Liu Xu,Yu Xiafeng,Liu Yiwei,et al.Surgical treatments for sternal defects in children with pentalogy of Cantrell[J].Journal of Clinical Pediatric Surgery,,22():517-524.[doi:10.3760/cma.j.cn101785-202302042-004]
Surgical treatments for sternal defects in children with pentalogy of Cantrell
- Keywords:
- Pentalogy of Cantrell; Sternal Defect; Orthopedic Procedures; Child
- Abstract:
- Objective To optimize the surgical strategies for sternal defect in children with pentalogy of Cantrell (PC) through comparing two types of sternal repair and outcomes.Methods From January 2009 to December 2022, clinical data were retrospectively reviewed for 11 PC children.They underwent stage I surgeries at a median age of 181 days (interquartile range:49 to 343 days) and with an average weight of (7.9±4.9) kg.There were substernal segment defects and congenital heart structural deformities, including complete PC(n=7) and incomplete PC (n=4).Stage I cardiac retraction and sternal reconstruction were performed.According to the mode of repairing sternum, they were assigned into two groups of direct closure (n=6) and non-direct closure (GORE-TEX? patch repair, n=3;artificial sternum, n=1;titanium mesh, n=1).According to the imaging data before stage I cardiac retraction operation, transverse dimension of the thorax on the level of diaphragm at the middle of anterior thoracic wall, the transverse dimension of sternal defect on the same level were measured, and the distance from the lowest margin of residual sternum to the above-mentioned level was measured as longitudinal dimension of sternal defect.We proposed a sternal defect index(SDI) and calculated, that is, the transverse dimension of sternal defect×2/(longitudinal dimension of sternal defect+transverse dimension of thorax×0.2).Results In the whole cohort, 2(2/11) died from cardiac failure post-operation.Nine survivors (9/11) were followed up for 6 to 120 months.One (1/9) child did not undergo complete cardiac surgery while the remainders (8/9) obtained excellent cardiac function, satisfactory thoracic appearance and decent quality-of-life.SDI value of direct closure group was lower than that of non-direct closure group with significant statistic difference (P=0.003);No significant inter-group differences existed in average age, body weight, proportion of complete PC or complex cardiac deformities (P>0.05).Conclusion The prognosis of PC is dependent largely upon the severity of cardiac heterotopia and intracardiac malformation.Different techniques of sternal reconstruction may be guided by SDI.It is an innovative tool for evaluating the severity of sternal defect.For children aged under 1 year with SDI<1, direct sternal closure is recommended;if direct closure is not available, artificial material repair is a practical alternative.
References:
[1] Cantrell JR, Haller JA, Ravitch MM.A syndrome of congenital defects involving the abdominal wall, sternum, diaphragm, pericardium, and heart[J].Surg Gynecol Obstet, 1958, 107(5):602-614.
[2] Kheir AEM, Bakhiet EA, Elhag SMM, et al.Pentalogy of Cantrell:case report and review of the literature[J].Sudan J Paediatr, 2014, 14(1):85-88.
[3] Ergeno?lu MA, Yeniel A?, Peker N, et al.Prenatal diagnosis of Cantrell pentalogy in first trimester screening:case report and review of literature[J].J Turk Ger Gynecol Assoc, 2012, 13(2):145-148.DOI:10.5152/jtgga.2011.77.
[4] Williams AP, Marayati R, Beierle EA.Pentalogy of cantrell[J].Semin Pediatr Surg, 2019, 28(2):106-110.DOI:10.1053/j.sempedsurg.2019.04.006.
[5] Toyama WM.Combined congenital defects of the anterior abdominal wall, sternum, diaphragm, pericardium, and heart:a case report and review of the syndrome[J].Pediatrics, 1972, 50(5):778-792.
[6] Singh N, Bera ML, Sachdev MS, et al.Pentalogy of Cantrell with left ventricular diverticulum:a case report and review of literature[J].Congenit Heart Dis, 2010, 5(5):454-457.DOI:10.1111/j.1747-0803.2009.00375.x.
