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3. Edoxaban for Thromboembolism Prevention in Pediatric Patients With Cardiac Disease

Author

Brothers, Matthew, Marino, Bradley, Canter, Charles, Law, Mark, Nguyen, Nguyenvu, Sang, Charlie, Shimano, Kristin, Gupta, Dipankar, Portman, Michael, Williams, Derek, Glass, Lauren, Sperrazza, Charles, Herold, Steven, Garg, Ruchira, Vranicar, Mark, Awad, Sawsan, Asante-Korang, Alfred, Druzgal, Colleen, Ozment, Caroline, Del Toro, Kamill, Roses, Ferran, Jux, Christian, Gravenhorst, Verena, Schweigmann, Ulrich, Bhatt, Mihir, Sabapathy, Christine, Dahdah, Nagib, Bartonicek, Dototea, Tulzer, Gerald, Basargina, Elena, Zvereva, Tatiana, Pertels, Tatiana, Plotnikova, Irina, Pierre-Emmanuel, S.E.G.U.E.L.A., Amedro, Pascal, Yves, Dulac, BONNET, Damien, Saraco, Paola, Rimini, Alessandro, Digtiar, Valerii, Gonchar, Margaryta, Kryuchko, Tetyana, Yablon, Olga, Bedi, Varinder Singh, Patel, Jashvant, Mitra, Monjori, Kusa, Jacek, Domagala, Kowalczyk, KÖRNYEI, László, BERECZKI, Csaba, ABLONCZY, László, Levitas, Vivianne Aviva, Mishali, David, Revel-Vilk, Shoshana, Harlev, Dan, Sasmaz, Hatice Ilgen, Ozbek, Namik Yasar, Unal, Sule, Patıroglu, Türkan, Malbora, Baris, Agin, Hasan, Karakas, Zeynep, Kavakli, Ramazan Kaan, Chalmers, Elizabeth, Bu'Lock, Frances, Daubeney, Piers, Hamza, Hala, Badr, Mohamed, Elalfy, Mohsen, Mansour, Ahmed, Hassab, Hoda, Sabry, Ayman, Daou, Linda, Bitar, Fadi, Portman, Michael A., Jacobs, Jeffrey P., Newburger, Jane W., Berger, Felix, Grosso, Michael A., Duggal, Anil, Tao, Ben, and Goldenberg, Neil A.

Published
2022
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4. Histopathology of congestive nephropathy: a case description and literature review.

Author

Husain‐Syed, Faeq, Rangaswami, Janani, Núñez, Julio, Skrzypek, Susanne, Jux, Christian, Gröne, Hermann‐Josef, and Birk, Horst‐Walter

Subjects
CARDIO-renal syndrome, ACUTE kidney failure, LITERATURE reviews, HEART failure, CELL differentiation, KIDNEY diseases, INTRA-abdominal hypertension
Abstract

Congestive nephropathy is an underappreciated manifestation of cardiorenal syndrome and is characterized by a potentially reversible kidney dysfunction caused by a reduced renal venous outflow secondary to right‐sided heart failure or intra‐abdominal hypertension. To date, the histological diagnostic criteria for congestive nephropathy have not been defined. We herein report a case of acute renal dysfunction following cardiac allograft failure and present a review of the relevant literature to elucidate the current understanding of the disease. Our case demonstrated that congestion‐driven nephropathy may be histopathologically characterized by markedly dilated veins and peritubular capillaries, focally accentuated low‐grade acute tubular damage, small areas of interstitial fibrosis, and tubular atrophy on a background of normal glomeruli and predominantly normal tubular cell differentiation. [ABSTRACT FROM AUTHOR]

Published
2024
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8. Associated Anomalies and Outcome in Patients with Prenatal Diagnosis of Aortic Arch Anomalies as Aberrant Right Subclavian Artery, Right Aortic Arch and Double Aortic Arch.

