American Journal of Pediatrics

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Cantrell Syndrome – A Rare Complex Congenital Anomaly: A Case Report and Literature Review

Received: Jan. 10, 2024    Accepted: Jan. 29, 2024    Published: Feb. 05, 2024
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Abstract

Cantrell’s pentalogy is a rare and complex white line congenital anomaly, it is first described by Cantrell et al. in 1958. It affects 5.5 children per 1,000,000 live births worldwide. The full spectrum of this syndrome includes five anomalies: an anterior diaphragm defect, a medial supraumbilical abdominal wall defect, a diaphragmatic pericardium defect, a lower sternum defect with intracardiac anomalies. The complete form that associates the five malformations of the Cantrell pentalogy is less common than the incomplete form. The embryogenesis of Cantrell's pentalogy can be caused by a problem in isolation of the frontal mesoderm around 14-18 days of gravidity; The etiopathogenesis of this pattern isn't yet fully understood, numerous factors can be indicted. The prognosis depends on the complexity of the cardiac malformation. We report the case of a 3 month old infant with a complete Cantrell pentalogy and complex congenital hear desease as this is the first case that has been reported in Morocco. And we highlight through our work the importance of prenatal diagnosis for early care. This case highlights the value of gestation monitoring and antenatal comforting and perfecting the quality of complaint operation services during the first days of life.

DOI 10.11648/ajp.20241001.13
Published in American Journal of Pediatrics ( Volume 10, Issue 1, April 2024 )
Page(s) 13-17
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2024. Published by Science Publishing Group

Keywords

Congenital Heart Disease, Thoracoabdominal Wall Defect, Ectopia Cordis, Complete Form, Pentalogy of Cantrell

References
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[2] S. Marcella, P. Damiano, AC Colangelo. Conservative management of Cantrell syndrome: a case report. J Surg Case Rep. 2020, 100: 10.1093/jscr/rjaa100.
[3] AM Korver, F Haas, MW Freund, J Strengers: Pentalogy of Cantrell: successful early correction. Pediatr Cardiol. 2008, 29: 146-149. 10.1007/s00246-007-9032.
[4] RI Kylat: Complete and Incomplete Pentalogy of Cantrell Children. 2019, 6: 109. 10.3390.
[5] A Vŭlkova, K Kovacheva, R Rosmanova: Pentalogy of Cantrell--a case report. Akush Ginekol (Sofiia). 2007, 46: 41-3.
[6] T Mendaluk, A Mościcka: The incomplete pentalogy of Cantrell - A case report. Pediatria Polska. 2015, 90: 241-44.
[7] AR Patil, LS Praveen: V A mbica. Pentalogy of cantrell: A case report. BJR Case Rep. 2015201520140002, 10.1259/bjrcr.20140002.
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[9] Steiner MB, Vengoechea J, Collins RT: II Duplication of the ALDH1A2 gene in association with pentalogy of Cantrell: A case report. J Med Case Rep. 2013, 7: 287.
[10] N Singh, ML Bera: Pentalogy of Cantrell with left ventricular diverticulum: a case report and review of literature. Congenit Heart Dis. 2010, 5: 454-7. 10.1111/j.1747-0803.2009.00375.x.
[11] JH Van Hoorn, RM Moonen: two patients and a review to determine prognostic factors for optimal approach. Eur J Pediatr. 2008, 167: 29-35. 10.1007/s00431-007-0578-9.
[12] Naburi, E Assenga, S Patel et Al: Class II pentalogy of Cantrell. 2015, 8: 318. 10.1186/s13104-015-1293-7.
[13] Ranganath P, Pradhan M: Complete pentalogy of Cantrell with craniorachischisis: A case report. J Prenat Med. 2012, 6: 10-12.
[14] Chen CP: Prenatal diagnosis of pentalogy of Cantrell associated with hypoplasia of the right upper limb andectrodactyly.. Prenat Diagn. 2006, 27: 86-87. 10.1002/pd.1610.
[15] Correa-Rivas MS: Pentalogy of Cantrell: a case report with pathologic findings. Pediatr Dev Pathol. 2004, 7: 649-652. 10.1007/s10024-004-9104-5.
[16] Aslan A, Karaguzel G, Unal I, Aksoy N, Melikoglu M: Two rare cases of the pentalogy of Cantrell or its variants.. Acta Med Austriaca. 2004, 31: 85-87.
[17] Uygur D: An infant with pentalogy of Cantrell and limb defects diagnosed prenatally.. Clin Dysmorphol. 2004, 13: 57-58. 10.1097/00019605-200401000-00018.
[18] Bittmann S: Combined pentalogy of Cantrell with tetralogy of Fallot, gallbladder agenesis, and polysplenia: a case report. J Pediatr Surg. 2004, 39: 107-109. 10.1007/s00431-007-0578-9.
[19] N Alphonso, PS Venugopal: Complete thoracic ectopia cordis. Eur J Cardio-Thorac Surg. 2003, 23: 426-8. 10.1016/s1010-7940(02)00811-4.
[20] Adele P. Williams, M. D: Pentalogy of Cantrell Semin Pediatr Surg. 2019, 28: 106-110. 10.1053.
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[22] Y Ootaki, J Strainic, RM Ungerleider: Pentalogy of Cantrell with double-outlet right ventricle: a case of surgical correction. Cardiol Young. 2011, 21: 235-237. 10.1017/S1047951110001770.
[23] Y Sakasai, BQ Thang: Staged repair of pentalogy of Cantrell with ectopia cordis and ventricular septal defect. J Card Surg. 2012, 27: 390-392. 10.1111/j.1540-8191.2012.01456.x.
[24] Gao Z: Duan QJ et al. Prognosis of pentalogy of Cantrell depends mainly on the severity of the intracardiac anomalies and associated malformations. Eur J Pediatr. 2009, 168: 1413-1414. 10.1007/s00431-008-0905-9.
[25] JY Wan: SH Zhao, SL Jiang Pentalogy of Cantrell associated with a double-outlet right ventricle. Chin Med J. 2012, 125: 3359-3360. 10.3760.
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Cite This Article
  • APA Style

