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Received: 24 March 2023 | Accepted: 10 September 2023

DOI: 10.1002/ppul.26703

ORIGINAL ARTICLE

Conservative and operative management of spontaneous


pneumothorax in children and adolescents: Are we abusing
of CT?

Angelo Zarfati MD | Valerio Pardi MD | Simone Frediani MD, PhD |


Ivan Pietro Aloi MD | Antonella Accinni MD | Arianna Bertocchini MD |
Silvia Madafferi MD | Alessandro Inserra MD

General and Thoracic Pediatric Surgery Unit,


Bambino Gesù Children's Hospital, IRCCS, Abstract
Rome, Italy
Background: No age‐specific pediatric guidelines exist for the management of
Correspondence spontaneous pneumothorax (SP) in children and adolescents. Treatment remains
Simone Frediani, MD, PhD, General and heterogeneous and center dependent. The role of computed tomography (CT) has
Thoracic Pediatric Surgery Unit, Bambino
Gesù Children's Hospital, IRCCS, Rome, Italy. yet to be defined.
Email: simone.frediani@opbg.net Aims: Review the management of SP in children and adolescents, with emphasis on

Funding information
conservative management and role of CT.
None Methods: Retrospective analysis of 61 consecutive patients with SP at single tertiary
center. Clinical, radiological, surgical data, follow‐up, and outcomes were revised.
Results: First‐line management was conservative for 32 (53%) patients and operative
for 29 (47%). Asymptomatic/paucisymptomatic patients managed conservatively
experienced less first‐line treatment failure. Furthermore, the patients needing at least
a chest drain or surgery during the follow‐up were significantly lower in the
conservative group. Conservative and operative patients showed no significant
differences regarding ipsilateral recurrences or contralateral occurrences. Of the 61
overall CTs performed, 14 (23%) had an impact on management. Forty‐three (70%)
patients had at least a CT, in 22 (51%) the CT was positive for blebs. For 10 of these
patients (45%) the presence of blebs had an impact on management. Patients with and
without blebs showed no differences regarding ipsilateral recurrence, contralateral
occurrences, or the need for at least a chest drain or surgery during the follow‐up.
Conclusions: First‐line conservative management had a significantly shorter hospitaliza-
tion and better outcome, with a similar incidence of recurrences. The presence of blebs at
CT does not predict the risk of recurrence. The CT scan should be reserved for a small
number of selected patients who have post‐VATS refractory or recurrent pneumothorax.

KEYWORDS
children, computed tomography, drainage, pneumothorax, VATS

Abbreviations: CT, computed tomography; SP, spontaneous pneumothorax; VATS, video assisted thoracic surgery.

This is an open access article under the terms of the Creative Commons Attribution‐NonCommercial‐NoDerivs License, which permits use and distribution in any
medium, provided the original work is properly cited, the use is non‐commercial and no modifications or adaptations are made.
© 2023 The Authors. Pediatric Pulmonology published by Wiley Periodicals LLC.

Pediatric Pulmonology. 2023;1–7. wileyonlinelibrary.com/journal/ppul | 1


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2 | ZARFATI ET AL.

1 | INTRODUCTION After the first chest X‐ray performed in the emergency room the
timing of a repeat X‐ray depended on the clinical status. In case of
Spontaneous pneumothorax (SP) is a relatively rare condition in favorable evolution, the X‐ray was performed at symptom remission
pediatric population.1 Tall, thin, male adolescents are more likely to to allow the discharge. Otherwise, the X‐ray was repeated in any
develop SP.1–3 Today, no age‐specific pediatric guidelines exist for moment of degradation. The failure of the first‐line management was
the management of children and adolescents with SP, and therapeu- defined as the need for surgery for a SP refractory or persistent to
tic approaches are drawn from adult literature. There are a number of the treatment. In the authors' institution postoperative management
adult recommendations, however there are still big discrepancies of the chest tube and discharge criteria were the following: in the
between them in terms of diagnosis and indications.4–7 Management absence of an air leak, the chest tube is closed. A chest X‐ray is
in pediatric patients remains heterogeneous and center dependent.4,8 performed to rule out any persistent pneumothorax after 12–24 h of
The use of CT remains controversial. CT is not routinely closure. The tube is removed if there is no or very little persistent
performed in pediatric SP, and its role is not yet clear. While it has pneumothorax. After the removal of the chest tube, the patients are
been reported that the finding of blebs/bullous lesions on CT is an released.
9
indication for surgery, some authors have reported that these are We considered that a CT with an “impact on the management”
not predictive of outcome or recurrence.8,10,11 However, some when it changed the patients care (e.g., indication to drain placement,
authors reserve a role for CT in selected cases.10,12 Others reported indication to surgery, etc). We defined a CT positive for blebs with an
even a useless overuse by clinicians.10 “impact on the management” when it changed the patients care (e.g.,
The aim of the present study was to analyze the management of indication to surgery, etc).
spontaneous pneumothorax in children and adolescents, with Categorical variables were reported as absolute and relative
particular emphasis on the role of conservative management in frequencies (%). Continuous variables were reported as median and
asymptomatic/paucisymptomatic patients. Additionally, we wanted range. As necessary, categorical variables were compared between
to review the role of the CT versus video‐assisted thoracic surgery groups using the Fisher exact test or the χ2 test. Differences between
(VATS), focusing on their impact and value on outcomes and groups for continuous variables were determined using a non-
recurrence. parametric test, Mann–Whitney U test. All p values were two‐sided,
and a p < .05 was considered significant.