Author

Axt-Fliedner, Roland, Nazar, Asia, Bedei, Ivonne, Schenk, Johanna, Reitz, Maleen, Rupp, Stefan, Jux, Christian, and Wolter, Aline

Subjects
SUBCLAVIAN artery, THORACIC aorta, PRENATAL diagnosis, MIRROR images, DOWN syndrome, SURVIVAL rate
Abstract

We aimed to evaluate retrospectively associated anomalies and outcome in prenatal aortic arch anomalies (AAAs). We included ninety patients with aberrant right subclavian artery (ARSA), right aortic arch (RAA) with mirror image branching (RAA-mirror) or aberrant left subclavian artery (RAA-ALSA) and double aortic arch (DAA) between 2011 and 2020. In total, 19/90 (21.1%) had chromosomal anomalies, the highest rate being within the ARSA subgroup (17/46, 37%). All (13/13) of the RAA-mirror subgroup, 10/27 (37.0%) of RAA-ALSA, 13/46 (28.3%) of ARSA and 0/4 within the DAA subgroup had additional intracardiac anomaly. The rate of extracardiac anomalies was 30.7% in RAA-mirror, 28.3% in ARSA, 25.0% in DAA and 22.2% in the RAA-ALSA subgroup. A total of 42/90 (46.7%) had isolated AAAs: three (7.1%) with chromosomal anomalies, all trisomy 21 (3/26, 11.5%) within the ARSA subgroup. Out of 90, 19 (21.1%) were lost to follow-up (FU). Two (2.2%) intrauterine deaths occurred, and six (6.7%) with chromosomal anomalies terminated their pregnancy. In total, 63 (70.0%) were liveborn, 3/63 (4.8%) with severe comorbidity had compassionate care and 3/60 (5.0%) were lost to FU. The survival rate in the intention-to-treat cohort was 53/57 (93%). Forty-one (77.4%) presented with vascular ring/sling, two (4.9%) with RAA-ALSA developed symptoms and one (2.4%) needed an operation. We conclude that intervention due to vascular ring is rarely necessary. NIPT could be useful in isolated ARSA cases without higher a priori risk for trisomy 21 and after exclusion of other anomalies. [ABSTRACT FROM AUTHOR]

Published
2024
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10. Somatic Development in Children with Congenital Heart Defects

