaire (GEPEA), Mines Nantes (Mines Nantes)-Université de Nantes - UFR des Sciences et des Techniques (UN UFR ST), Université de Nantes (UN)-Université de Nantes (UN)-Ecole Nationale Vétérinaire, Agroalimentaire et de l'alimentation Nantes-Atlantique (ONIRIS)-Centre National de la Recherche Scientifique (CNRS), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Centre Hospitalier Régional Universitaire de Nancy (CHRU Nancy), Centre hospitalier universitaire de Nantes (CHU Nantes), Centre Hospitalier Régional Universitaire de Tours (CHRU Tours), Centre Hospitalier Régional Universitaire [Montpellier] (CHRU Montpellier), Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand (CHU Dijon), Hôpital de la Timone [CHU - APHM] (TIMONE), Service de Cardiologie Maladies Vasculaires [CHU Clermont-Ferrand], CHU Gabriel Montpied [Clermont-Ferrand], CHU Clermont-Ferrand-CHU Clermont-Ferrand, CHU Toulouse [Toulouse], Unité de recherche de l'institut du thorax (ITX-lab), Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Université de Nantes - UFR de Médecine et des Techniques Médicales (UFR MEDECINE), Université de Nantes (UN)-Université de Nantes (UN), Centre Chirurgical Marie Lannelongue (CCML), Université de Rennes (UR)-Institut National de la Santé et de la Recherche Médicale (INSERM), Université de Nantes (UN)-Université de Nantes (UN)-École nationale vétérinaire, agroalimentaire et de l'alimentation Nantes-Atlantique (ONIRIS)-Centre National de la Recherche Scientifique (CNRS), Service Cardiologie pédiatrique [CHU Toulouse], Pôle Enfants [CHU Toulouse], Centre Hospitalier Universitaire de Toulouse (CHU Toulouse)-Centre Hospitalier Universitaire de Toulouse (CHU Toulouse), Le Corre, Morgane, and Centre Hospitalier Régional Universitaire de Tours (CHRU TOURS)

Subjects
Male, Parents, MESH : Retrospective Studies, Heart malformation, MESH : NAV1.5 Voltage-Gated Sodium Channel, MESH : Prevalence, MESH : Aged, Electrocardiography, 0302 clinical medicine, MESH: Pregnancy, MESH : Child, Pregnancy, Prenatal Diagnosis, MESH: Child, Medicine, Sinus rhythm, genetics, Child, 0303 health sciences, MESH: Middle Aged, MESH: Genetic Testing, MESH : Infant, MESH: Infant, 3. Good health, MESH : Phenotype, MESH: Young Adult, MESH : Electrocardiography, Child, Preschool, Cardiology and Cardiovascular Medicine, medicine.medical_specialty, conduction, MESH : Young Adult, Prenatal diagnosis, MESH: Phenotype, Sudden death, 03 medical and health sciences, [SDV.MHEP.CSC]Life Sciences [q-bio]/Human health and pathology/Cardiology and cardiovascular system, Physiology (medical), MESH : Adolescent, MESH : Genetic Testing, Humans, MESH : Middle Aged, Genetic Testing, MESH: Prenatal Diagnosis, MESH : Prenatal Diagnosis, MESH: Prevalence, Aged, Retrospective Studies, [SDV.IB] Life Sciences [q-bio]/Bioengineering, MESH: Adolescent, MESH: Parents, MESH: Humans, MESH: Child, Preschool, MESH : Humans, Infant, MESH: Adult, MESH: Retrospective Studies, medicine.disease, MESH : Pregnancy, MESH : Genetic Predisposition to Disease, Atrioventricular block, MESH: Female, Pediatrics, ECG screening, 030204 cardiovascular system & hematology, MESH : Child, Preschool, NAV1.5 Voltage-Gated Sodium Channel, atrioventricular block, Prevalence, Mass Screening, MESH : Female, [ SDV.IB ] Life Sciences [q-bio]/Bioengineering, Family history, MESH : Parents, MESH: Aged, MESH: Infant, Newborn, MESH: Genetic Predisposition to Disease, Middle Aged, MESH : Adult, [ SDV.MHEP.CSC ] Life Sciences [q-bio]/Human health and pathology/Cardiology and cardiovascular system, [SDV.MHEP.CSC] Life Sciences [q-bio]/Human health and pathology/Cardiology and cardiovascular system, MESH : Atrioventricular Block, Phenotype, Female, [SDV.IB]Life Sciences [q-bio]/Bioengineering, medicine.symptom, Adult, Adolescent, pediatrics, MESH : Male, MESH : Infant, Newborn, Asymptomatic, Young Adult, MESH: Atrioventricular Block, Genetic Predisposition to Disease, MESH: Mass Screening, PR interval, 030304 developmental biology, MESH : Mass Screening, business.industry, Infant, Newborn, MESH: NAV1.5 Voltage-Gated Sodium Channel, MESH: Male, MESH: Electrocardiography, business
Abstract