2 | MATERIALS AND METHODS


3 | RESULTS
A retrospective review of consecutive patients with SP at single
tertiary center (Pediatric Surgery Unit, Bambino Gesù Pediatric The overview of the study is presented in Figure 1. Baseline and
Hospital, IRCCS) between January 2010 and January 2022 was general characteristics are detailed in Table 1. Sixty‐one patients
undertaken. Clinical, radiological, and surgical data, follow‐up and were identified with a median age of 14.2 years (range 2.3–17.9).
outcomes were revised. Exclusion criteria included incomplete data, Fourteen patients (22%) were female. Eight (13%) had Marfan
being lost to follow‐up and older than 18 at the time of diagnosis. The syndrome, and seven (11%) had a history of asthma. At presentation,
number of cases managed at our Institution during the study period 53 (87%) were paucisymptomatic, seven (11%) were highly sympto-
determined the sample size. Locally and nationally referred patients matic and one (2%) was asymptomatic. The pneumothorax was on
composed the study population. There was no randomization, and the left side in 35 (57%) of the cases, on the right side in 25 (41%),
management was determined by the surgeon's discretion. and bilateral in 1 (2%).
Patients were defined as asymptomatic in the absence of First‐line management was conservative for 32 (53%) and
symptoms, paucisymptomatic in case of mild chest pain, cough or operative for 29, and their comparison is presented in Table 2. The
mild dyspnea, and highly symptomatic in the case of severe pain operative management was drainage for 24 and surgery for 5. In the
or dyspnea. The first line management may be conservative or surgically treated subgroup, apical blebs were discovered in three of
operative. At the authors' institution conservative management the five paintings after a VATS exploration, which was performed on
consisted in hospitalization for clinical and vital signs monitoring, all of them. In the remaining two the exploration was negative. One
painkillers, and oxygen therapy. Instead, at our institution, the of these patients underwent pleurodesis through scarification. In
operative management included chest tube placement and VATS. every case, a postoperative chest tube was inserted for a median of 9
The chest tube placement, except emergent life threating days (range: 3–15 days). Overall, the median hospital stay was 6 days
situations, was ed in the operative room under general anesthe- (range 0–45). The median hospital stay was significantly shorter in
sia. Instead, the thoracoscopic approach consisted in an the conservative group (4 days [range 0–33] vs. 8 [7–45], p < .00001).
exploration of pleural space and lung parenchyma. When Overall, 10 (16%) patients needed further surgery for the treatment
bullae/blebs were found at exploration, the lesions were of first‐line management failure. All 10 patient received a VATS
resected. Furthermore, the procedure may consist in a mechani- exploration, and 8 of them had their apical blebs removed. In such
cal or chemical pleurodesis. subsets, two individuals underwent an additional pleurodesis (one
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ZARFATI ET AL. | 3

FIGURE 1 Overview of the study.

TABLE 1 Baseline and general data.

Patients 61

Median age at first episode (range) 14.2 (2.3–17.9)

Females 22% (14)

Marfan 13% (8)

Asthma 11% (7)

Presentation Asymptomatic 2% (1)

Paucisymptomatic 87% (53)

Highly symptomatic 11% (7)

Side Left 57% (35)

Right 41% (25)

Bilateral 2% (1)

First‐line management Conservative 53% (32)

Operative Overall 47% (29)

Drainage 24

Surgery 5

Median age at follow‐up end (range) 17.2 (5.5–27)

Median years of follow‐up (range) 1.2 (0.1–12)

TABLE 2 Comparison of first‐line management outcomes and follow‐up.