Author

Kerst, Gunter, Vazquez-Jimenez, Jaime F., Gkalpakiotis, Dimitrios, Schedifka, Andrea, Buheitel, Gernot, Streble, Joachim, II, Kececioglu, Deniz, Sandica, Eugen, Trusen, Burkhard, Berger, Felix, Miera, Oliver, Ovroutski, Stanislav, Peters, Björn, Schmitt, Katharina, Schubert, Stephan, Photiadis, Joachim, Opgen-Rhein, Bernd, Weiss, Katja, Berns, Christoph, Blumenthal-Barby, Carl-Christian, Boeckel, Thomas, Krusche, Susanne, Busse, Jürgen, Kästner, Andreas, Koch, Heike, Köpcke, Christian, Streichan, Frank, Timme, Jens, Franzbach, Birgit, Senft, Gabriela, Beyer, Frank, Winter, Klaus, Breuer, Johannes, Welz, Armin, Bahlmann, Jens, Griese, Eberhard, Lê, Trong Phi, Hebe, Joachim, Nürnberg, Jan-Hendrik, Magsaam, Annette, Müller, Ronald, Potthoff, Ludger, Voigt, Renate, Krüger, Tim, Gerleve, Hubert, Kleideiter, Ulrich, Schneider-Kulla, Dirk, Feil, Elmo, Menke, Thomas, Lehn, Martin, Grohmann, Martina, Tomczak, Helge, Krogmann, Otto N., Tarusinov, Gleb, Scheld, Michael, Mayatepek, Ertan, Pillekamp, Frank, Lichtenberg, Artur, Terpeluk, Christiane, von Jan, Veronika, Dittrich, Sven, Gundlach, Ulrike, Cesnjevar, Robert, Neudorf, Ulrich, Morf, Geert, Esmaeili, Anoosh, Backhoff, Stephan, Stiller, Brigitte, Beyersdorf, Friedhelm, Kroll, Johannes, Wildberg, Anke, Siaplaouras, Jannos, Masri-Zada, Antje, Jux, Christian, Böning, Andreas, Akintürk, Hakan, Paul, Thomas, Tirilomis, Theodor, Schürer, Gabriele, Hartmann, Johannes, Grabitz, Ralph, Liebaug, Uta, Rotzsch, Claudius, Kozlik-Feldmann, Rainer, Sachweh, Jörg, Riso, Arlindo, Petersen, Volker, Friedrich, Bernd, Schlobohm, Otmar, Beerbaum, Philipp, Böthig, Dietmar, Horke, Alexander, Bauersachs, Johann, Westhoff-Bleck, Mechthild, Gorenflo, Matthias, Karck, Matthias, Loukanov, Tsvetomir, Sebening, Christian, Schrüfer, Hermann, Wilken, Martin, Abdul-Khaliq, Hashim, Rädle-Hurst, Tanja, Rentzsch, Axel, Schäfers, Hans-Joachim, Reichert, Hagen, Kriebel, Thomas, Boysen, Arnulf, Kramer, Hans-Heiner, Cremer, Joachim Thomas, Scheewe, Jens, Buchholz-Berdau, Regina, Möller, Peter, Ram, Wolfgang, Brockmeier, Konrad, Bennink, Gerardus B.W.E., Gillor, Alex, Niehues, Tim, Terhoeven, Peter, Leidig, Steffen, Dähnert, Ingo, Kinzel, Peter, Kostelka, Martin, Kändler, Liane, Bethge, Martin, Köster, Stefan, Schröder, Christoph, Karstedt, Jens, Seitz, Uwe, Kampmann, Christoph, Vahl, Christian-Friedrich, Stahl, Frank, Barth, Helmut, Müller-Scholden, Joachim, Ewert, Peter, Hager, Alfred, Kaemmerer, Harald, Nagdyman, Nicole, Schoetzau, Jörg, Tutarel, Oktay, Lange, Rüdiger, Haas, Nikolaus A., Hakami, Lale, Hauser, Michael, Roithmaier, Alexander, Kehl, Hans Gerd, Malec, Edward, Baumgartner, Helmut, Diller, Gerhard, Bahle, Roswitha, Hofner, Gerald, Zink, Stefan, Reif, Roland, Parlasca, Christoph, Freund, Matthias W., Schumacher, Michael, Darrelmann, Christine, Motz, Reinald, Willmann, Olaf, Schmiedl, Norbert, Quick, Peter, Hillebrand, Dirk, Hirsemann, Petra, Eiselt, Stephan Michele, Nekarda, Torsten, Eberhard, Michael, Schneider, Martin, Asfour, Boulos, Baier, Georg, Uhlemann, Frank, Scheid, Michael, Beyer, Alexander, Binz, Gudrun, Teufel, Thomas, Handke, Ronald-Peter, Hofbeck, Michael, Kaulitz, Renate, Sieverding, Ludger, Schlensak, Christian, Apitz, Christian, Kupferschmid, Christoph, Holtvogt, Jürgen, Wippermann, Carl-Friedrich, Heusch, Andreas, Wirbelauer, Johannes, Brosi, Wolfgang, Poryo, Martin, Paes, Laura Antonia, Pickardt, Thomas, Bauer, Ulrike M.M., Meyer, Sascha, and Wagenpfeil, Stefan

Published
2018
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18. Genetic Screening Reveals Heterogeneous Clinical Phenotypes in Patients with Dilated Cardiomyopathy and Troponin T2 Variants.

