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Indian J Pediatr (2010) 77:10251027

DOI 10.1007/s12098-010-0154-6

CLINICAL BRIEFS

Pulmonary Interstitial Emphysema Complicating


Pneumonia in an Unventilated Term Infant
Hyun Seung Lee & Soo Ah Im

Received: 26 January 2010 / Accepted: 15 June 2010 / Published online: 27 August 2010
# Dr. K C Chaudhuri Foundation 2010

Abstract A case of pulmonary interstitial emphysema with


pneumothorax and pneumomediastinum complicating
pneumonia in a 6-week-old infant is reported. The patient
had no history of resuscitation, bag and mask ventilation,
nasal continuous positive airway pressure or mechanical
ventilation.
Keywords Pulmonary interstitial emphysema .
Air leak syndrome . Pneumonia
Pulmonary interstitial emphysema (PIE) is a form of air
collection in the pulmonary interstitial tissue caused by
alveolar air-leakage; it has been associated with the use of
mechanical ventilation, prematurity and the respiratory
distress syndrome [1, 2]. However, the occurrence of PIE
associated with pneumonia has not been frequently reported
[3, 4]. There is only one case report of PIE in a full-term
infant published, that was probably associated with a
previous viral pneumonia [5].
A full-term infant with an acute lobar PIE and
associated tension pneumothorax with pneumomediastinum that occurred in a pneumonic process, with no
history of mechanical ventilation, was successfully managed non-surgically.

H. S. Lee
Department of Pediatrics, College of Medicine,
The Catholic University of Korea,
Seoul, South Korea
S. A. Im (*)
Department of Radiology, College of Medicine,
The Catholic University of Korea,
Seoul, South Korea
e-mail: saim@catholic.ac.kr

Report of Case
A previously healthy 6-week-old infant girl was admitted to
the hospital with grunting and poor oral feeding for 1 day.
The patient was born at term following an uneventful
delivery and had no medical problems. The physical
examination revealed slightly ill appearing infant with
temperature of 37.4C, HR 128/min, BP 90/50, and RR of
42/min with intermittent grunting respirations and decreased breath sounds over both lung fields. The initial
chest radiograph showed obscure infiltrates in both lower
lung fields. Laboratory evaluation demonstrated a leukocytosis and elevated C-reactive protein. Empirical intravenous
antibiotics were started. However, 7 h following admission
the patient became dyspneic, and developed tachypnea up
to 60 breaths/min, prolonged grunting, chest retractions,
and fever of 37.8C, with metabolic acidosis (pH, 7.28;
HCO3 concentration, 17.4 mmol/L; pCO2, 37.9 mmHg;
pO2, 58.4 mmHg; and base excess, 9.6 mEq/L). The
administration of 2 L/min oxygen via a nasal cannula was
required. The chest radiograph showed bilateral bronchopneumonia with perivascular linear lucencies in the right lung
field, suggestive of a PIE and a right-sided pneumothorax.
On day 2 of admission, the fever subsided; however, the
respiratory distress persisted with tachypnea up to 70
breaths/min, subcostal retraction, and prolonged grunting.
The follow-up chest radiograph demonstrated that the rightsided PIE and pneumothorax had worsened and an
associated pneumomediastinum developed (Fig. 1). A
subsequent chest CT demonstrated the presence of bilateral
bronchopneumonia with PIE in the right upper and middle
lobe, and an associated right tension pneumothorax and
pneumomediastinum (Fig. 2). Arterial blood gas analysis
showed the following: pH, 7.42; HCO3 concentration,
19.1 mmol/L; pCO2, 30 mmHg; pO2, 78.4 mmHg; base

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Fig. 1 Chest AP obtained on admission day 2 demonstrates the


presence of bilateral pulmonary infiltrates, a hyperinflated right lung
with more prominent perivascular linear and cystic lucencies of PIE
distributed diffusely in the lung (arrows). Right-sided pneumothorax
(arrowheads) and the newly developed pneumomediastinum (thin
arrows) are also noted

excess, 5.6 mEq/L. A chest tube connected to a water seal


suction device was inserted, and the symptoms gradually
improved. Given the diagnosis of acute lobar PIE, the
patient was managed non-surgically; the air leakage did not
worsen and mechanical respiration was not needed. The
patient was closely monitored.
By day 6, the PIE, pneumomediastinum and pneumothorax were completely resolved on the chest radiograph.
The suction was stopped and the chest tube was removed.
On day 9, the patient was discharged to continue the
course of antibiotics orally for 5 days. Follow-up chest
radiographs obtained after 5 days and at 4 months showed
total resolution of the lung findings without sequelae.

