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Table of Contents   
ORIGINAL ARTICLE
Year : 2023  |  Volume : 28  |  Issue : 6  |  Page : 508-513
 

Thoracoscopic resection of pulmonary lesions in Israel: The mentorship approach


1 Department of Pediatric Surgery, Meir Medical Center, Affiliated to Sackler School of Medicine, Tel Aviv University, Kfar Saba, Israel
2 Division of Pediatric Surgery, The Rocky Mountain Hospital for Children, Denver, Colorado, USA
3 Institute of Pulmonology, Schneider Children's Medical Center of Israel (Affiliated to Sackler School of Medicine, Tel Aviv University), Petah-Tiqwa, Israel
4 Department of Anesthesiology, Schneider Children's Medical Center of Israel, Petah-Tiqwa, Israel
5 Department of Pediatric and Adolescent Surgery, Schneider Children's Medical Center of Israel (Affiliated to Sackler School of Medicine, Tel Aviv University), Petah-Tiqwa, Israel
6 Department of Thoracic Surgery, Beilinson Hospital (Affiliated to Sackler School of Medicine, Tel Aviv University), Petah-Tiqwa, Israel

Date of Submission21-May-2023
Date of Decision13-Jul-2023
Date of Acceptance30-Jul-2023
Date of Web Publication02-Nov-2023

Correspondence Address:
Emmanuelle Seguier-Lipszyc
Department of Pediatric Surgery, Meir Medical Center, Affiliated to Sackler School of Medicine, Tel Aviv University, 59, Tchernihovski, Kfar Saba
Israel
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.jiaps_115_23

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   Abstract 


Background: Most congenital pulmonary airway malformations (CPAMs) are detected antenatally. The majority of newborns are asymptomatic. Patients are prone to subsequent respiratory complications and to a lesser extent malignant transformation remains concerning. In Israel, until 2013, pediatric surgeries were performed by thoracotomy. To minimize its morbidity, we introduced thoracoscopy using a mentorship approach. We present our experience with thoracoscopic resections coordinated by the mentorship of a pediatric worldwide leader in his field and compare our results with resections performed by thoracotomy.
Materials and Methods: A retrospective review of records of children operated between 2013 and 2020 was conducted. Data were compared using t-test for quantitative variables.
Results: Fifty patients were operated by thoracoscopy with a median age of 4 years, a thoracoscopic lobectomy performed in 68%. There was no conversion with a median length of stay (LOS) of 3½ days. Thirty patients were operated by thoracotomy by a thoracic surgeon with a median age of 3.5 years. A lobectomy was performed in 87% with a median LOS of 7 days.
Conclusions: Thoracoscopic lobectomy is a technically demanding procedure with a long learning curve, strongly related to the low volume of cases. The role of a mentorship program in acquiring those surgical skills is crucial through standardization of the technique applied and supervised by the mentor. Early thoracoscopy for congenital pulmonary lesions at an early age can be achieved with a low conversion rate and minimal complications creating a change in the paradigm of practice when considering surgery for CPAM in Israel.


Keywords: Bronchopulmonary sequestration, congenital pulmonary airway malformation, lobectomy, thoracoscopy


How to cite this article:
Seguier-Lipszyc E, Rothenberg S, Mei-Zahav M, Stafler P, Zeitlin Y, Samuk I, Peysakhovich Y, Kravarusic D. Thoracoscopic resection of pulmonary lesions in Israel: The mentorship approach. J Indian Assoc Pediatr Surg 2023;28:508-13

How to cite this URL:
Seguier-Lipszyc E, Rothenberg S, Mei-Zahav M, Stafler P, Zeitlin Y, Samuk I, Peysakhovich Y, Kravarusic D. Thoracoscopic resection of pulmonary lesions in Israel: The mentorship approach. J Indian Assoc Pediatr Surg [serial online] 2023 [cited 2023 Nov 28];28:508-13. Available from: https://www.jiaps.com/text.asp?2023/28/6/508/389313





   Introduction Top


Congenital pulmonary airway malformations (CPAMs) are a group of mixed entities, including congenital cystic adenomatous malformations (CCAMs), bronchopulmonary sequestration (BPS), congenital lobar emphysema (CLE), and bronchogenic cysts (BCs). With the improving accuracy of prenatal ultrasound, they are increasingly detected antenatally. About 10% of all lesions will cause symptoms during the neonatal period and thus lead to surgical resection.[1] The management of asymptomatic cases remains controversial with families and physicians having to balance the risks of resection with the risks of conservative follow-up. Early surgery prevents the development of respiratory complications and minimizes the risk of missing malignant lesions at diagnosis or malignant transformation.