    Ennadif, B., Marzouki, N., Inboui, F. A., Salimi, S., Slaoui, B., et al. (2024). Cantrell Syndrome – A Rare Complex Congenital Anomaly: A Case Report and Literature Review. American Journal of Pediatrics, 10(1), 13-17. https://doi.org/10.11648/ajp.20241001.13

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    ACS Style

    Ennadif, B.; Marzouki, N.; Inboui, F. A.; Salimi, S.; Slaoui, B., et al. Cantrell Syndrome – A Rare Complex Congenital Anomaly: A Case Report and Literature Review. Am. J. Pediatr. 2024, 10(1), 13-17. doi: 10.11648/ajp.20241001.13

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    AMA Style

    Ennadif B, Marzouki N, Inboui FA, Salimi S, Slaoui B, et al. Cantrell Syndrome – A Rare Complex Congenital Anomaly: A Case Report and Literature Review. Am J Pediatr. 2024;10(1):13-17. doi: 10.11648/ajp.20241001.13

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  • @article{10.11648/ajp.20241001.13,
      author = {B. Ennadif and N. Marzouki and F. Alaoui Inboui and S. Salimi and B. Slaoui and Elkorchi and K. Chbani},
      title = {Cantrell Syndrome – A Rare Complex Congenital Anomaly: A Case Report and Literature Review},
      journal = {American Journal of Pediatrics},
      volume = {10},
      number = {1},
      pages = {13-17},
      doi = {10.11648/ajp.20241001.13},
      url = {https://doi.org/10.11648/ajp.20241001.13},
      eprint = {https://download.sciencepg.com/pdf/10.11648.ajp.20241001.13},
      abstract = {Cantrell’s pentalogy is a rare and complex white line congenital anomaly, it is first described by Cantrell et al. in 1958. It affects 5.5 children per 1,000,000 live births worldwide. The full spectrum of this syndrome includes five anomalies: an anterior diaphragm defect, a medial supraumbilical abdominal wall defect, a diaphragmatic pericardium defect, a lower sternum defect with intracardiac anomalies. The complete form that associates the five malformations of the Cantrell pentalogy is less common than the incomplete form. The embryogenesis of Cantrell's pentalogy can be caused by a problem in isolation of the frontal mesoderm around 14-18 days of gravidity; The etiopathogenesis of this pattern isn't yet fully understood, numerous factors can be indicted. The prognosis depends on the complexity of the cardiac malformation. We report the case of a 3 month old infant with a complete Cantrell pentalogy and complex congenital hear desease as this is the first case that has been reported in Morocco. And we highlight through our work the importance of prenatal diagnosis for early care. This case highlights the value of gestation monitoring and antenatal comforting and perfecting the quality of complaint operation services during the first days of life.
    },
     year = {2024}
    }
    