Indian J Pediatr (2010) 77:10251027

bronchial intubation and antibiotics. Crosswell et al. [7].


reported an unventilated preterm infant with acute unilateral
PIE associated with presumed pneumonia, who was treated
with supplemental oxygen and antibiotics. Pursnani et al.
[5] reported an unventilated full-term infant with persistent
localized PIE causing an intractable pneumothorax and
associated with a previous viral pneumonia, who had surgery
for treatment and a definitive diagnosis. Boisset et al.
[4]. reported pulmonary interstitial air-leakage associated
with artificial ventilation, in their autopsy cases with
staphylococcal pneumonia, measles interstitial pneumonia
and pulmonary tuberculosis.
PIE should be differentiated from other cystic lung
lesions including congenital adenomatoid malformation,
congenital lobar emphysema, lymphangiectasia, a bronchogenic cyst, cystic lymphangioma, and diaphragmatic hernia
[2]. These conditions were excluded by the initial chest
radiograph and CT scan in this case.
Pneumonia may contribute to the development of pulmonary air leaks by at least three mechanisms: 1) air trapping
from mechanical or check-valve obstruction within the bronchi
by mucus and inflammatory exudates, 2) reduced strength or
direct disruption of the alveolar lining from parenchymal
inflammation or necrosis as commonly seen in necrotizing
pneumonia and 3) decreasing lung compliance [4, 5].
Primarily, PIE should be managed conservatively to
avoid the complications associated with radical intervention. Management includes lateral decubitus positioning,
supplemental oxygen, contralateral selective intubation or

Discussion
The initial event of air leaks including PIE reflects over
distension and rupture of the alveoli, which can arise from
any condition increasing intra-alveolar pressure or volume
combined with any underlying condition that decreases
lung compliance [1]. The extra-alveolar air producing a
PIE can dissect along bronchovascular sheaths toward the
hilum and can create a pneumomediastinum, pneumothorax, pneumopericardium, subcutaneous emphysema or
pneumoperitoneum [1, 6].
Few cases of PIE associated with pneumonia have been
documented. ODonovan et al. [3]. reported a ventilated
preterm infant with acute unilateral PIE after staphylococcal
pneumonia, who was treated with prolonged selective

Fig. 2 Chest CT scan with lung window obtained on the same day
depicts air tracking along the vascular tree to the mediastinum creating
a suspended linear branch or dot appearance in the right lung field as a
characteristic finding of PIE (arrows). Right-sided pneumothorax
(arrowheads) and pneumomediastinum (thin arrows) are observed.
Air-space consolidations and ground glass densities in the both lungs
are noted

Indian J Pediatr (2010) 77:10251027

selective bronchial occlusion, a short course of steroids, and


high-frequency ventilation [3]. Surgical intervention may
be considered for some persistent cases with resectable
expansile lesions causing pulmonary dysfunction, cardiovascular compromise, recurrent infections or recurrent
pneumothoraces, or for a definitive diagnosis in equivocal
cases [8].
This case demonstrates the natural history of PIE within
the course of pneumonia in a term infant without any
history of assisted ventilation. While many of such cases of
PIE may need active surgical management, some, such as
this case, may do well even with conservative management.
However this should come with a strict advise for close
clinical and radiological monitoring.

Contributions HSL; involved in patient management, literature


review and drafting of the manuscript. SAI; involved in the
radiological diagnosis and would be the guarantor.

Conflict of Interest None.

Role of Funding Source None.

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References
1. Macklin MT, Macklin CC. Malignant interstitial emphysema of the
lungs and mediastinum as an important occult complication in
many respiratory diseases and other conditions: an interpretation of
the clinical literature in the light of laboratory experiment.
Medicine. 1944;23:281358.
2. Stocker JT, Madewell JE. Persistent interstitial pulmonary emphysema: another complication of the respiratory distress syndrome.
Pediatrics. 1977;59:84757.
3. ODonovan D, Wearden M, Adams J. Unilateral pulmonary
interstitial emphysema following pneumonia in a preterm infant
successfully treated with prolonged bronchial intubation. Am J
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4. Boisset GF. Subpleural emphysema complicating staphylococcal
and other pneumonias. J Pediatr. 1972;81:25966.
5. Pursnani SK, Amodio JB, Guo H, Greco MA, Nadler EP. Localized
persistent interstitial pulmonary emphysema presenting as a
spontaneous tension pneumothorax in a full term infant. Pediatr
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6. Carey B. Neonatal air leaks: pneumothorax, pneumomediastinum,
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Netw. 1999;18:814.
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8. Jassal MS, Benson JE, Mogayzel Jr PJ. Spontaneous resolution of
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