Journal of Pediatric Surgery Case Reports 73 (2021) 102000

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Journal of Pediatric Surgery Case Reports

Expanding pneumatocele in an ELBW infant

Theresa R. Price a, *, Mechelle A. Miller a, Alicia C. Prescott a, Jeffery M. Meadows b,
Benjamin D. Tabak c
a Department of Pediatrics, Tripler Army Medical Center, Honolulu, HI, USA
b Department of Radiology, Tripler Army Medical Center, Honolulu, HI, USA
c Department of Surgery, Tripler Army Medical Center, Honolulu, HI, USA

ARTICLE INFO ABSTRACT

Keywords: Pneumatoceles are gas-filled cysts that can develop within lung parenchyma, often as the result of barotrauma.
Pneumatocele Pneumatoceles in extremely low birth weight (ELBW) premature infants present unique challenges, particularly
Extremely low birthweight infant when conservative management fails. We present a case of an enlarging pneumatocele that resulted in severe he-
Premature
modynamic compromise which required surgical intervention. We review the described interventional tech-
Infant
niques for pneumatocele management in the EBLW population and their indications.
Very low birthweight infant
NICU
Barotrauma
Volutrauma
Ventilator
Pneumonia
Non-invasive positive pressure ventilation
Video-assisted thorascopic surgery
Bronchopulmonary dysplasia
Lung injury
Chest tube
Pneumothorax
Pigtail catheter
Mini-thoracotomy

1. Introduction 1.1. Case presentation

The etiology of pneumatoceles has long been attributed to mechani-
cal ventilation or complications of bacterial pneumonia [1–7]. In the
era of gentle ventilation, antenatal steroids, and surfactant therapy, the
incidence in the neonatal population is low, estimated at 1.8% [8].
There are no established criteria for when or how to intervene on pneu-
matoceles in ELBW infants which do not respond to conservative man-
agement. We present a rare case of an expanding pneumatocele in an
ELBW infant, which ultimately resulted in hemodynamic compromise,
and discuss the unique challenges to treatment in this population.
A female infant, twin A of mono-di twin gestation, was born via Ce-
sarean section to a 29- year-old, gravida 1, para 1 mother at 24 + 5
weeks gestation due to refractory pre-eclampsia with severe features.
The prenatal course was otherwise uncomplicated and the infant's
mother received a complete course of antenatal steroids. The infant's
birth weight was 500 g and Apgar scores were 6 and 8 at 1 and 5 min-
utes, respectively. She was intubated, given surfactant, and placed on
high-frequency oscillatory ventilation (HFOV). Two weeks later, chest
radiographs were significant for diffuse bilateral infiltrates and labs
suggested ongoing sepsis. Vancomycin, Piperacillin/Tazobactam,
Flagyl, and Fluconazole were administered and adjusted based on sensi-

Abbreviations: ELBW, extremely low birthweight infant; VLBW, very low birthweight infant; DOL, day of life; HFOV, high frequency oscillatory ventilation; PPV,
positive pressure ventilation; PIE, pulmonary interstitial emphysema; CMV, conventional mechanical ventilation; NIPPV, non-invasive positive pressure ventilation;
CT, computed tomography; NICU, neonatal intensive care unit; VATS, video-assisted thoracoscopic surgery.
* Corresponding author. Department of Pediatrics, Tripler Army Medical Center, 1 Jarrett White Road, Honolulu, HI, 96859, USA.

E-mail address: theresa.r.price.mil@mail.mil (T.R. Price).