Author

Weis, Angelika, Krueck, Svenja, Dombrowsky, Gregor, Schänzer, Anne, Jux, Christian, Uebing, Anselm, Voges, Inga, Hitz, Marc-Phillip, and Rupp, Stefan

Subjects
GENETIC testing, DILATED cardiomyopathy, TROPONIN, PHENOTYPES, PATIENTS' families, MICROFILAMENT proteins
Abstract

Background: Cardiomyopathies (CMs) are a heterogeneous and severe group of diseases that shows a highly variable cardiac phenotype and an incidence of app. 1/100.000. Genetic screening of family members is not yet performed routinely. Patients and methods: Three families with dilated cardiomyopathy (DCM) and pathogenic variants in the troponin T2, Cardiac Type (TNNT2) gene were included. Pedigrees and clinical data of the patients were collected. The reported variants in the TNNT2 gene showed a high penetrance and a poor outcome, with 8 of 16 patients dying or receiving heart transplantation. The age of onset varied from the neonatal period to the age of 52. Acute heart failure and severe decompensation developed within a short period in some patients. Conclusion: Family screening of patients with DCM improves risk assessment, especially for individuals who are currently asymptomatic. Screening contributes to improved treatment by enabling practitioners to set appropriate control intervals and quickly begin interventional measures, such as heart failure medication or, in selected cases, pulmonary artery banding. [ABSTRACT FROM AUTHOR]

Published
2023
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21. Exercise-dependent changes in ventricular–arterial coupling and aortopulmonary collateral flow in Fontan patients: a real-time CMR study.

Author

Latus, Heiner, Hofmann, Lucas, Gummel, Kerstin, Khalil, Markus, Yerebakan, Can, Waschulzik, Birgit, Schranz, Dietmar, Voges, Inga, Jux, Christian, and Reich, Bettina

Subjects
PULMONARY circulation, VENA cava superior, COLLATERAL circulation, MAGNETIC resonance imaging, EXERCISE, RESEARCH funding, EMPLOYEES' workload, AORTA, CARDIOPULMONARY bypass, ERGOMETRY, VENA cava inferior, HEMODYNAMICS, HEART failure
Abstract

Aims Inefficient ventricular–arterial (V–A) coupling has been described in Fontan patients and may result in adverse haemodynamics. A varying amount of aortopulmonary collateral (APC) flow is also frequently present that increases volume load of the single ventricle. The aim of the study was to assess changes in V–A coupling and APC flow during exercise CMR. Methods and results Eighteen Fontan patients (age 24 ± 3 years) and 14 controls (age 23 ± 4 years) underwent exercise CMR using a cycle ergometer. Ventricular volumetry and flow measurements in the ascending aorta (AAO), inferior (IVC), and superior (SVC) vena cava were assessed using real-time sequences during stepwise increases in work load. Measures of systemic arterial elastance Ea, ventricular elastance Ees, and V–A coupling (Ea/Ees) were assessed. APC flow was quantified as AAO − (SVC + IVC). Ea remained unchanged during all levels of exercise in both groups (P = 0.39 and P = 0.11). Ees increased in both groups (P = 0.001 and P < 0.001) with exercise but was lower in the Fontan group (P = 0.04). V–A coupling was impaired in Fontan patients at baseline (P = 0.04). Despite improvement during exercise (P = 0.002) V–A coupling remained impaired compared with controls (P = 0.001). Absolute APC flow in Fontan patients did not change during exercise even at maximum work load (P = 0.98). Conclusions Inefficient V–A coupling was already present at rest in Fontan patients and aggravated during exercise due to a limited increase in ventricular contractility which demonstrates the importance of a limited functional reserve of the single ventricle. APC flow remained unchanged suggesting no further increase in volume load during exercise. [ABSTRACT FROM AUTHOR]

Published
2023
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24. Somatic Development in Children with Congenital Heart Defects