In the last decade, in certain specialized centers, thoracoscopic resection of these lesions is described as common practice, but the learning curve is quite long since, technically these remain demanding procedures and the worldwide volume of patients is limited.[2] To lessen the complication and conversion rate and shorten the learning curve, a mentorship approach has been applied in our institution, allowing for the gradual introduction of surgeons to this technique.


   Materials and Methods Top


We retrospectively reviewed the medical records of all patients who underwent resection of a congenital lung lesion at Schneider Children's Medical Center of Israel (SCMCI), a tertiary pediatric hospital, between December 2013 and October 2020, both through thoracoscopy and thoracotomy. Patients were evaluated for indication of surgery, type of surgery performed, age at surgery, length of stay (LOS), complications, pathological examination, and follow-up. Statistical analysis was performed using SAS/STAT software version 9.4 by Statistical Analysis System Corporation, North Carolina, United States of America. Data were compared using t-test to compare quantitative variables. P < 0.05 was considered statistically significant.

Pediatric thoracoscopic lobectomy is a very technically demanding procedure, and a mentorship approach was adopted in our institution since 2013. The mentor, Dr. Steven Rothenberg, came once to twice a year, and surgeries were concentrated on 2–3 days.

The study was approved by the Institutional Review Board of SCMCI and Rabin Medical Center (0671-18-RMC).


   Results Top


The summarized results of the two groups of patients that underwent surgery by thoracoscopic versus thoracotomy approach are presented in [Table 1].
Table 1: Data on thoracoscopic surgeries versus thoracotomy

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During the 7-year period, 50 patients underwent resection through thoracoscopy and 30 through thoracotomy, the median age (range) was 4 years (5 months to 17.8 years) and 3.5 years (13 days to 17.3 years), respectively. All thoracoscopic procedures were completed by or under the guidance/supervision of a single mentor surgeon, Dr. Steven Rothenberg (The Rocky Mountain Hospital for Children, Denver, Colorado, USA), with a zero conversion rate. The first 3 years, Dr. Rothenberg did all the thoracoscopic lobectomies himself and described step by step the procedure and its eventual pitfalls. The following 3 years, he helped the local assistant on parts of the procedure to finally assisting only in 2020. The modality of choice for diagnosis before surgery was computed tomography (CT) scan in all but one patient who underwent a magnetic resonance imaging (MRI) (performed at age 1 month to 2.8 years with a median of 10 months). Chest tubes were left in place from 1 to 15 days after surgery in 44 cases (all lobectomies and segmentectomies) with a mean duration of 3 days. Early postoperative complication rate was 7% and consisted of 4 pneumothoraces after drain removal that required insertion of a new thoracic drain for a period of 2–10 additional days. The follow-up ranges from 6 months to 8 years and is uneventful. All thoracotomy procedures were done by a thoracic surgeon. The modality of choice for diagnosis before surgery was a CT scan in all. Chest tubes were left in place from 1 to 14 days after surgery in all cases, with a mean duration of 4 days. Early postoperative complication rate was 13% and consisted of three pneumothoraces after drain removal and one bronchopulmonary fistula requiring a redo-thoracotomy 14 days postoperatively for closure of the fistula. The follow-up ranges from 1.6 to 7 years, two patients presented with pneumonia on the side operated on 1 month after surgery.

Using a t-test two-sample assuming unequal variances, variables of age at surgery, time of surgery, and antenatal diagnosis are without statistical difference. Variables of LOS (3 vs. 7 days, P = 0.006) and of time of thoracic drain (3 vs. 4 days, P = 0.04) are statistically significant with a shorter time for thoracoscopic surgery compared to thoracotomy. Early postoperative complications (7% vs. 13%, P = 0.0017) are statistically significant, with less complications for thoracoscopic surgery.