Background— The origin of congenital or childhood nonimmune isolated atrioventricular (AV) block remains unknown. We hypothesized that this conduction abnormality in the young may be a heritable disease. Methods and Results— A multicenter retrospective study (13 French referral centers, from 1980–2009) included 141 children with AV block diagnosed in utero, at birth, or before 15 years of age without structural heart abnormalities and without maternal antibodies. Parents and matched control subjects were investigated for family history and for ECG screening. In parents, a family history of sudden death or progressive cardiac conduction defect was found in 1.4% and 11.1%, respectively. Screening ECGs from 130 parents (mean age 42.0±6.8 years, 57 couples) were compared with those of 130 matched healthy control subjects. All parents were asymptomatic and in sinus rhythm, except for 1 with undetected complete AV block. Conduction abnormalities were more frequent in parents than in control subjects, found in 50.8% versus 4.6%, respectively ( P P P P SCN5A mutation screening identified 2 mutations in 2 patients among 97 children. Conclusions— ECG screening in parents of children affected by idiopathic AV block revealed a high prevalence of conduction abnormalities. These results support the hypothesis of an inheritable trait in congenital and childhood nonimmune isolated AV block.

Published
2012
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4. Characteristics and long-term outcome of non-immune isolated atrioventricular block diagnosed in utero or early childhood: a multicentre study