Author

Poryo, Martin, Paes, Laura Antonia, Pickardt, Thomas, Bauer, Ulrike M.M., Meyer, Sascha, Wagenpfeil, Stefan, Abdul-Khaliq, Hashim, Kerst, Gunter, Vazquez-Jimenez, Jaime F., Gkalpakiotis, Dimitrios, Schedifka, Andrea, Buheitel, Gernot, Streble, Joachim, II, Kececioglu, Deniz, Sandica, Eugen, Trusen, Burkhard, Berger, Felix, Miera, Oliver, Ovroutski, Stanislav, Peters, Björn, Schmitt, Katharina, Schubert, Stephan, Photiadis, Joachim, Opgen-Rhein, Bernd, Weiss, Katja, Berns, Christoph, Blumenthal-Barby, Carl-Christian, Boeckel, Thomas, Krusche, Susanne, Busse, Jürgen, Kästner, Andreas, Koch, Heike, Köpcke, Christian, Streichan, Frank, Timme, Jens, Franzbach, Birgit, Senft, Gabriela, Beyer, Frank, Winter, Klaus, Breuer, Johannes, Welz, Armin, Bahlmann, Jens, Griese, Eberhard, Lê, Trong Phi, Hebe, Joachim, Nürnberg, Jan-Hendrik, Magsaam, Annette, Müller, Ronald, Potthoff, Ludger, Voigt, Renate, Krüger, Tim, Gerleve, Hubert, Kleideiter, Ulrich, Schneider-Kulla, Dirk, Feil, Elmo, Menke, Thomas, Lehn, Martin, Grohmann, Martina, Tomczak, Helge, Krogmann, Otto N., Tarusinov, Gleb, Scheld, Michael, Mayatepek, Ertan, Pillekamp, Frank, Lichtenberg, Artur, Terpeluk, Christiane, von Jan, Veronika, Dittrich, Sven, Gundlach, Ulrike, Cesnjevar, Robert, Neudorf, Ulrich, Morf, Geert, Esmaeili, Anoosh, Backhoff, Stephan, Stiller, Brigitte, Beyersdorf, Friedhelm, Kroll, Johannes, Wildberg, Anke, Siaplaouras, Jannos, Masri-Zada, Antje, Jux, Christian, Böning, Andreas, Akintürk, Hakan, Paul, Thomas, Tirilomis, Theodor, Schürer, Gabriele, Hartmann, Johannes, Grabitz, Ralph, Liebaug, Uta, Rotzsch, Claudius, Kozlik-Feldmann, Rainer, Sachweh, Jörg, Riso, Arlindo, Petersen, Volker, Friedrich, Bernd, Schlobohm, Otmar, Beerbaum, Philipp, Böthig, Dietmar, Horke, Alexander, Bauersachs, Johann, Westhoff-Bleck, Mechthild, Gorenflo, Matthias, Karck, Matthias, Loukanov, Tsvetomir, Sebening, Christian, Schrüfer, Hermann, Wilken, Martin, Abdul-Khaliq, Hashim, Rädle-Hurst, Tanja, Rentzsch, Axel, Schäfers, Hans-Joachim, Reichert, Hagen, Kriebel, Thomas, Boysen, Arnulf, Kramer, Hans-Heiner, Cremer, Joachim Thomas, Scheewe, Jens, Buchholz-Berdau, Regina, Möller, Peter, Ram, Wolfgang, Brockmeier, Konrad, Bennink, Gerardus B.W.E., Gillor, Alex, Niehues, Tim, Terhoeven, Peter, Leidig, Steffen, Dähnert, Ingo, Kinzel, Peter, Kostelka, Martin, Kändler, Liane, Bethge, Martin, Köster, Stefan, Schröder, Christoph, Karstedt, Jens, Seitz, Uwe, Kampmann, Christoph, Vahl, Christian-Friedrich, Stahl, Frank, Barth, Helmut, Müller-Scholden, Joachim, Ewert, Peter, Hager, Alfred, Kaemmerer, Harald, Nagdyman, Nicole, Schoetzau, Jörg, Tutarel, Oktay, Lange, Rüdiger, Haas, Nikolaus A., Hakami, Lale, Hauser, Michael, Roithmaier, Alexander, Kehl, Hans Gerd, Malec, Edward, Baumgartner, Helmut, Diller, Gerhard, Bahle, Roswitha, Hofner, Gerald, Zink, Stefan, Reif, Roland, Parlasca, Christoph, Freund, Matthias W., Schumacher, Michael, Darrelmann, Christine, Motz, Reinald, Willmann, Olaf, Schmiedl, Norbert, Quick, Peter, Hillebrand, Dirk, Hirsemann, Petra, Eiselt, Stephan Michele, Nekarda, Torsten, Eberhard, Michael, Schneider, Martin, Asfour, Boulos, Baier, Georg, Uhlemann, Frank, Scheid, Michael, Beyer, Alexander, Binz, Gudrun, Teufel, Thomas, Handke, Ronald-Peter, Hofbeck, Michael, Kaulitz, Renate, Sieverding, Ludger, Schlensak, Christian, Apitz, Christian, Kupferschmid, Christoph, Holtvogt, Jürgen, Wippermann, Carl-Friedrich, Heusch, Andreas, Wirbelauer, Johannes, and Brosi, Wolfgang