https://doi.org/10.1016/j.epsc.2021.102000
Received 29 July 2021; Accepted 8 August 2021
Available online 9 August 2021
2213-5766/© 2021 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
T.R. Price et al.
tivities and culture results, which ultimately grew pan-sensitive Staphy-
lococcus aureus and Acinetobacter junii.
On day of life (DOL)18, chest radiographs revealed, for the first
time, a round 1.7 cm radiolucency concerning for a developing pneu-
matocele. Over the next 2 weeks the defect nearly tripled in size to 4.8
cm causing a significant mediastinal shift (Fig. 1a). Tension physiology
developed rather suddenly, and a dopamine infusion was initiated for
new onset hypotension. Progressive difficulty with oxygenation and
ventilation required upward titration on the ventilator to 100% FiO2
and a mean airway pressure of 17 cmH2O. Despite these interventions,
the patient's oxygen saturation remained in the low 90's which
prompted urgent surgical consultation.
Percutaneous chest tube placement under fluoroscopy or CT guid-
ance was considered, but due to the infant's size and clinical instability,
it was unsafe to transport the baby to a radiology suite. Bedside ultra-
sound-guided drainage was considered, but acoustic shadowing from
the normal lung and pneumatocele, both of which are air-filled, made
the two structures indistinguishable. The decision was therefore made
to perform a “mini” posterolateral thoracotomy through an incision just
large enough to identify the pneumatocele, which was visibly discol-
ored relative to the normal lung (Fig. 2).
A 6 French pigtail catheter was inserted into the pneumatocele us-
ing a micropuncture kit and serial dilation via a modified Seldinger
technique (Fig. 1b). Appropriate placement in the pneumatocele was
confirmed with injection of contrast through the pigtail and a portable
chest radiograph. An additional straight chest tube was placed into the
right pleural cavity to prevent development of post-operative pneu-
mothorax. The infant tolerated the surgery well and had immediate im-
provement in ventilation and oxygenation. The patient weighed 880 g
at the time of the procedure.
On DOL 55 she was extubated to NIPPV, and the straight chest tube
was removed. The pigtail catheter was removed on DOL 72. The pneu-
matocele was no longer evident on chest radiographs by DOL 99, and
she was discharged home on DOL 151 with low flow oxygen (Fig. 1c).
The infant undergoes routine monitoring by her pulmonologist for
bronchopulmonary dysplasia management.
Fig. 1. Serial progression of pneumatocele. a) Large pneumatocele with mediastinal shift. b) Pigtail catheter placed surgically into the pneumatocele; straight
chest tube in the pleural space. c) Pneumatocele resolved by day of discharge.
2
Journal of Pediatric Surgery Case Reports 73 (2021) 102000
1.2. Review and discussion
ELBW infants are susceptible to pneumatocele formation, primarily
in the lower lobes, which bear the brunt of ventilator-associated lung
injury. The incidence of pneumatocele formation is thought to have de-
creased in the post-surfactant era, but persistent pneumatoceles may be
associated with higher mortality [6]. We conducted an extensive litera-
ture search using PubMed, Embase, and Google Scholar including the
search words: pneumatocele, pneumatocoele, ELBW, VLBW, infant,
preterm, pulmonary interstitial emphysema, pneumonia, percutaneous
drainage, ultrasound, thoracotomy, and chest tube. We identified 16 ar-
ticles that discussed treatment of pneumatoceles in ELBW infants (Table
1, online content only).
Conservative management is the mainstay of initial treatment of
pneumatoceles, with reduction of mean airway pressures and treatment
of underlying infections. In rare cases, pneumatoceles will continue to
expand over time, despite maximal medical therapy. This expansion
may result in tension physiology with life-threatening hemodynamic
compromise. Whereas in cases of tension pneumothorax, in which air
escapes into the pleural space and can be easily accessed by blind place-
ment of a chest tube, pneumatoceles exert pressure from within the
lung parenchyma, usually within the lower lobes.
Several anatomical factors must be considered during evacuation of
a pneumatocele in the ELBW infant. Firstly, the technique convention-
ally utilized for evacuation of a pneumothorax, with a blunt-tipped
chest tube oriented towards the apex, may be expected to miss the
pneumatocele, or push it away. A sharp instrument is required to pierce
the lung parenchyma and access the pneumatocele. Whereas it is gener-
ally safe to blindly insert a blunt instrument through the intercostal
space to relieve a pneumothorax, blind advancement of a sharp instru-
ment into a neonatal chest may result in life-threatening damage to ad-
jacent vital organs.
Secondly, as pneumatoceles are most commonly in the right lower
lobe (18/19 cases in one series [4]), the chest tube may need to be
placed at a lower intercostal space, or oriented inferiorly. A common
rule-of-thumb for pneumothoraces is to blindly place chest tubes at or
above the nipple line to avoid inadvertent placement into the abdomen,
T.R. Price et al. Journal of Pediatric Surgery Case Reports 73 (2021) 102000

Fig. 2. Open thoracotomy for chest tube placement in the large right lower lobe
pneumatocele. Note the difference in color between the pneumatocele (purple,
left) and normal lung parenchyma (light pink, right). (For interpretation of the
references to color in this figure legend, the reader is referred to the Web ver-
sion of this article.)
as the diaphragm tends to rise higher in the chest than anticipated. In-
advertent placement of a needle or chest tube beneath the diaphragm in
a neonate, in particular, could result in life-threatening liver hemor-
rhage. The liver of an ELBW infant produces notoriously difficult-to-
control bleeding, and could result in exsanguination before the injury is
fully appreciated.
Image guidance is thus recommended for any attempts at percuta-
neous insertion of chest tubes in ELBW infants, but the availability of
advanced imaging techniques is often limited. In one report, CT-
guidance was planned in an ELBW infant, but could not be done due to
instability and high ventilator settings. The infant eventually had CT-
guided bilateral pigtail catheters placed a few weeks later, but the in-
fant died several days later [13]. A few authors have described the use
of fluoroscopy for guiding chest tube placement [16,18], but this often
requires transport to a radiology or surgical suite, which may not be
feasible for a neonate that is hemodynamically unstable or on maxi-
mum ventilator settings.
There is one recent report in the literature describing percutaneous
drainage of a left-sided pneumatocele using ultrasound guidance [17].
This modality may be ideal for the unstable ELBW infant, as it can be
performed at the bedside. However, the pneumatocele may not be dis-
tinguishable from normal lung parenchyma. Pneumatoceles are gener-
ally filled with air, not fluid, which is more easily drained under ultra-
sound guidance. Acoustic shadowing may prevent clear identification
of a pneumatocele, which can be indistinguishable from surrounding
lung parenchyma. Inadvertent puncture of the normal lung tissue could
result in additional problems, including bronchopleural fistula.
Surgical management has also been reported, often after failed chest
tube insertion [20]. Thoracotomy offers the ability to immediately and
definitively remove the pneumatocele. However, complete resection
could result in life-threatening hemorrhage or permanent respiratory
insufficiency. Even if a wedge resection or partial lobectomy can be
achieved, some functional lung tissue will almost certainly be removed
in the process. Two authors have described the use of video-assisted
thoracoscopic surgery (VATS) to guide resection or pigtail catheter in-
sertion for pneumatoceles in babies over 1 kg [19,9]. Another tech-
nique afforded by thoracotomy is decompression of the pneumatocele
using an electrosurgical device, such as Ligasure ® (Covidien, USA)
[15].
We present herein another surgical alternative, a “mini-
thoracotomy”, for surgical placement of a chest tube. Just enough of a
thoracotomy incision is needed to identify the pneumatocele, which
tends to have a distinct color relative to normal lung parenchyma. This
allows precise placement of a pigtail catheter in a very controlled set-
ting at bedside, without the need to transport the baby. It avoids the
risks of percutaneous chest tube placement and poses little more overall
procedural risk, given the limited and muscle-sparing incision utilized.
Clinical improvement can be observed soon after the chest tube place-
ment. The patient may be monitored with the chest open for several
minutes, and if no improvement is noted, the thoracotomy incision can