Total (n = 61) Conservative (n = 32) Operative (n = 29) p Value

Median days of hospitalization at first episode (range) 6 (0–45) 4 (0–33) 8 (3–45) <.00001

First‐line treatment failure needing further surgery 16% (10) 6% (2) 27% (8) .037

≥1 ipsilateral recurrence 39% (24) 31% (10) 48% (14) .199

≥1 contralateral occurrence 14% (9) 9% (3) 20% (6) .287

≥1 surgery or chest drain during the follow‐up 36% (22) 21% (7) 51% (15) .018
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4 | ZARFATI ET AL.

TABLE 3 Comparison of first‐line management outcomes and follow‐up depending on the type of operative management.

Total (n = 29) Drainage (n = 24) Surgery (n = 5) p Value

Median days of chest tube (range) 8 (3–45) 8 (3–45) 9 (3–15) .756

First‐line treatment failure needing further surgery 27% (8) 33% (8) 0% (0) .283

≥1 ipsilateral recurrence 48% (14) 54% (13) 20% (1) .329

≥1 contralateral occurrence 20% (6) 25% (6) 0% (0) .552

≥1 surgery or chest drain during the follow‐up 51% (15) 54% (13) 40% (2) .650

T A B L E 4 Role of CT for management and follow‐up of SP in Regarding the role of the CT (Table 4), 43 (70%) patients had at
children and adolescents. least a CT at diagnosis, during management, or at follow‐up, while 12

Patients with ≥1 CT 70% (43) (19%) had at least two CTs. Despite the complete absence of
symptoms, the CT was repeated in 10 of the 12 patients to rule out
Patients with ≥2 CT 19% (12)
the presence of blebs during follow‐up visits after discharge. For the
Overall CTs 61 other two, a repeat CT was done at the first episode due to a
CT timing I episode 22% (14) persistent pneumothorax. Overall, 61 CTs were performed. Fourteen

Recurrence 13% (8) CTs (23%) had an impact on management. Regarding the timing, 14
(23%) were performed at the first episode, 8 (13%) at recurrence and
Follow‐up 63% (39)
39 (63%) during the follow‐up.
CT with an impact on management 23% (14) Of the 43 patients with at least one CT, 22 (51%) had at least one
Patients with ≥1 CT with an impact on management 54% (33) CT scan that was positive for blebs (Table 5). In 10 of these patients

Patients with ≥1 CT with Blebs 51% (22/43) (45%) the presence of blebs on CT had an impact on management.
However, patients with and without blebs at CT showed no
Patients with ≥1 CT with blebs with an impact on 45% (10/22)
differences regarding ipsilateral recurrence (59% [13], vs. 33% [7],
management
p = .129), contralateral occurrences (13% [3], vs. 28% [6], p = .280), or
Abbreviations: CT, computed tomography; SP, spontaneous
the need for at least a chest drain or surgery during the follow‐up
pneumothorax.
(54% [12], vs. 28% [6], p = .124).

talcage, one scarification). The remaining two had a negative


exploration. In every case, a postoperative chest tube was inserted 4 | D IS CU SS IO N
for a median of 8 days (range: 4–16 days). This failure was
significantly lower in the conservative group (6% [2/32] vs. 27% In our experience, patients who were managed conservatively at the
[8/29], p = .037). Patients were followed for a median of 1.2 years first episode had significantly shorter hospitalizations and experi-
(range 0.1–12) and until a median age of 17.2 years (range 5.5–27). enced fewer treatment failures requiring surgery during the same
During the follow‐up, 24 patients (39%) experienced ipsilateral stay. Furthermore, these patients showed no statistically significant
recurrence and nine (14%) had contralateral occurrences. Conserva- differences during the follow‐up for ipsilateral recurrence and
tive and operative groups showed no statistically significant differ- contralateral occurrence. However, the patients needing at least
ences regarding ipsilateral recurrence (31% [10], vs. 48% (14), one surgery or chest drain during the follow‐up were significantly
p = .199) or contralateral occurrences (9% [3] vs. 20% [6], p = .287). fewer in the conservative group. The first systematic review and
However, significantly fewer conservatively managed patients meta‐analysis of pediatric spontaneous pneumothorax management
needed at least a chest drain or surgery during the follow‐up (21% was recently published by Miscia et al.8
[7] vs. 51% [15], p = .018). These authors concluded that first‐line surgery reduced the risk
The comparison of the first line operatively managed patients of recurrence without increasing the hospital stay. Instead in the
is shown in Table 3. Patients treated with drainage and surgery present study, we found that first‐line operative management was
showed no statistically significant differences regarding days of associated with an increased hospitalization length without decreas-
chest tube (8 [3–45] vs. 9 [3–15] p = .756), first‐line treatment ing the risk of recurrence. This may be explicated by a possible
failure needing further surgery (33% [8] vs. 0% [0], p = .283), selection bias of our methodology. Due to the lack of randomization
ipsilateral recurrence (54% [13] vs. 20% [1], p = .329), contra- or prospective design, the most severe cases were more likely be
lateral occurrences (25% [6] vs. 0% [0], p = .552), and the need for treated operatively. So, we could have selected the patients who
at least a chest drain or surgery during the follow‐up (54% [13] were at higher risk of treatment failure, increased hospital stay
vs. 40% [2], p = .650). and recurrence.
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ZARFATI ET AL. | 5

