Current Practice in the Management of Spontaneous Pneumothorax in Children

Abstract

Introduction:

There is a lack of pediatric-specific guidelines for the workup and management of primary spontaneous pneumothorax (PSP) in children. The aim of this study was to describe current practices among North American pediatric surgeons.

Materials and Methods:

An online survey comprising 18 questions was sent out through the American Pediatric Surgical Association Outcomes and Clinical Trials Committee to all members. Bivariate analysis was performed using Chi-square analysis.

Results:

A total of 287 surveys were completed (33% response rate). For a first episode of PSP, 57% of surgeons opt for chest tube drainage, 4% for upfront video-assisted thoracoscopic surgery (VATS), 3% for needle aspiration, and 29% for only oxygen administration. Eighty-one percent of surgeons report that the size of the pneumothorax influences management. However, neither practice setting (P = .87) nor years in practice (P = .11) correlated with initial management strategy. For patients with a persistent air leak after chest tube placement, there is wide variation in duration of observation before performing VATS, with 40% operating after 3 days, but 21% waiting at least 5 days. The use of chest computed tomography (CT) is also highly variable. Eighty-two percent of respondents perform surgery only after the second episode of PSP. Most perform a stapled apical blebectomy and mechanical pleurodesis for both initial and recurrent PSP.

Conclusion:

There is significant variation among pediatric surgeons in the management of spontaneous pneumothorax, including the use of CT, timing of operation, and duration of observation for air leak before performing surgery. Prospective data are needed to better inform guidelines and standardize practice.

Introduction

Spontaneous pneumothorax affects in excess of 20,000 patients per year in the United States and is associated with health care expenditure exceeding $130,000,000.^1,2 Because this condition primarily affects adolescents and young adults, pediatric general and thoracic surgeons care for a significant fraction of affected patients. The highest incidence of primary spontaneous pneumothorax (PSP) in the pediatric population is in tall, thin adolescent males, aged 13–16 years.^3,4 Spontaneous pneumothorax is considered primary if there is no underlying predisposing condition such as pulmonary pathology, connective tissue disease, or infection.⁴ The pathophysiology of PSP is believed to involve rupture of blebs or bullae. While chest radiographs or computed tomography (CT) scans may visualize these blebs, and the presence of blebs has been associated with a need for surgical intervention, the sensitivity and specificity of these imaging findings for predicting clinical outcome remain poor.^5–7 Treatment is therefore driven largely by clinical factors, including patient and provider preferences.

Often initial treatment of symptomatic pneumothorax involves drainage of the extrapulmonary air by needle aspiration or tube thoracostomy. However, recurrence rates after this nonoperative approach are high, ranging from 22% to 55%.^3,8–11 Surgical management is usually apical wedge resection of blebs/bullae and pleurodesis or pleurectomy. This operation is now typically performed using a minimally invasive approach with video-assisted thoracoscopic surgery (VATS). VATS has been shown to be safe, effective, and associated with low recurrence rates (10%–13%), decreased hospital stay, and decreased postoperative pain and morbidity compared to traditional open thoracotomy.^3,10–14

Although high rates of recurrence with nonoperative management have been consistently reported across studies, significant morbidity and recurrence have also been reported after operative intervention.^3,7–11 Factors indicative of treatment failure with nonoperative management are also debated. Persistent air leakage from the ruptured blebs, failure of adequate lung expansion on chest radiograph, and recurrence of pneumothorax after tube removal indicate failure of nonoperative management, which may necessitate an operation.¹⁵ However, the reported definition of a persistent air leak varies in reported studies from 3 to 10 days.^3,16,17 A prolonged air leak can significantly increase the preoperative duration of hospitalization for those who will invariably require surgery.^3,15–17

Both the American College of Chest Physicians (ACCP) and the British Thoracic Society (BTS) have published practice guidelines for the management of spontaneous pneumothorax.^15,18 The ACCP recommends observation in the emergency department for clinically stable patients with small pneumothoraces, whereas for those with large pneumothoraces, the recommendation is for chest tube or pigtail catheter placement and hospitalization.¹⁵ Furthermore, the ACCP panel recommends surgical evaluation for patients with air leak persisting beyond 4 days and intervention to prevent recurrence in those with a second pneumothorax occurrence.¹⁵ The BTS guidelines emphasize that “the size of a pneumothorax is less important than the degree of clinical compromise” and also recommend observation for small and even large pneumothoraces without significant dyspnea.¹⁸ Needle aspiration is recommended as an initial strategy for those requiring intervention and early surgical referral for those who have had a chest tube with a persistent air leak for 3–5 days.¹⁸ However, these are expert consensus guidelines for which there is limited level 1 evidence and are intended for the management of adult patients.