[7] van Hoorn JHL, Moonen RMJ, Huysentruyt CJR, et al.Pentalogy of Cantrell:two patients and a review to determine prognostic factors for optimal approach[J].Eur J Pediatr, 2008, 167(1):29-35.DOI:10.1007/s00431-007-0578-9.
[8] Mallula KK, Sosnowski C, Awad S.Spectrum of Cantrell’s pentalogy:case series from a single tertiary care center and review of the literature[J].Pediatr Cardiol, 2013, 34(7):1703-1710.DOI:10.1007/s00246-013-0706-4.
[9] Schiavone M, Pizzol D, Colangelo AC, et al.Conservative management of Cantrell syndrome:a case report[J].J Surg Case Rep, 2020, 2020(5):rjaa100.DOI:10.1093/jscr/rjaa100.
[10] 陈昌, 赵枫, 肖明第.Cantrell五联症九例围手术期处理分析[J].中华外科杂志, 2013, 51(4):379-380.DOI:10.3760/cma.j.issn.0529-5815.2013.04.025. Chen C, Zhao F, Xiao MD.Analysis of perioperative management of 9 cases of pentalogy of Cantrell[J].Chin J Surg, 2013, 51(4):379-380.DOI:10.3760/cma.j.issn.0529-5815.2013.04.025.
[11] 孙明利, 吕滨, 荆志成, 等.Cantrell综合征的诊断与治疗[J].中华心血管病杂志, 2011, 39(9):836-839.DOI:10.3760/cma.j.issn.0253-3758.2011.09.013. Sun ML, Lyu B, Jing ZC, et al.Clinic diagnoses and treatments of patients with Cantrell syndrome[J].Chin J Cardiol, 2011, 39(9):836-839.DOI:10.3760/cma.j.issn.0253-3758.2011.09.013.
[12] Sakasai Y, Thang BQ, Kanemoto S, et al.Staged repair of pentalogy of Cantrell with ectopia cordis and ventricular septal defect[J].J Card Surg, 2012, 27(3):390-392.DOI:10.1111/j.1540-8191.2012.01456.x.
[13] Yuan SM, Shinfeld A, Mishaly D.An incomplete pentalogy of Cantrell[J].Chang Gung Med J, 2008, 31(3):309-313.
[14] Abdallah HI, Marks LA, Balsara RK, et al.Staged repair of pentalogy of Cantrell with tetralogy of Fallot[J].Ann Thorac Surg, 1993, 56(4):979-980.DOI:10.1016/0003-4975(93)90373-p.
[15] Zhang XQ, Xing QS, Sun J, et al.Surgical treatment and outcomes of pentalogy of Cantrell in eight patients[J].J Pediatr Surg, 2014, 49(8):1335-1340.DOI:10.1016/j.jpedsurg.2014.06.003.
[16] Usha MK, Mahimaiha J, Shivanna DN.Incomplete pentalogy of Cantrell:single stage repair[J].Heart, 2012, 98(15):1183.DOI:10.1136/heartjnl-2011-301510.
[17] Korver AMH, Haas F, Freund MW, et al.Pentalogy of Cantrell:successful early correction[J].Pediatr Cardiol, 2008, 29(1):146-149.DOI:10.1007/s00246-007-9032-z.
[18] 肖明第, 冯晓东, 张建卿, 等.8例Cantrell五联症的手术治疗[J].中华胸心血管外科杂志, 2010, 26(4):221-223.DOI:10.3760/cma.j.issn.1001-4497.2010.04.002. Xiao MD, Feng XD, Zhang JQ, et al.Summary of surgical experience of eight cases with pentalogy of Cantrell[J].Chin J Thorac Cardiovasc Surg, 2010, 26(4):221-223.DOI:10.3760/cma.j.issn.1001-4497.2010.04.002.
[19] de Campos JRM, Filomeno LTB, Fernandez A, et al.Repair of congenital sternal cleft in infants and adolescents[J].Ann Thorac Surg, 1998, 66(4):1151-1154.DOI:10.1016/s0003-4975(98)00596-7.