Author

François Marçon, Caroline Bonnet, Sophie Guillaumont, Elisabeth Villain, Véronique Gournay, François Godart, Albin Behaghel, Yves Dulac, Jean-Benoit Thambo, Swanny Fouchard, Vincent Probst, Jean-Jacques Schott, Jean-Marc Schleich, Alain Chantepie, Francis Rouault, Christophe Leclercq, Alban-Elouen Baruteau, Alain Fraisse, Hervé Le Marec, Jean-Claude Daubert, Jean-René Lusson, Philippe Mabo, Service de cardiologie et maladies vasculaires, Université de Rennes 1 ( UR1 ), Université de Rennes ( UNIV-RENNES ) -Université de Rennes ( UNIV-RENNES ) -Hôpital Pontchaillou-CHU Pontchaillou [Rennes], CIC-IT Rennes, Hôpital Pontchaillou-Institut National de la Santé et de la Recherche Médicale ( INSERM ), Institut du thorax, Université de Nantes ( UN ) -IFR26-Institut National de la Santé et de la Recherche Médicale ( INSERM ), Laboratoire Traitement du Signal et de l'Image ( LTSI ), Université de Rennes ( UNIV-RENNES ) -Université de Rennes ( UNIV-RENNES ) -Institut National de la Santé et de la Recherche Médicale ( INSERM ), unité de recherche de l'institut du thorax UMR1087 UMR6291 ( ITX ), Université de Nantes ( UN ) -Institut National de la Santé et de la Recherche Médicale ( INSERM ) -Centre National de la Recherche Scientifique ( CNRS ), Service de cardiologie pédiatrique [CHU Necker], Assistance publique - Hôpitaux de Paris (AP-HP)-CHU Necker - Enfants Malades [AP-HP], Service cardiologie pédiatrique [Bordeaux], CHU Bordeaux [Bordeaux], Service de Cardiologie Infantile [CHRU Nancy], Centre Hospitalier Régional Universitaire de Nancy ( CHRU Nancy ), Service de cardiologie pédiatrique [Nantes], Centre hospitalier universitaire de Nantes ( CHU Nantes ), Cabinet de Cardiologie pédiatrique, Service de cardiologie pédiatrique [Tours], CHU Tours, Service de cardiologie Pédiatrique [Montpellier], Centre Hospitalier Régional Universitaire [Montpellier] ( CHRU Montpellier ), Service de cardiologie Pédiatrique [Lille], Centre Hospitalier Régional Universitaire [Lille] ( CHRU Lille ), Service de cardiologie pédiatrique et congénitale adulte [CHU de Dijon], Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand ( CHU Dijon ), Service de cardiologie Pédiatrique [Marseille], Hôpital de la Timone [CHU - APHM] ( TIMONE ), Service de cardiologie [Clermont-Ferrand], CHU Clermont-Ferrand, Service pédiatrie-cardiologie, CHU Toulouse [Toulouse]-Hôpital des Enfants, 'Protocole Hospitalier de Recherche Clinique' 2001 R20/03 and 2004 R20/07 from the University Medical Center of Nantes, France, the 2009 Philippe Coumel Research Grant from the French Society of Cardiology, grant no 05 CVD 01 (Preventing Sudden Death) from the Foundation Leducq Trans-Atlantic Network of Excellence, 'Agence Nationale de la Recherche' grant 05-MRAR-028., Service de cardiologie et maladies vasculaires [Rennes] = Cardiac, Thoracic, and Vascular Surgery [Rennes], CHU Pontchaillou [Rennes], Hôpital Pontchaillou-Institut National de la Santé et de la Recherche Médicale (INSERM), Université de Nantes (UN)-IFR26-Institut National de la Santé et de la Recherche Médicale (INSERM), Laboratoire Traitement du Signal et de l'Image (LTSI), Université de Rennes 1 (UR1), Université de Rennes (UNIV-RENNES)-Université de Rennes (UNIV-RENNES)-Institut National de la Santé et de la Recherche Médicale (INSERM), unité de recherche de l'institut du thorax UMR1087 UMR6291 (ITX), Université de Nantes - UFR de Médecine et des Techniques Médicales (UFR MEDECINE), Université de Nantes (UN)-Université de Nantes (UN)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Centre Hospitalier Régional Universitaire de Nancy (CHRU Nancy), Centre hospitalier universitaire de Nantes (CHU Nantes), Centre Hospitalier Régional Universitaire de Tours (CHRU Tours), Centre Hospitalier Régional Universitaire [Montpellier] (CHRU Montpellier), Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand (CHU Dijon), Hôpital de la Timone [CHU - APHM] (TIMONE), Service de Cardiologie Maladies Vasculaires [CHU Clermont-Ferrand], CHU Gabriel Montpied [Clermont-Ferrand], CHU Clermont-Ferrand-CHU Clermont-Ferrand, CHU Toulouse [Toulouse], Université de Rennes (UR)-Institut National de la Santé et de la Recherche Médicale (INSERM), Unité de recherche de l'institut du thorax (ITX-lab), Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Université de Nantes - UFR de Médecine et des Techniques Médicales (UFR MEDECINE), Université de Nantes (UN)-Université de Nantes (UN), Service Cardiologie pédiatrique [CHU Toulouse], Pôle Enfants [CHU Toulouse], Centre Hospitalier Universitaire de Toulouse (CHU Toulouse)-Centre Hospitalier Universitaire de Toulouse (CHU Toulouse), and Centre Hospitalier Régional Universitaire de Tours (CHRU TOURS)