Published
2018
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28. Tuberculosis-Associated HLH in an 8-Month-Old Infant: A Case Report and Review

Author

Hauch, Holger, Skrzypek, Susanne, Woessmann, Wilhelm, Lehmberg, Kai, Ehl, Stephan, Speckmann, Carsten, Schneck, Emmanuel, Koerholz, Dieter, Jux, Christian, and Neuhäuser, Christoph

Subjects
tuberculosis, hemophagocytic lymphohistiocytosis, lung failure, case report, extracorporeal membrane oxygenation, ECMO, Pediatrics, infant
Abstract

Hemophagocytic lymphohistiocytosis (HLH) is a rare immunological disease, which can be mistaken for sepsis easily. Among the infectious causes that may trigger secondary HLH, tuberculosis (TBC), a rather rare pathogen nowadays, is typical. To our knowledge, this is the first case report of an infant suffering from TBC-associated HLH-induced acute respiratory failure who was treated successfully using extracorporeal membrane oxygenation. An 8-month-old boy with fever (over the last 8 wk) and pancytopenia was transferred to our institution with acute respiratory failure and for extracorporeal membrane oxygenation therapy. Bone marrow biopsy revealed hemophagocytosis. Immunological work-up for familial HLH was negative. In a desperate search for the cause of secondary HLH, an interferon-gamma release assay for TBC returned positive. However, microscopy for acid-fast bacteria as well as polymerase chain reaction for TBC were initially negative. Despite this, the child was treated with tuberculostatic therapy. TBC was finally confirmed. The child remained on extracorporeal membrane oxygenation for 28 d. Further work-up showed typical lesions of disseminated TBC. The mother was identified as the source of TBC. The boy presents with mild sequelae (fine motor skills). In infants with suspected septicemia, TBC should be considered as differential diagnosis even if the results are initially negative.

Published
2020

39. Single Nuclei Sequencing Reveals Novel Insights Into the Regulation of Cellular Signatures in Children With Dilated Cardiomyopathy.

Author

Nicin, Luka, Abplanalp, Wesley T., Schänzer, Anne, Sprengel, Anke, John, David, Mellentin, Hannah, Tombor, Lukas, Keuper, Matthias, Ullrich, Evelyn, Klingel, Karin, Dettmeyer, Reinhard B., Hoffmann, Jedrzej, Akintuerk, Hakan, Jux, Christian, Schranz, Dietmar, Zeiher, Andreas M., Rupp, Stefan, and Dimmeler, Stefanie

Published
2021
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40. Symptom-orientated oral hygiene for children after heart transplantation: Effectiveness of a standardized prophylactic program.