In this study, members of the American Pediatric Surgical Association (APSA) were surveyed to establish current practice in the workup and management of PSP.

Materials and Methods

Study population

An electronic survey was used to investigate the initial management of PSP, imaging preferences, and operative approaches to both initial and recurrent PSP among pediatric surgeons in North America (Supplementary Data 1). Study data were collected and managed using Research Electronic Data Capture (REDCap^®) tools hosted at Northwestern University.¹⁹ The survey was self-administered through REDCap with dissemination of a hyperlink using the APSA email server. The survey comprised 18 questions focused on initial management of PSP, imaging preferences, operative techniques, and approach to recurrent disease. The survey was distributed by the APSA Outcomes and Evidence Based Practice Committee to 870 associate and full members. After initial survey distribution, reminder emails were sent 12 and 21 days later. Institutional Review Board (IRB) approval was obtained, and the study was deemed exempt (IRB No. 2017-1157).

Statistical analysis

Data were analyzed using STATA, version 13 (College Station, TX). Descriptive analysis was performed on the answers to each survey question, with reporting of frequencies and percentages. Differences in preferences were explored using Chi-square or Fisher's exact tests for categorical variables. Associations of preferences with experience or institutional type were tested using multivariable linear regression. A P value <.05 was considered statistically significant.

Results

Demographics

There were 287 responses to the survey, for a response rate of 33%. Most responses were from pediatric surgeons working either in a freestanding children's hospital (55%) or a pediatric hospital within a general adult hospital (41%). Most responders had been in practice for at least 11 years (58%) with 18% in practice for 5 years or less.

Initial management

Tube thoracostomy was the preferred initial management by a majority (57%) of respondents for the first episode of PSP, with only 4% recommending VATS as the first intervention. Initial management was not significantly associated with either practice setting (P = .923) or years in practice (P = .635). Persistent air leak (94%) and recurrence after chest tube removal (78%) were the most common factors prompting VATS during the initial admission. Most surgeons (70%) would wait at least 3 days after chest tube placement for resolution of air leak before performing VATS, including 20% who would wait 5 days or more (Table 1).

Table 1.

Initial Management of Primary Spontaneous Pneumothorax Among North American Pediatric Surgeons

	%
Initial management
Oxygen administration	29
Needle aspiration	3
Tube thoracostomy (chest tube)	57
Upfront VATS	4
Other	7
Factors prompting VATS during initial admission
Persistent air leak	94
Failure of complete lung expansion	57
Recurrence after chest tube removal	78
Other	3
Observation of air leak before performing VATS, days
1	2
2	27
3	39
4	11
5 or more	20
Influence of size of pneumothorax on management
Strong	43
Moderate	38
None	19
Timing of surgery
First episode	14
Second episode	82
Third or more episodes	4
Chest CT for operative decision-making
Routinely	26
Selectively	35
Rarely/never	39
Surgery offered based on blebs on CT
Yes	42
No	35
N/A	23
Surgery offered on asymptomatic side based on presence of blebs
Yes	27
No	58
N/A	15

CT, computed tomography; N/A, not applicable; VATS, video-assisted thoracoscopic surgery.

Imaging

The size of the pneumothorax on initial imaging influenced their choice of initial management in the majority of responses. Chest CT was utilized rarely or never in the initial workup of PSP, with only 26% obtaining a CT routinely. When CT revealed blebs/bullae on the contralateral (asymptomatic) side, most (58%) surgeons would not recommend bilateral intervention.

Timing of intervention

One quarter of surgeons with <5 years of experience offered VATS after the first episode of PSP compared to only 7% of those in practice for >20 years (P = .04). However, nearly 80% of respondents perform surgery after the second episode of PSP (first recurrence).

Operative management

VATS was the favored operative approach for both initial and recurrent PSP, and most surgeons perform a stapled apical blebectomy and mechanical pleurodesis. Only a minority of respondents (9%) use staple line reinforcement and in those who perform chemical pleurodesis, talc was the preferred agent. When a second operation is required for recurrent PSP, more respondents chose to use chemical pleurodesis, pleurectomy, fibrin sealant, and staple line reinforcement (Table 2).

Table 2.