Subjects
Male, Pacemaker, Artificial, Heart disease, MESH : Retrospective Studies, law.invention, Electrocardiography, 0302 clinical medicine, MESH: Pregnancy, MESH : Child, Pregnancy, Prenatal Diagnosis, MESH: Child, Age of Onset, Child, MESH: Treatment Outcome, MESH: Bundle-Branch Block, Clinical outcome, Cardiac Pacing, Artificial, MESH : Infant, MESH: Infant, 3. Good health, MESH : Age of Onset, Pacemaker, MESH: Young Adult, MESH : Electrocardiography, Child, Preschool, Disease Progression, MESH: Pacemaker, Artificial, MESH: Disease Progression, Cardiology and Cardiovascular Medicine, medicine.medical_specialty, MESH : Bundle-Branch Block, MESH: Age of Onset, MESH : Young Adult, MESH: Cardiac Pacing, Artificial, Disease-Free Survival, 03 medical and health sciences, [SDV.MHEP.CSC]Life Sciences [q-bio]/Human health and pathology/Cardiology and cardiovascular system, MESH : Adolescent, Humans, MESH: Prenatal Diagnosis, MESH : Prenatal Diagnosis, Retrospective Studies, MESH: Adolescent, MESH: Humans, MESH: Child, Preschool, MESH : Humans, Infant, MESH: Adult, MESH: Retrospective Studies, MESH : Disease Progression, medicine.disease, MESH : Pregnancy, MESH: Disease-Free Survival, Atrioventricular block, MESH: Female, Pediatrics, Paediatric electrocardiology, 030204 cardiovascular system & hematology, MESH : Child, Preschool, Risk Factors, law, MESH: Risk Factors, MESH : Female, [ SDV.IB ] Life Sciences [q-bio]/Bioengineering, medicine.diagnostic_test, Dilated cardiomyopathy, MESH : Adult, [ SDV.MHEP.CSC ] Life Sciences [q-bio]/Human health and pathology/Cardiology and cardiovascular system, MESH : Risk Factors, MESH : Atrioventricular Block, Treatment Outcome, MESH : Disease-Free Survival, Female, [SDV.IB]Life Sciences [q-bio]/Bioengineering, medicine.symptom, Adult, Adolescent, MESH : Male, Bundle-Branch Block, MESH : Treatment Outcome, Asymptomatic, Young Adult, MESH: Atrioventricular Block, 030225 pediatrics, medicine, business.industry, MESH : Pacemaker, Artificial, Retrospective cohort study, MESH: Male, Surgery, MESH: Electrocardiography, MESH : Cardiac Pacing, Artificial, Artificial cardiac pacemaker, Age of onset, business
Abstract

International audience; AIMS: The natural history of congenital or childhood non-immune, isolated atrioventricular (AV) block is poorly defined. METHODS AND RESULTS: We retrospectively studied 141 children with isolated, non-immune AV block diagnosed in utero, or up to 15 years of age, at 13 French medical centres, between 1980 and 2009. Patients with structural heart disease or maternal antibodies were excluded. Atrioventricular block was asymptomatic in 119 (84.4%) and complete in 100 (70.9%) patients. There was progression to complete AV block in 29/41 (70.7%) patients with incomplete AV block over 2.8 ± 3.4 years (1-155 months), but all patients with incomplete AV block may not have been included in the study. Narrow QRS complex was present in 18 of 26 patients (69.2%) with congenital, and 106 of 115 (92.2%) with childhood AV block. Pacemakers were implanted in 112 children (79.4%), during the first year of life in 18 (16.1%) and before 10 years of age in 90 (80.4%). The mean interval between diagnosis of AV block and pacemaker implants was 2.6 ± 3.9 years (0-300 months). The pacing indication was prophylactic in 70 children (62.5%). During a mean follow-up of 11.6 ± 6.7 years (1-32 years), no patient died or developed dilated cardiomyopathy (DCM). The long-term follow-up was uncomplicated in 127 children (90.1%). CONCLUSION: In this large multicentre study, the long-term outcome of congenital or childhood non-immune, isolated AV block was favourable, regardless of the patient's age at the time of diagnosis. No patient died or developed DCM, and pacemaker-related complications were few.