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    T1  - Cantrell Syndrome – A Rare Complex Congenital Anomaly: A Case Report and Literature Review
    AU  - B. Ennadif
    AU  - N. Marzouki
    AU  - F. Alaoui Inboui
    AU  - S. Salimi
    AU  - B. Slaoui
    AU  - Elkorchi
    AU  - K. Chbani
    Y1  - 2024/02/05
    PY  - 2024
    N1  - https://doi.org/10.11648/ajp.20241001.13
    DO  - 10.11648/ajp.20241001.13
    T2  - American Journal of Pediatrics
    JF  - American Journal of Pediatrics
    JO  - American Journal of Pediatrics
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    EP  - 17
    PB  - Science Publishing Group
    SN  - 2472-0909
    UR  - https://doi.org/10.11648/ajp.20241001.13
    AB  - Cantrell’s pentalogy is a rare and complex white line congenital anomaly, it is first described by Cantrell et al. in 1958. It affects 5.5 children per 1,000,000 live births worldwide. The full spectrum of this syndrome includes five anomalies: an anterior diaphragm defect, a medial supraumbilical abdominal wall defect, a diaphragmatic pericardium defect, a lower sternum defect with intracardiac anomalies. The complete form that associates the five malformations of the Cantrell pentalogy is less common than the incomplete form. The embryogenesis of Cantrell's pentalogy can be caused by a problem in isolation of the frontal mesoderm around 14-18 days of gravidity; The etiopathogenesis of this pattern isn't yet fully understood, numerous factors can be indicted. The prognosis depends on the complexity of the cardiac malformation. We report the case of a 3 month old infant with a complete Cantrell pentalogy and complex congenital hear desease as this is the first case that has been reported in Morocco. And we highlight through our work the importance of prenatal diagnosis for early care. This case highlights the value of gestation monitoring and antenatal comforting and perfecting the quality of complaint operation services during the first days of life.
    
    VL  - 10
    IS  - 1
    ER  - 

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Author Information
  • Pediatrics II Department, A. Harouchi Children’s Hospital, IBN ROCHD University Hospital Center, Faculty of Medicine, Hassan II University Casablanca, Casablanca, Morocco

  • Pediatrics II Department, A. Harouchi Children’s Hospital, IBN ROCHD University Hospital Center, Faculty of Medicine, Hassan II University Casablanca, Casablanca, Morocco

  • Pediatrics II Department, A. Harouchi Children’s Hospital, IBN ROCHD University Hospital Center, Faculty of Medicine, Hassan II University Casablanca, Casablanca, Morocco

  • Pediatrics II Department, A. Harouchi Children’s Hospital, IBN ROCHD University Hospital Center, Faculty of Medicine, Hassan II University Casablanca, Casablanca, Morocco

  • Pediatrics II Department, A. Harouchi Children’s Hospital, IBN ROCHD University Hospital Center, Faculty of Medicine, Hassan II University Casablanca, Casablanca, Morocco

  • Radiology Department, A. Harouchi Children’s Hospital, IBN ROCHD University Hospital Center, Faculty of Medicine, Hassan II University Casablanca, Casablanca, Morocco

  • Radiology Department, A. Harouchi Children’s Hospital, IBN ROCHD University Hospital Center, Faculty of Medicine, Hassan II University Casablanca, Casablanca, Morocco

  • Section