Operative Management of Initial and Recurrent Primary Spontaneous Pneumothorax Among North American Pediatric Surgeons

	Initial PSP, %	Recurrent PSP, %
Operative approach
VATS	98	96
Thoracotomy	1	2
Other	1	2
Interventions
Stapled apical blebectomy	97	84
Mechanical pleurodesis	77	68
Chemical pleurodesis	16	37
Pleurectomy	25	40
Fibrin sealant	11	21
Other	2	2
Staple line reinforcement
Yes	9	13
No	91	87
Agent for chemical pleurodesis
Talc	70	60
Minocycline	0	2
Doxycycline	30	38
Other	0	0

PSP, primary spontaneous pneumothorax.

Discussion

This survey of North American pediatric surgeons demonstrates that considerable variability exists in the management of pediatric PSP. The majority of surgeons manage a first episode of PSP nonoperatively with chest tube drainage and use VATS for ongoing air leak for 3 or more days or for recurrent pneumothorax. Both patient and provider factors influence clinical decision-making.

Tube thoracostomy was the initial management strategy for PSP used by a majority of respondents in this survey, in agreement with findings from both a national administrative database and from single institution retrospective reviews of pediatric patients.^19,20 The choice of initial approach, according to both the BTS and ACCP adult guidelines, should be made based on patient symptomatology and the degree of lung collapse on imaging.^15,18 Patients who are clinically unstable or those with a large pneumothorax should undergo hospitalization with needle aspiration or tube thoracostomy, while those who are stable with a small pneumothorax may be candidates for no intervention after a brief period of observation and short-interval follow-up.^15,18 This initial nonoperative approach in children however, compared to early VATS, has been associated with a higher risk of recurrence and an increasing probability of requiring surgery the longer patients remain hospitalized.^10,11,19 Furthermore, most pediatric patients with PSP ultimately require VATS, even after initial nonoperative management.²⁰ One study suggested that <40% of pediatric patients with PSP were definitively treated without surgery.³ Despite showing a high risk of recurrence and substantial likelihood of ultimately requiring VATS, there is insufficient current evidence to definitively recommend upfront surgery and most pediatric surgeons still manage a first episode of PSP nonoperatively.

Needle aspiration is an alternative to tube thoracostomy for the initial nonoperative management of PSP. According to the most recent BTS guidelines, needle aspiration is as effective as tube thoracostomy for management of symptomatic PSP and may be associated with reduced hospitalization and length of stay.¹⁸ The ACCP guidelines, however, do not include needle aspiration in the management algorithm, and it was chosen as initial management by only a minority of respondents to our survey. Furthermore, in one retrospective review of PSP management at two pediatric U.K. hospitals, 53% of needle aspirations failed, suggesting that in the pediatric population this technique may not be as effective.²¹ In the pediatric patient population where sedation may be required for needle aspiration or chest tube placement, providers may opt for a more definitive modality of chest drainage to minimize the risk of requiring repeat procedures and anesthesia.

For most respondents, the size of the pneumothorax on imaging influenced their choice of initial management. It has been shown that the size of the pneumothorax on imaging does not correlate well with clinical symptoms but may determine the rate of resolution.¹⁸ There are also different methods of size estimation among the guidelines from professional societies.²² For instance, the ACCP uses the apex-to-cupola distance and the BTS uses the interpleural distance at the level of the hilum. Further efforts are required in the pediatric population to understand whether imaging findings can predict which patients will improve rapidly without intervention and which will require pneumothorax drainage.

Only one quarter of respondents to this survey routinely obtained CT in the workup of PSP. Similarly, in a review of a large national database, less than a third of pediatric patients with PSP underwent CT imaging.¹⁹ CT can theoretically identify the presence or absence of pulmonary blebs or bullae for which operative intervention may reduce the risk of treatment failure and relapse.^23,24 However other groups have shown that CT has low sensitivity for the detection of blebs and therefore limited utility in the management of pediatric patients with PSP.⁶ The infrequent utilization of CT may also relate to the recommendation against contralateral surgery by a majority of surgeons, even when contralateral blebs are seen. Assessment of the contralateral side is one of the potential benefits of preoperative imaging, but the responses suggest that most surgeons believe that the risks of contralateral surgery outweigh the benefits. In the adult guidelines, the BTS recommends CT for “uncertain or complex cases,” and the ACCP panel does not recommend routine use for patients with a first-time pneumothorax.¹⁵