Author

Schulz-Weidner, Nelly, Bulski, Julia C., Siahi-Benlarbi, Rachida, Bauer, Jürgen, Logeswaran, Thushiha, Jux, Christian, Wetzel, Willi-Eckhard, and Krämer, Norbert

Subjects
ORAL hygiene, CHILDREN'S health, HEART transplantation, GINGIVITIS, MOUTHWASHES, GINGIVAL hyperplasia, TOOTHBRUSHES
Abstract

Children with CHD, especially heart-transplanted patients, are predisposed to have caries lesions, gingivitis and other oral findings like gingival hyperplasia. The aim of the study was the implementation of a specific oral hygiene program in these patients and its effect on the improvement of oral health, especially gingival overgrowth. For this, we used a newly developed systematic GHI to evaluate and describe this gingival alteration. Thirty-three children, aged 6 to 15 years with cardiac transplants (9 girls, 24 boys), were examined and introduced into a specific oral hygiene program. Each child showed evidence of gingival hyperplasia. They were randomly divided into three groups with the following oral care measurements: Group ZZ tooth brushing, Group ZZS tooth brushing and mouth rinsing, Group ZZSS tooth brushing, mouth rinsing and the use of an additional single and sulcus toothbrush. A significant decline of all oral health parameters could be proven in all groups. Gingival hyperplasia (GHI) improved as well as plaque accumulation (QHI). The children who used in addition to toothbrushing rinsing solutions and/or additional miniature toothbrushes showed better parameters of the gingival hygiene indexes from the baseline examination until the end of the study. The results show that any infant with cardiac transplant has to be introduced into an individualized oral hygiene program underlining the need of comprehensive dental care in cooperation with pediatric cardiology. [ABSTRACT FROM AUTHOR]

Published
2020
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42. Interrelationship Between Hemodynamics, Brain Volumes, and Outcome in Hypoplastic Left Heart Syndrome.

Author

Reich, Bettina, Heye, Kristina N., O'Gorman Tuura, Ruth, Beck, Ingrid, Wetterling, Kristina, Hahn, Andreas, Aktintürk, Hakan, Schranz, Dietmar, Jux, Christian, Kretschmar, Oliver, Hübler, Michael, Latal, Beatrice, and Knirsch, Walter

Abstract

The long-term impact of altered hemodynamics after stage II in children with hypoplastic left heart syndrome (HLHS) and hypoplastic left heart complex (HLHC) on cerebral growth and neurodevelopmental outcome is unknown. We aimed to investigate whether elevated central venous and atrial filling pressures before the Fontan procedure may be associated with smaller brain volumes and poorer neurodevelopmental outcome after the initial hybrid procedure. In a two-center cohort study semiautomated segmentation of cerebral magnetic resonance imaging scans was conducted in 25 children with HLHS/HLHC (25 hybrid) before the Fontan procedure (27.6 ± 4.3 months) and in 8 healthy control subjects (29.7 ± 9.5 months). Study patients were evaluated with the Bayley Scales of Infant and Toddler Development III (Bayley-III) and a neurologic examination. Hemodynamic measures after stage II were assessed with cardiac catheterization at 2 years of age before Fontan completion. Children with known genetic comorbidities were excluded. In HLHS/HLHC patients higher atrial filling pressures (6 ± 3 mm Hg; range, 2–14) were correlated with reduced brain volumes and lower language composite score, whereas higher Glenn pressures (10 ± 3 mm Hg; range, 6–16) were related to higher cerebrospinal fluid, reduced brain volumes, and lower cognitive, language, and motoric composite scores in the Bayley-III. Compared with control subjects white matter volumes were reduced and cerebrospinal fluid volumes increased in study patients. These data suggest that altered cardiovascular hemodynamics after stage II influence brain growth and neurodevelopmental outcome in infants with HLHS/HLHC. [ABSTRACT FROM AUTHOR]

Published
2019
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44. Intracoronary bone marrow cell application for terminal heart failure in children