[20] Jadhav V, Rao S, D’Cruz A.Autologous repair of isolated complete sternal cleft in an adolescent[J].J Pediatr Surg, 2009, 44(12):2414-2416.DOI:10.1016/j.jpedsurg.2009.09.019.
[21] Araujo Júnior E, Carrilho MC, Toneto BR, et al.Pentalogy of Cantrell:prenatal diagnosis, delivery, and immediate postnatal surgical repair[J].J Neonatal Surg, 2017, 6(2):32.DOI:10.21699/jns.v6i2.503.
[22] Kim CW, Cho HM, Son BS, et al.Neo-sternum reconstruction using costal cartilage approximation and small Permacol? patch repair in the treatment of Cantrell pentalogy:a case report[J].J Cardiothorac Surg, 2015, 10:40.DOI:10.1186/s13019-015-0241-x.
[23] 胡玥, 计晓娟.Cantrell综合征的诊断与治疗[J].国际儿科学杂志, 2020, 47(4):235-238.DOI:10.3760/cma.j.issn.1673-4408.2020.04.004. Hu Y, Ji XJ.Diagnoses and treatments of pentalogy of Cantrell[J].Int J Pediatr, 2020, 47(4):235-238.DOI:10.3760/cma.j.issn.1673-4408.2020.04.004.
[24] Chia HL, Rasheed MZ, Ong KK, et al.Repair of ectopia cordis using a resorbable poly-L-lactic-polyglycolic acid plate in a patient with pentalogy of Cantrell[J].J Pediatr Surg, 2012, 47(6):e1-e4.DOI:10.1016/j.jpedsurg.2012.01.072.
[25] Chaudhry IUH, Cheema A, Aqeel C, et al.Modified surgical reconstruction technique for a rare isolated congenital sternal cleft:In a six-year-old child[J].Ann Med Surg (Lond), 2021, 65:102280.DOI:10.1016/j.amsu.2021.102280.
[26] Razafimanjato NNM, Tsiambanizafy GO, Ravelomihary TDN, et al.Successful surgical repair of a sternum cleft using composite mesh:a case report and new technical note[J].Afr J Thorac Crit Care Med, 2021, 27(2):66-69.DOI:10.7196/AJTCCM.2021.v27i2.103.
[27] Fouilloux V, Bertin F, Peltier E, et al.First sternal cleft repair using a porous alumina ceramic prosthesis in a 9-year-old child[J].European J Pediatr Surg Rep, 2019, 7(1):e20-e23.DOI:10.1055/s-0039-1688775.
[28] 习林云, 李洪波, 吴春, 等.Cantrell五联症的外科治疗及胸骨缺损修复探讨[J].中华小儿外科杂志, 2019, 40(5):432-435.DOI:10.3760/cma.j.issn.0253-3006.2019.05.010. Xi LY, Li HB, Wu C, et al.Surgical repairing of sternal defects with pentalogy of Cantrell[J].Chin J Pediatr Surg, 2019, 40(5):432-435.DOI:10.3760/cma.j.issn.0253-3006.2019.05.010.
[29] 曾春, 金柯含, 李凯, 等.聚醚醚酮与钛网在颅骨修补术中的应用分析[J].中华神经外科杂志, 2021, 37(12):1219-1222.DOI:10.3760/cma.j.cn112050-20210702-00321. Zeng C, Jin KH, Li K, et al.Analysis of application of polyetheretherketone and titanium mesh during cranioplasty[J].Chin J Neurosurg, 2021, 37(12):1219-1222.DOI:10.3760/cma.j.cn112050-20210702-00321.
[30] Chia HL, Yeow VKL.Repair of inferior sternal cleft using bilateral sternal bar turnover flaps in a patient with pentalogy of Cantrell[J].Arch Plast Surg, 2014, 41(1):77-80.DOI:10.5999/aps.2014.41.1.77.
Memo
收稿日期:2023-02-27。
通讯作者:孙彦隽,Email:sunyanjuan@scmc.com.cn