   Discussion Top


Congenital lung malformations are uncommon disorders and include CPAMs (previously termed CCAM), BPS, CLE, and BCs. Previously, these malformations had a reported incidence of 1/10,000–35,000 pregnancies, but with the improvement of prenatal imaging (ultrasound and MRI), an incidence of fetal lung masses was recalculated and reported as 1/2500 in 2015.[3] A prenatal resolution of CPAMs is found in 19%–56% of pregnancies continued to term.[4] However, up to 40% of lesions with apparent prenatal resolution on ultrasound still persist on postnatal imaging.[5] For symptomatic patients, which represent 20%–40% of infants,[6] surgical resection is the standard of care. Management of asymptomatic patients remains controversial. Despite the lack of symptoms, these patients are susceptible to pulmonary infections, with half occurring in the first year of life.[7] Morikawa et al.[8] describe in these children recurrent pneumonia by the age of 3 years. In a study of older patients from the Mayo Clinic, 82.3% of patients required resection for symptoms, suggestive of recurrent pneumonia and 17.7% for suspected malignancy.[9] A nationwide Japanese study reported that all asymptomatic cases beyond the newborn period became symptomatic by 10 years of age.[10] Another study showed that more than 25% of asymptomatic cases presented pathology with either subclinical infection or malignancy.[6] When symptoms do develop, they can be very severe including life-threatening sepsis. The likelihood of patients becoming symptomatic after the neonatal period varies from 3% to 86%.[11] Conservative management may be a reasonable option for selected cases, as demonstrated by the GOSH team.[12] However, the need for serial CT scans and a strict follow-up which is often difficult to ensure, contravenes this management strategy. Faced with the debate of “take it out or just do naught,” a less invasive treatment modality might sway physicians and families toward removal.

Elective planned surgery is safer than emergency surgery, especially in neonates and infants who present a three-time increased risk of adverse events after emergency surgery.[13] The primary risk of conversion from thoracoscopy to open thoracotomy is the presence of preoperative infections that create adhesions and inflammation, leading to a more difficult dissection, rate of conversion ranges from 1% to 50%.[14] Surgical complications in patients operated on when symptomatic consist of longer LOS, longer pleural drainage, higher rate of fistula, bleeding, or need for second surgery. Furthermore, operating early might avoid the inflammatory changes associated with clinically and subclinically infections.[14] Moreover, if left untreated for several decades, the lesions may undergo malignant transformation. Moreover, a histological overlap between type I pleuroblastoma (PPB) and type IV CPAM exists.[15]

The “wait and see” approach requires serial investigations with serial yearly CT scans that pose the problem of cumulated dose of ionizing radiation[16] or MRIs that require general anesthesia. Early resection also comes with the potential for compensatory lung growth. A study using radionuclide imaging for measuring long-term pulmonary function and postnatal lung growth after lobectomy showed that alveolar multiplication mostly occurs in patients younger than 1 year, whereas if the surgery is performed after, emphysematous overinflation occurs.[17] A long-term lung function study in children following lobectomy for congenital lung malformations suggests surgery before 1 year of age for asymptomatic patients to enable an adequate long-term pulmonary function.[18] Long-term pulmonary function test is superior in patients who underwent thoracoscopic lobectomy compared to thoracotomy.[19]

A long-term follow-up study from Australia showed a high incidence (86%) of complications in initially asymptomatic cases at a median age of 2 years for symptom development.[20] A recent meta-analysis showed that even if the child remains asymptomatic beyond the neonatal period, resection is still recommended to avoid the emergence of symptoms.[21] Another recent systematic review found that at least 25% of asymptomatic CPAM patients will develop symptoms and these tend to develop around 6–7 months of age.[6] In our series, four patients were operated before 6 months of age and 14 between 6 months and 1 year.

Operative time increases significantly with age (before and after 6 months) without significant differences in major complications, readmission or conversion to open.[22] This is also supported by our results with a lower complication rate in our patients who were operated before 6 months of age. The suggested reason is anatomic factors rendering the surgery more difficult, like more complete fissures with age and infections. Meta-analyses have demonstrated shorter hospital LOS and comparable operative times of thoracoscopy compared to thoracotomy.[13] This is also supported by the current results with a shorter hospital stay in the thoracoscopic group, which was statistically significant.

Thus, many advocate early resection of asymptomatic CPAM before the development of symptoms and to enable lung growth compensation.[6],[14],[22],[23],[24]

For those who argue for conservative nonoperative management of these lesions in asymptomatic patients, despite the high incidence of infection, two cases of unsuspected PPB were reported by Dr. Rothenberg in his series in 2015 and 3 PPB were found in 129 suspected cases of CPAM in the Ontario series in 2010 and more recently in 2020 were reported two malignant lesions (one PPB and one bronchioalveolar carcinoma) out of 63 congenital lung malformations. The Oxford experience in 2017[24] presents also in asymptomatic patients evidence of microscopic disease, including infection and two cases of tumors (PPB and rhabdomyomatous dysplasia seen in CPAM). A systematic review concluded that in CPAM, lesions detected radiographically might exist a 4% risk of PPB and an undefined risk for late malignant transformation into other epithelial and mesenchymal malignancies.[11]

Thus, the risks of recurrent infection and possible malignancy outweigh the risks of intervention if a thoracoscopic approach is used in an institution with experience in these procedures.[14]