Author

Rupp, Stefan, Jux, Christian, Bönig, Halvard, Bauer, Jürgen, Tonn, Torsten, Seifried, Erhard, Dimmeler, Stefanie, Zeiher, Andreas M., Schranz, Dietmar, and Justus Liebig University Giessen

Subjects
ddc:610
Abstract

Introduction In spite of tremendous progress in the medical and surgical treatment of children with congenital heart disease and dilated cardiomyopathy achieved during the past few decades, for some children a heart transplant remains the only option. Clinically relevant benefits of intracoronary injection of autologous stem cells on cardiac function and remodelling have been demonstrated in adult patients with acute myocardial infarction. Experience with autologous stem cell therapy in children with severe congenital or acquired pump failure is limited to a small number of case reports. Method and results Between 2006 and 2010, nine severely ill children were treated with intracoronary infusion of autologous bone marrow-derived mononuclear cells as part of a compassionate therapy in our centre. No procedure-related unexpected adverse events occurred. There was one patient on extracorporeal membrane oxygenation who died of haemorrhage unrelated to the procedure; three patients proceeded to heart transplantation once a donor heart became available. The other five patients showed an improvement with respect to New York Heart Association classification (greater than or equal to 1), brain natriuretic peptide serum levels, and ejection fraction. Conclusion Similar to adults, intracoronary injection of autologous bone marrow cell is technically feasible and safe for children. On the basis of our data, we propose to perform a pilot study for children with congestive heart failure, to formally assess the efficacy of intracoronary autologous bone marrow cell therapy.

Published
2012
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47. Cardiovascular magnetic resonance assessment of ventricular function and myocardial scarring before and early after repair of anomalous left coronary artery from the pulmonary artery.

Author

Latus, Heiner, Gummel, Kerstin, Rupp, Stefan, Mueller, Matthias, Jux, Christian, Kerst, Gunter, Akintuerk, Hakan, Bauer, Juergen, Schranz, Dietmar, and Apitz, Christian

Subjects
HEART ventricles, MAGNETIC resonance imaging, MYOCARDIUM, RESEARCH funding, SCARS, T-test (Statistics), PRE-tests & post-tests, CONTRAST media, RETROSPECTIVE studies, DATA analysis software, DESCRIPTIVE statistics, BLAND-White-Garland syndrome, PHYSIOLOGY
Abstract

Background In patients with anomalous left coronary artery from the pulmonary artery (ALCAPA) left ventricular (LV) dilatation and dysfunction evolves due to diminished myocardial perfusion caused by coronary steal phenomenon. Using late gadolinium enhanced cardiovascular magnetic resonance (LGE-CMR) imaging, myocardial scarring has been shown in ALCAPA patients late after repair, however the incidence of scarring before surgery and its impact on postoperative course after surgical repair remained unknown. Methods 8 ALCAPA-patients (mean age 10.0 ± 5.8 months) underwent CMR before and early after (mean 4.9 ± 2.5 months) coronary reimplantation procedures. CMR included functional analysis and LGE for detection of myocardial scars. Results LV dilatation (mean LVEDVI 171 ± 94 ml/m2) and dysfunction (mean LV-EF 22 ± 10 %) was present in all patients and improved significantly after surgery (mean LVEDV 68 ± 42 ml/m2, p = 0.02; mean LV-EF 58 ± 19 %, p < 0.001). Preoperative CMR revealed myocardial scarring in 2 of the 8 patients and did not predict postoperative course. At follow-up CMR, one LGE-positive patient showed delayed recovery of LV function while myocardial scarring was still present in both patients. In two patients new-onset transmural scarring was found, although functional recovery after operation was sufficient. One of them showed a stenosis of the left coronary artery and required resurgery. Conclusions Despite diminished myocardial perfusion and severely compromised LV function, myocardial scarring was preoperatively only infrequently present. Improvement of myocardial function was independent of new-onset scarring while the impact of preoperative scarring still needs to be defined. [ABSTRACT FROM AUTHOR]

Published
2014
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