TABLE 5 Comparison of outcomes and follow‐up of patients with blebs at CT and patients with a negative CT.

Patients with ≥1 CT (n = 43) ≥1 CT with Blebs (n = 22) No blebs (n = 21) p Value

≥1 ipsilateral recurrence 46% (20) 59% (13) 33% (7) .129

≥1 contralateral occurrence 20% (9) 13% (3) 28% (6) .280

≥1 surgery or chest drain during the follow‐up 41% (18) 54% (12) 28% (6) .124

Abbreviation: CT, computed tomography.

In a recently published mixed adolescents‐adults randomized surgical intervention. However, patients with and without blebs at CT
13
control trial, Brown and colleagues reported interesting findings. showed no differences regarding ipsilateral recurrence, contralateral
The study has been conducted in a mixed adolescent‐adult occurrences, or the need for at least a chest drain or surgery during
population (14–50 years), with a first‐known, unilateral, moderate‐ the follow‐up. Therefore, from this limited retrospective experience,
to‐large primary spontaneous pneumothorax. The outcomes of the presence of blebs does not seem to have a major impact of the
conservative management are really promising in this population. outcomes of SP patients.
However, the trial does not seem to be adequate for a pediatric In the present series, 70% of patients received at least one CT.
tertiary center for some methodological reasons. Pediatric studies, as However, of all the 61 CTs performed only 14 (23%) had an impact
the present one, include a population quite different from the trial on the patient's management. Indeed, 77% of the CT performed
(1–18 years, rather 14–50 years). Furthermore, the main criteria for could have been avoided. Moreover, even in the 22 (51%) patients
patient selection in this trial was the size. However, the size has been with blebs at CT, only 45% of these had an impact on management.
calculated with the Collins and colleagues formula.14 Since its first Finally, the blebs and no blebs groups showed no statistically
description in 1995, the clinical everyday use of this formula is significant difference during the follow‐up for ipsilateral recurrence,
extremely limited and, to the authors best knowledge, the formula contralateral occurrence or the need for surgery or drainage. In the
has never been validated for a use in pediatric patients. Therefore, aforementioned paper Miscia and colleagues concluded that the
any conclusion regarding the impact of the size on the outcomes on presence of blebs does not indicate a higher risk of recurrence.8 In
pediatric cases may be misleading. Anyway, this trial has several this sense, our experience is consistent with the available literature.
interesting and promising outcomes that need to be validation in a The analysis of our experience showed no difference of
pediatric setting. A similar trial should be conducted in pediatric outcomes between the two types of operative treatment considered,
patients. This study may helpful to clarify many ambiguous aspects of VATS and drain. Therefore, these findings let us sometimes
the management of the spontaneous pneumothorax in this setting, as reconsider our operative approach to highly symptomatic patients,
size definition (e.g., large pneumothorax), best first‐management (e.g., conservative management failure, persistent, or recurrent pneumo-
role of conservative management), role of CT, risk factors for thorax. In our institution, in this selected group of complicate cases,
persistence or recurrence, and so forth. sometimes we may favor VATS over drainage‐only when there is an
One of the aims of the present study was to perform a critical indication for operative treatment. Therefore, given that both the
review the role of the CT, focusing on its impact and value on procedures require general anesthesia, we believe that in selected
outcomes and recurrence. We considered that a CT with had an cases the thoracoscopic exploration adds valuable diagnostic
“impact on the management” when it changed the patients care (e.g., information regarding the presence of blebs or associated anomalies.
indication to drain placement, indication to surgery, etc). The Furthermore, after exploration the surgeon has three possibilities:
objective was to try to understand the number of avoidable CTs perform blebs resection, perform a pleurodesis, or leave only a chest
(and radiations to pediatric patients) we performed. As quite evident tube. The present series represent a clear and critical review of more
for the reader, it appears that CT was not helpful in most cases and than a decade of experience of our large volume pediatric tertiary
may lead to unnecessary interventions and radiation exposure. center. This study consists in a report a period of stable and
Hoverer, the CT scan should be reserved for a small number of homogeneous management. When we realized that some of our
patients with complex scenarios, as post‐VATS refractory or practices may be improved we performed this critical analysis to
recurrent pneumothorax. ameliorate the patient's care. Therefore, from our findings and the
Furthermore with the same logic, we defined a CT positive for recent literature we developed a protocol for the management of SP
blebs with had an “impact on the management” when it changed the in children and adolescents.7,8,11,12 (Figure 2). Our proposal had three
patients care (e.g., indication to surgery, etc). The aim as always to purposes: to redefine the role of conservative treatment, VATS and
define the role of this finding on the patient's care. Of the patients CT, support the need for age‐specific management, and promote
with at least one CT, 51% had at least one CT scan that was positive debate between experts from tertiary centers. At presentation the
for blebs. In 45% of these patients, the presence of blebs on CT had management should be guided by clinical status and symptoms.
an impact on management. From our limited experience, is still Asymptomatic and pauci‐symptomatic patients at first episode
unclear if blebs or bullae seen on CT or on VATS should lead to should be admitted to the hospital and treated conservatively with
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6 | ZARFATI ET AL.