Thoracoscopic lobectomy is one of the most technically demanding minimally invasive procedures performed in the pediatric population. The complex nature of the disease process and surgical dissection, the risk of bleeding, and the anesthetic issues have limited the number of procedures being attempted. A learning curve for pediatric thoracoscopic lobectomy is necessary and to acquire expertise a mentorship is a strong advantage. It is already in place at Columbia University[14] and in Spain.[25] Since 2013, Dr. Rothenberg has arrived at our institution once to twice a year to perform and assist with these thoracoscopic surgeries and gradually teaches the techniques to local surgeons. It has enabled us to offer these advanced procedures immediately, without any major operative complication, with a zero conversion rate, eliminating the disadvantage to the patient and to the team during the learning curve period. Pulmonologists refer more easily patients knowing the procedure will be performed thoracoscopically. This represents a great example of the valuable art of mentorship,[26] the most significant benefit being for the patients now and in future.

The outcomes of our cooperation are advanced skills and confidence of the local team, and in November 2020, a thoracoscopic right lobar lobectomy in a 1-year-old boy, and in December 2020, a thoracoscopic right upper lobectomy in a 6-month-old girl was successfully performed by our local surgeons without conversion or complications.

These procedures are technically easier in infants at or near 5 kg despite the smaller working space.[14] And lately, with the development of a 5 mm stapler and a 3 mm sealer, surgery in small weight infants has become easier and safer.[27]

Single lung ventilation is helpful in most cases and mandatory for some operations. In patients who are too young or too small for a double-lumen endotracheal tube, single lung ventilation can be achieved with contralateral mainstem intubation or insertion of a bronchial blocker.

Another advantage of the thoracoscopic approach for CPAM, which has been shown to be safe and cost-effective, is the lower incidence of postoperative musculoskeletal complications.[1],[14] Early resection before the development of respiratory complications such as pneumonia may also avoid the risk of conversion owing to adhesions and poor visibility.[14],[28]

With the COVID-19 pandemic and its travel restrictions, our mentor was unable to come, so we started to do the procedures by ourselves with the confidence built up through the years. Telementoring helped us prepare for the surgeries ahead and contributed to the success.


   Conclusion Top


When summarizing our developing experience with mentorship in a continuing medical education design, we are confident to conclude that the paradigm of surgical practice for CPAM in Israel has changed. The primary thoracoscopic approach with the standardization of the technique for selected CPAM and various lung lesions is safe, with rapid recovery, superior cosmetic results, and satisfactory outcomes.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Adams S, Jobson M, Sangnawakij P, Heetun A, Thaventhiran A, Johal N, et al. Does thoracoscopy have advantages over open surgery for asymptomatic congenital lung malformations? An analysis of 1626 resections. J Pediatr Surg 2017;52:247-51.  Back to cited text no. 1
    
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Criss CN, Musili N, Matusko N, Baker S, Geiger JD, Kunisaki SM. Asymptomatic congenital lung malformations: Is nonoperative management a viable alternative? J Pediatr Surg 2018;53:1092-7.  Back to cited text no. 3
    
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Kantor N, Wayne C, Nasr A. Symptom development in originally asymptomatic CPAM diagnosed prenatally: A systematic review. Pediatr Surg Int 2018;34:613-20.  Back to cited text no. 5
    
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Durell J, Thakkar H, Gould S, Fowler D, Lakhoo K. Pathology of asymptomatic, prenatally diagnosed cystic lung malformations. J Pediatr Surg 2016;51:231-5.  Back to cited text no. 6
    
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Parikh DH, Rasiah SV. Congenital lung lesions: Postnatal management and outcome. Semin Pediatr Surg 2015;24:160-7.  Back to cited text no. 7
    
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Morikawa N, Kuroda T, Honna T, Kitano Y, Fuchimoto Y, Terawaki K, et al. Congenital bronchial atresia in infants and children. J Pediatr Surg 2005;40:1822-6.  Back to cited text no. 8
    
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Makhija Z, Moir CR, Allen MS, Cassivi SD, Deschamps C, Nichols FC 3rd, et al. Surgical management of congenital cystic lung malformations in older patients. Ann Thorac Surg 2011;91:1568-73.  Back to cited text no. 9
    
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Kuroda T, Nishijima E, Maeda K, Fuchimoto Y, Hirobe S, Tazuke Y, et al. Clinical features of congenital cystic lung diseases: A report on a nationwide multicenter study in Japan. Eur J Pediatr Surg 2016;26:91-5.  Back to cited text no. 10
    