Published
2012
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5. 341 Parental electrocardiographic screening identifies a high degree of inheritance for congenital and childhood non-immune isolated atrioventricular block

Author

Alain Fraisse, Albin Behaghel, Jean-Marc Schleich, Swanny Fouchard, Alban-Elouen Baruteau, Vincent Probst, Francis Rouault, Yves Dulac, Alain Chantepie, Jean-René Lusson, Christian Dina, Raphaël P. Martins, Elisabeth Villain, François Godart, Sophie Guillaumont, Jean-Claude Daubert, Caroline Bonnet, Stéphanie Chatel, Jean-Jacques Schott, Véronique Gournay, Jean-Benoit Thambo, Hervé Le Marec, François Marçon, and Philippe Mabo

Subjects
Genetics, Inheritance (object-oriented programming), Immune system, business.industry, medicine, medicine.disease, business, Cardiology and Cardiovascular Medicine, Atrioventricular block, Degree (temperature)
Published
2012
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6. Congenital portocaval fistula associated with hepatopulmonary syndrome: ligation vs liver transplantation

Author

Francis Rouault, Stéphane Tercier, Celine Roman, Jean Breaud, Arnauld Delarue, and Philippe Petit

Subjects
Male, medicine.medical_specialty, medicine.medical_treatment, Portal venous pressure, Fistula, Vena Cava, Inferior, Liver transplantation, Asymptomatic, Medicine, Humans, Hepatopulmonary syndrome, Ligation, Vascular Fistula, business.industry, Portal Vein, General Medicine, medicine.disease, Hypoplasia, Surgery, Pulmonary Arteriovenous Fistula, Liver Transplantation, Child, Preschool, Pediatrics, Perinatology and Child Health, Portal hypertension, Radiology, medicine.symptom, business, Hepatopulmonary Syndrome
Abstract

A 4-year-old boy underwent pulmonary testing for diagnosis of exercise-induced dyspnea and subsequent cyanosis. Findings demonstrated the presence of multiple pulmonary arteriovenous fistulas resulting in oxygen desaturation owing to shunting (Pa o 2, 44 mm Hg). Abdominal ultrasound, abdominal computer tomography, and mesenteric angiography revealed an extrahepatic portocaval fistula (PCF), absence of a patent portal vein, and no evidence of portal hypertension. Because these findings were consistent with hepatopulmonary syndrome (HPS), liver transplantation was initially considered. However, subsequent workup using cavofistulography revealed the presence of a hypoplastic portal vein that selective catheterization showed to be threadlike but patent. Based on this finding, a definitive diagnosis of a congenital PCF with hypoplasia of the portal vein (type 2 Abernethy malformation) was made and surgical ligation with transection of the fistula was performed at the age of 5. Treatment was successful without subsequent development of portal hypertension and pulmonary symptoms disappeared. Follow-up examination 4 years later showed that the boy was asymptomatic and that the intrahepatic portal system was patent with normal hepatopetal flow. This is the first reported case of HPS because of portal type 2 Abernethy malformation. Anatomical types of PCF and corresponding therapeutic options in case of HPS are discussed.

Published
2006

7. 325 Clinical presentation and long-term clinical outcomes of non immune, isolated atrioventricular block diagnosed in utero or early childhood

Author

Caroline Bonnet, Francis Rouault, Hervé Le Marec, Alban-Elouen Baruteau, Albin Behaghel, Swanny Fouchard, Yves Dulac, François Marçon, Jean-René Lusson, Sophie Guillaumont, Alain Chantepie, Philippe Mabo, Jean-Benoit Thambo, Christophe Leclercq, Jean-Claude Daubert, Elisabeth Villain, François Godart, Alain Fraisse, Jean-Jacques Schott, Jean-Marc Schleich, Véronique Gournay, Vincent Probst, and Stéphanie Chatel

Subjects
Pediatrics, medicine.medical_specialty, business.industry, medicine.disease, Term (time), Immune system, In utero, embryonic structures, Medicine, Early childhood, Presentation (obstetrics), business, Cardiology and Cardiovascular Medicine, Atrioventricular block, reproductive and urinary physiology
Published
2012
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8. Amiodarone and digoxin for refractory fetal tachycardia

Author

Pierre Djiane, Jean Paul Cano, Andre Serradimigni, Michele Llurens, Philippe Arnoux, Philippe Seyral, Francis Rouault, Alain Potier, and Daniel Unal