Table 1
Literature Review Summary of Pneumatoceles in ELBW/VLBW preterm infants.
Management Authors Year Age (weeks) Weight Procedure Outcome
(grams)

Non- Williams et al. [4] 1988 20 infants; 26–31 750–1250 19 infants managed non-operatively; Survived
operative weeks 1 required surgical resection (unspecified)
Andreou et al. [9] 2001 28 1280 Contralateral single lung ventilation Survived
Hussain et al. [6] 2009 19 infants; Multiple All 19 infants managed non- operatively 15/19
≤30 weeks Survived
Joseph et al. [10] 2010 25 85 Contralateral single lung ventilation Survived
Puthiyachirakkal 2018 27 738 Non-operative Survived
[11] 25 590 Non-operative Survived
Parappil et al. [12] 2020 26 1000 Non-operative Survived
Chest tube Arias-Camison et al. 2001 24 725 Fluoroscopy-guided pigtail catheter Survived
[13]
Fujii et al. [8] 2003 23 533 Fluoroscopy-guided pigtail catheter Survived
Jackson et al. [14] 2008 30 Not Failed bilateral pigtail catheters; VATS-guided chest tube placement Survived
provided
Kumar et al. [7] 2018 26 900 CT-guided bilateral pigtail catheters Died
Munoz et al. [15] 2019 28 1240 Percutaneous pigtail catheter Survived
Muniraman et al. 2021 25 810 Ultrasound-guided pigtail catheter Survived
[16]
Surgical Fujii et al. [17] 2008 28 1240 VATS-guided pigtail catheter Survived
Sacks et al. [18] 2014 3 infants; 23–24 500–695 3 infants failed chest tube; 2 infants emergent thoracotomy; 1 infant thoracotomy 3/3
weeks with lobectomy Survived
Harada et al. [19] 2018 23 593 Thoracotomy; Resection with Ligasure Survived
Min et al. [20] 2018 27 1000 VATS-guided resection Survived
Puthiyachirakkal 2018 26 955 Right lower lobectomy Survived
[11]
Present case 2021 24 500 Mini-thoracotomy with pigtail catheter Survived

3
T.R. Price et al.
be extended to perform a surgical resection or decompression with an
electrosurgical device.
Given that many pneumatoceles in this population will resolve
spontaneously, a conservative initial approach with gentle ventilation
strategies and treatment of any suspected infection seems appropriate.
Clinical deterioration in the form of worsening respiratory status or he-
modynamic instability should prompt consideration of procedural in-
tervention. These clinical indicators should be taken into consideration
along with radiographic changes, including expansion of the pneumato-
cele and accompanying mediastinal shift.
2. Conclusion
Pneumatoceles pose unique challenges in the ELBW infant popula-
tion. Conservative management with gentle ventilation is the mainstay
of treatment. Pneumatoceles that continue to expand and cause clinical
deterioration should prompt intervention prior to the development of
tension physiology. Due to multiple anatomical considerations in the
ELBW infant, blind chest tube placement is ill-advised. We present a
‘mini’ thoracotomy technique to address these problems safely in un-
stable infants. We also present a literature review of alternative proce-
dural options, including percutaneous chest tube insertion under fluo-
roscopic, CT, VATS, or sonographic guidance.
Consent
The parent of patient consented to publication of the case although
no identifiable information is available in the manuscript.
Funding
No funding or grant support.
Authorship
All authors attest that they meet the current ICMJE criteria for Au-
thorship.
Declaration of competing interest
The authors declare that they have no known competing financial
interests or personal relationships that could have appeared to influ-
ence the work reported in this paper.
Acknowledgements
Thank you to Dr. Milissa Jones for her review of the manuscript.
Journal of Pediatric Surgery Case Reports 73 (2021) 102000
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