FIGURE 2 The Bambino Gesù children's hospital protocol for management of spontaneous pneumothorax in children and adolescents.

oxygen. In cases of favorable evolution, with resolution or decrease reducing the risks and stress of a longer hospital stay for the
of the pneumothorax, the patient should be discharged with a clinical patient.13,14
and radiological check in 7–10 days. In the event of symptoms or if We recognize the limitations of the present experience. The first
the pneumothorax persists or worsens, the patients should have a is the retrospective nature and the limited monocentric sample of the
thoracoscopic exploration. In case of recurrence, patients should be series. Additionally, this study lacks randomization and a prospective
hospitalized and treated with oxygen therapy and VATS. Additionally, design because of the methodology. Prospective and multicenter
during their hospital stay, these patients should have a multi- studies are needed to generalize our findings.
disciplinary examination for comorbidities, which at our institution In Conclusion, first‐line conservative management had a signifi-
includes a genetic, cardiologic, and ophthalmologic work‐up. Instead, cantly shorter hospitalization and experienced less treatment failure
patients highly symptomatic at presentation should be hospitalized requiring surgery, with a similar incidence of recurrences. The
and treated with oxygen and VATS. A chest drain should be presence of blebs at CT does not predict the risk of recurrence.
immediately placed in case of suspect of tensive pneumothorax. The CT scan should be reserved for a small number of patients who
Every time a VATS is performed the pleural space should be carefully have post‐VATS refractory or recurrent pneumothorax.
explored. In cases of blebs, a bullectomy should be performed.
Otherwise, a talcage should be considered in the absence of obvious A UT H O R C O N T R I B U TI O NS
blebs. In any case, a chest drain should be placed. In cases of Angelo Zarfati: Conceptualization; investigation; funding acquisition;
postoperative persistence of pneumothorax or postoperative ipsi- writing–original draft; methodology; validation; visualization; writing—
lateral recurrence, a chest CT should be considered to assess any review & editing; software; formal analysis; project administration;
anatomical (residual blebs, etc.) or pathological (associated anomalies, data curation; supervision; resources. Valerio Pardi: Data curation;
etc.) etiology before the repeat of a VATS exploration. The targeted supervision; resources; project administration; formal analysis; soft-
use of CT is perfectly in accordance with the current culture of ware; methodology; validation; visualization; writing—review & editing;
radioprotection, favoring the thoracoscopic approach the radiological conceptualization; investigation; funding acquisition; writing—original
exposure is reduced and the therapeutic process is accelerated, also draft. Simone Frediani: Conceptualization; investigation; funding
10990496, 0, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/ppul.26703 by Cochrane Romania, Wiley Online Library on [23/11/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
ZARFATI ET AL. | 7

acquisition; writing—original draft; methodology; validation; visualiza- 3. Engwall‐Gill AJ, Weller JH, Rahal S, Etchill E, Kunisaki SM, Nasr IW.
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ORCID
Simone Frediani http://orcid.org/0000-0003-4629-8132

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