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Downard CD, Calkins CM, Williams RF, Renaud EJ, Jancelewicz T, Grabowski J, et al. Treatment of congenital pulmonary airway malformations: A systematic review from the APSA outcomes and evidence based practice committee. Pediatr Surg Int 2017;33:939-53.  Back to cited text no. 11
    
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Cook J, Chitty LS, De Coppi P, Ashworth M, Wallis C. The natural history of prenatally diagnosed congenital cystic lung lesions: Long-term follow-up of 119 cases. Arch Dis Child 2017;102:798-803.  Back to cited text no. 12
    
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Stanton M, Njere I, Ade-Ajayi N, Patel S, Davenport M. Systematic review and meta-analysis of the postnatal management of congenital cystic lung lesions. J Pediatr Surg 2009;44:1027-33.  Back to cited text no. 13
    
14.
Rothenberg SS, Middlesworth W, Kadennhe-Chiweshe A, Aspelund G, Kuenzler K, Cowles R, et al. Two decades of experience with thoracoscopic lobectomy in infants and children: Standardizing techniques for advanced thoracoscopic surgery. J Laparoendosc Adv Surg Tech A 2015;25:423-8.  Back to cited text no. 14
    
15.
Priest JR, Williams GM, Hill DA, Dehner LP, Jaffé A. Pulmonary cysts in early childhood and the risk of malignancy. Pediatr Pulmonol 2009;44:14-30.  Back to cited text no. 15
    
16.
Davenport M, Eber E. Long term respiratory outcomes of congenital thoracic malformations. Semin Fetal Neonatal Med 2012;17:99-104.  Back to cited text no. 16
    
17.
Komori K, Kamagata S, Hirobe S, Toma M, Okumura K, Muto M, et al. Radionuclide imaging study of long-term pulmonary function after lobectomy in children with congenital cystic lung disease. J Pediatr Surg 2009;44:2096-100.  Back to cited text no. 17
    
18.
Tocchioni F, Lombardi E, Ghionzoli M, Ciardini E, Noccioli B, Messineo A. Long-term lung function in children following lobectomy for congenital lung malformation. J Pediatr Surg 2017;52:1891-7.  Back to cited text no. 18
    
19.
Lau CT, Wong KK. Long-term pulmonary function after lobectomy for congenital pulmonary airway malformation: Is thoracoscopic approach really better than open? J Pediatr Surg 2018;53:2383-5.  Back to cited text no. 19
    
20.
Wong A, Vieten D, Singh S, Harvey JG, Holland AJ. Long-term outcome of asymptomatic patients with congenital cystic adenomatoid malformation. Pediatr Surg Int 2009;25:479-85.  Back to cited text no. 20
    
21.
Kapralik J, Wayne C, Chan E, Nasr A. Surgical versus conservative management of congenital pulmonary airway malformation in children: A systematic review and meta-analysis. J Pediatr Surg 2016;51:508-12.  Back to cited text no. 21
    
22.
Jelin EB, O'Hare EM, Jancelewicz T, Nasr I, Boss E, Rhee DS. Optimal timing for elective resection of asymptomatic congenital pulmonary airway malformations. J Pediatr Surg 2018;53:1001-5.  Back to cited text no. 22
    
23.
Singh R, Davenport M. The argument for operative approach to asymptomatic lung lesions. Semin Pediatr Surg 2015;24:187-95.  Back to cited text no. 23
    
24.
Thakkar HS, Durell J, Chakraborty S, Tingle BL, Choi A, Fowler DJ, et al. Antenatally detected congenital pulmonary airway malformations: The Oxford experience. Eur J Pediatr Surg 2017;27:324-9.9.  Back to cited text no. 24
    
25.
Cano I, Antón-Pacheco JL, García A, Rothenberg S. Video-assisted thoracoscopic lobectomy in infants. Eur J Cardiothorac Surg 2006;29:997-1000.  Back to cited text no. 25
    
26.
Entezami P, Franzblau LE, Chung KC. Mentorship in surgical training: A systematic review. Hand (N Y) 2012;7:30-6.  Back to cited text no. 26
    
27.
Rothenberg S. Thoracoscopic lobectomy in infants and children utilizing a 5 mm stapling device. J Laparoendosc Adv Surg Tech A 2016;26:1036-8.  Back to cited text no. 27
    
28.
Elhattab A, Elsaied A, Wafa T, Jugie M, Delacourt C, Sarnacki S, et al. Thoracoscopic surgery for congenital lung malformations: Does previous infection really matter? J Pediatr Surg 2021;56:1982-7.  Back to cited text no. 28
    



 
 
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