Because of the limitations of current imaging to predict the likelihood of success or failure with nonoperative management, surgeons rely on clinical findings, including persistent air leak, incomplete lung re-expansion, and pneumothorax recurrence to drive management. While 94% of respondents indicated that persistent air leak was a factor that would prompt them to offer VATS during the initial admission, there was considerable variability in the number of days they would wait before recommending that operation. More than 70% of responding surgeons would wait 3 or more days before recommending surgery. Similarly, the newest BTS guidelines recommend that an air leak persisting beyond 3–5 days should prompt referral to a thoracic surgeon.¹⁸ The ACCP panel recommends observation for 4 days before surgical evaluation.¹⁵ Other authors have suggested that early objective measurement of air leak volume may provide a form of risk assessment to determine which patients would benefit from early intervention.²⁵

Following successful nonoperative management of an initial episode of PSP, recurrence occurs in 22%–55% of patients.^3,8–11 The majority of surgeons in this survey would recommend operative intervention if and when patients develop a first recurrence of PSP. This is in keeping with the findings of several retrospective single-institution reviews.^3,8,14 This practice is also consistent with the BTS and ACCP adult guidelines, which both recommend surgical evaluation with the first recurrence.¹⁵ There may be some clinical situations where surgery should even be offered after the first episode of PSP. Fifteen percent of ACCP panel members would offer surgical intervention after the first occurrence particularly in patients whose activities, such as scuba diving or flying, placed them at increased risk.¹⁵ Similarly, 15% of respondents to this survey offer surgery after the first episode, more commonly those in practice for <5 years. Earlier operative intervention may reduce rates of readmission, shorter hospital length of stay, and lower hospital costs if the pneumothorax fails to resolve quickly during the first hospitalization.¹⁹ However there is still a nontrivial risk of recurrence (11%–40%) even after surgical intervention.^3,5,8,19 Furthermore, even with the higher recurrence rates associated with nonoperative management, the increased morbidity and cost of VATS with the first episode of PSP may not justify routine, initial operative intervention.^8,26

When operative intervention is performed, there is broad consensus among pediatric surgeons to utilize a minimally invasive approach (98%) and to perform stapled blebectomy (97%) and mechanical pleurodesis (77%). The advantages of VATS over thoracotomy have been established in a number of studies,^15,27 although there is some evidence that VATS may still be associated with a slightly higher recurrence rate.^11,18 There is good consensus that blebectomy/bullectomy in combination with some treatment of the pleura is associated with lower recurrence rates than either procedure alone and should be the twofold objective of any intervention for both adults and children.^12,21,26 Stapled bullectomy with pleurodesis or pleurectomy is recommended by both sets of guidelines and was the most popular option in this survey.^15,18 In the adult literature, there was thought to be some advantage of pleurectomy over pleural abrasion, but the comparative evidence for these two techniques is limited, and a combination of the two is often used.^18,28 With respect to agents used for chemical pleurodesis, talc is the most common. Tetracyclines are no longer recommended in the United Kingdom but doxycycline and, less commonly, minocycline are sometimes used in the United States. Staple line reinforcement is not specifically addressed in the guidelines but was used by only a minority of respondents.

This survey shows that among North American pediatric surgeons, there is some concordance with adult professional society guidelines for the management of PSP. It is still unclear, however, what potential differences may exist in children warranting pediatric-specific management considerations. The relative proportions of primary versus secondary disease among children, for example, are not known. Similarly, it has been suggested that recurrence rates are higher in younger patients^5,9,29,30 and that there may be a lower incidence of bullous disease.²⁶ As mentioned before, there is some evidence for improved outcomes with earlier surgical intervention in children, especially since the majority eventually undergoes surgery.^3,19,20 Randomized controlled trials comparing nonoperative management and operative intervention are needed to provide high quality evidence to inform pediatric-specific guidelines. Ultimately, an objective assessment of recurrence risk will best dictate the management approach.

There are limitations to this study, including the inherent risk of response bias in this type of voluntary survey. Furthermore, there was no mechanism to cross-check surgeons' responses against data on their actual operative practice. Finally, in the interest of preserving the brevity of the survey to maximize our response rate, additional clinical details about the exact clinical signs and symptoms guiding management were not ascertained.

Conclusion

There is significant practice variation in the management of pediatric PSP, including the use of CT, timing of VATS, and duration of observation for air leak before performing surgery. Long-term prospective data are needed to better inform guidelines and standardize practice.

Funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Authors' Contributions

K.W. assisted with data collection and analysis and was the primary author of the article. L.B. assisted with data collection and analysis. J.G. assisted with survey development and provided final editing of the article. T.B.L. conceptualized the project and supervised survey development, as well as preparation of the article.

Footnotes

Disclosure Statement

The authors report no proprietary or commercial interest in any product mentioned or concept discussed in this article.

Supplementary Material

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Supplementary Material

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