Subjects
Adult, Tachycardia, Digoxin, medicine.medical_specialty, medicine.medical_treatment, Amiodarone, Antiarrhythmic agent, Refractory, Pregnancy, Internal medicine, medicine, Humans, Fetus, business.industry, medicine.disease, Fetal Tachycardia, Fetal Diseases, Anesthesia, Cardiology, Female, medicine.symptom, Cardiology and Cardiovascular Medicine, business, medicine.drug
Abstract

A miodarone is an effective antiarrhythmic agent in adults and children. Its main detected metabolite is the compound monodesethyl. Few reports have been published of administration of amiodarone during pregnancy in the treatment of maternal 1-3 or fetal arrhythmias. 4 Its use during pregnancy has been limited because of insufficient knowledge of maternal-fetal transfer, the effects of iodine excess and the dosage needed to achieve efficient fetal concentrations.

Published
1987
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9. Should we close hypoxaemic patent foramen ovale and interatrial shunts on a systematic basis?

Author

Béatrice Bonello, Gilbert Habib, Alain Fraisse, Andreea Dragulescu, Bertrand Jop, Mohammad El Tahlawi, and Francis Rouault

Subjects
Male, Cardiac Catheterization, Time Factors, Heart disease, Septal Occluder Device, Heart Septal Defects, Atrial, Hypoxemia, Medicine, Respiratory system, Interventional closure, Foramen ovale perméable, Hypoxia, Chronic respiratory insufficiency, General Medicine, Middle Aged, Treatment Outcome, Cardiology, Female, France, Fermeture percutanée, medicine.symptom, Respiratory Insufficiency, Cardiology and Cardiovascular Medicine, Shunt (electrical), medicine.medical_specialty, Partial Pressure, Foramen secundum, Foramen Ovale, Patent, Persistent fetal circulation, Hypoxémie, Internal medicine, Humans, In patient, Adverse effect, Aged, Retrospective Studies, business.industry, Patient Selection, Hemodynamics, Retrospective cohort study, Recovery of Function, Oxygenation, medicine.disease, Pulmonary hypertension, Patent foramen ovale, Surgery, Oxygen, Respiratory failure, Chronic Disease, business, Hypoxaemia
Abstract

SummaryBackgroundRarely, hypoxaemia is associated with shunt reversal at the atrial level. Closure by interventional catheterization is the treatment of choice but indications and results have been studied insufficiently.PurposeTo describe our experience with interventional closure of atrial right-to-left shunts described as hypoxaemic and the impact on patient oxygenation and clinical status.MethodRetrospective study in two referral centres, including all patients undergoing closure of interatrial right-to-left shunt associated with hypoxaemia.ResultsSince 2001, 21 consecutive patients underwent interventional shunt closure using the “Amplatzer® device”; two patients had atrial septal defect and 19 had patent foramen ovale. Three patients had minor adverse events; two patients have a tiny residual shunt. Transcutaneous oxygen saturation and partial oxygen pressure increased significantly from 86±5 to 95±3% (p

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10. 242 Clinical presentation and long-term outcome of non immune and isolated atrioventricular block when congenital or diagnosed during childhood: a French multicentric study on 141 patients

Author

Sophie Guillaumont, Alban-Elouen Baruteau, Alain Fraisse, Jean-René Lusson, Jean-Benoit Thambo, Jean-Jacques Schott, Jean-Claude Daubert, Caroline Bonnet, Alexandre Bretonneau, Jean-Marc Schleich, Alice Maltret, Francis Rouault, Philippe Mabo, Vincent Probst, Yves Dulac, Alain Chantepie, Elisabeth Villain, François Godart, Véronique Gournay, Hervé Le Marec, and François Marçon

Subjects
Bradycardia, Pediatrics, medicine.medical_specialty, Heart malformation, business.industry, Dilated cardiomyopathy, medicine.disease, Asymptomatic, Natural history, In utero, medicine, medicine.symptom, Cardiology and Cardiovascular Medicine, business, Complication, Atrioventricular block
Abstract

Purpose When isolated and non immune, prevalence of congenital and childhood atrioventricular blocks (AVB) is extremely low and little is known about their natural history. Methods a multicentric study retrospective from 1980 to 2009 allowed inclusion of 141 children from 13 French referral centers. Included children presented an AVB diagnosed in utero, at birth or during childhood before the age of 15 years, without structural heart abnormalities and without maternal antibodies. Results 26 congenital and 114 childhood AVB were included. Symptoms lead to diagnosis in 15,6% whereas AVB was asymptomatic in 84,4%. 73% AVB were complete and 26,2% AVB were incomplete at first presentation. 21 of these incomplete blocks (56,7%) progressed to permanent complete AVB. Narrow QRS complexes were found in 69,2% congenital and 91,2% childhood AVB. In the 112 (79,4%) implanted children, mean duration between AVB diagnosis and pacemaker implantation was 35 months. Pacemaker primo-implantation occurred during the first year of life for 18 children and 90 children (63.8%) were paced before 10 years old. Pacing was required for symptomatic bradycardia in 37,5% whereas prophylactic cardiac pacing accounted for 61,6%. The median follow-up was 96 months (from 6 to 384 months). 85,1% experienced no complication and neither dilated cardiomyopathy nor death had occurred at last follow-up. Pacemaker-related complications appeared in 11,6%. Conclusion We describe the largest reported experience with isolated and non immune congenital and childhood AVB. Such a block is a nodal damage from unknown origin that may postnatally progress in incomplete forms. Outcome is not influenced by age at diagnosis. Prognosis is very good with no late-onset dilated cardiomyopathy, a few pacemaker-related complications in the modern technological era and no death at last follow-up.

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11. 294 Clinical presentation and long-term outcome of non immune and isolated atrioventricular block when congenital or diagnosed during childhood: a French multicentric study on 141 patients

Author

Yves Dulac, François Marçon, Hervé Le Marec, Jean-René Lusson, Caroline Bonnet, Alexandre Bretonneau, Sophie Guillaumont, Francis Rouault, Jean-Benoit Thambo, Véronique Gournay, Alban-Elouen Baruteau, Alice Maltret, Philippe Mabo, Jean-Marc Schleich, Elisabeth Villain, François Godart, Alain Fraisse, Vincent Probst, Jean-Jacques Schott, Alain Chantepie, and Jean-Claude Daubert

Subjects
Bradycardia, Pediatrics, medicine.medical_specialty, business.industry, Heart malformation, Dilated cardiomyopathy, medicine.disease, Asymptomatic, Natural history, In utero, Medicine, medicine.symptom, business, Complication, Cardiology and Cardiovascular Medicine, Atrioventricular block
Abstract

Purpose When isolated and non immune, prevalence of congenital and childhood atrioventricular blocks (AVB) is extremely low and little is known about their natural history. Methods A multicentric study retrospective from 1980 to 2009 allowed inclusion of 141 children from 13 French referral centers. Included children presented an AVB diagnosed in utero, at birth or during childhood before the age of 15 years, without structural heart abnormalities and without maternal antibodies. Results 26 congenital and 114 childhood AVB were included. Symptoms lead to diagnosis in 15,6% whereas AVB was asymptomatic in 84,4%. 73% AVB were complete and 26,2% AVB were incomplete at first presentation. 21 of these incomplete blocks (56,7%) progressed to permanent complete AVB. Narrow QRS complexes were found in 69,2% congenital and 91,2% childhood AVB. In the 112 (79,4%) implanted children, mean duration between AVB diagnosis and pacemaker implantation was 35 months. Pacemaker primo-implantation occurred during the first year of life for 18 children and 90 children (63.8%) were paced before 10 years old. Pacing was required for symptomatic bradycardia in 37,5% whereas prophylactic cardiac pacing accounted for 61,6%. The median follow-up was 96 months (from 6 to 384 months). 85,1% experienced no complication and neither dilated cardiomyopathy nor death had occurred at last follow-up. Pacemaker-related complications appeared in 11,6%. Conclusion We describe the largest reported experience with isolated and non immune congenital and childhood AVB. Such a block is a nodal damage from unknown origin that may postnatally progress in incomplete forms. Outcome is not influenced by age at diagnosis. Prognosis is very good with no late-onset dilated cardiomyopathy, a few pacemaker-related complications in the modern technological era and no death at last follow-up.

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