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417
Pulmonary Pseudocysts Newborn Infants with Respiratory Distress Syndrome
Thomas and
A.
David
K
Twelve
2
Edwards1
.
neonates
are and described. all were
who developed
intrathoracic
cystlike
in
structures
(“pseudocysts”)
treated All infants with had assisted clinical ventilation. and radiographic Pulmonary respiratory interstitial distress emphysema syndromepre-
ceded the development of the pseudocysts in all cases. The pseudocysts appeared between 0 and 6 days of life (average, 2.4 days), and radiographically documented disappearance occurred in 10 patients at 3-18 days old (average, 7.0 days). Two patients had persisting pseudocysts at 8 and 25 days of age, and no subsequent radiographs. In no case did these structures persist symptomatically or require surgical intervention such as has been described with patients in other series. In two cases, abrupt disappearance of the pseudocysts was accompanied by increased extraalveolar gas in other locations. Eight patients developed radiographic bronchopulmonary dysplasia, which was severe in two of the cases.
Pulmonary respiratory
interstitial emphysema distress syndrome (ROS)
ventilation
[1 -3].
often
is the
and
An
are benign in that they reports these structures requiring
air
leaks,
extensive
[4-6].
of intrathoracic by Campbell [1
ultimately
identification
of other
because
compromise
formation scribed
radiographic
forerunner
mediastinum, tory
The
(PIE) is a well recognized in premature newborns who
]
regress did
surgical
because
can
it
and
pneumo-
cause
respira-
associated
with
PIE
is
or ‘ ‘ pseudocysts. ‘ ‘ As initially deby others [2, 7], such pseudocysts
and disappear not regress
resection
emphysema
phenomenon
structures observed
is important
as pneumothorax
interstitial
uncommon
cystlike and later
of PIE
such
complication of require assisted
but
spontaneously. However, instead continued to
or contributing
to the
patients’
in other enlarge,
demise
[8,
9]. This
disparity
amine Received vision
May
January
accepted
after
prompted
outcome
at our
describe
1 2 such
Subjects
and Methods
institution
patients
who
us to clinically who
were
developed
encountered
and
radiographically
pulmonary during
ex-
pseudocysts.
a 3 year
We
period.
re-
of Radiology,
Hospital, Address
University
225 Dickinson St. reprint requests
.
of Cali-
San Diego, to D. K.
CA Ed-
wards.
Between
Present
address:
University
of
Rochester,
NY 14642.
Department
Rochester,
AJR 133:417-421, 0361-803X/79/1333-0417 © American Roentgen
601
September Ray
.
January
1 975 .
Cane .
2
of
patients
9. 1979.
Department fornia 92103.
3, 1 979;
the
diagnosis
1979
$00.00 Society
Ave.,
.
findings of
for
RDS
very
.
and
of events
mild
sequence initial
disease
chest
film
up to 5 for
with
each scored increasing
was
were .
admitted .
to the .
Intensive .
in San Otego with a primary admitting Of these patients, i 2 demonstrated the
.
.
of intrathoracic cystlike and the nadiographs
the
newborns .
Hospital (RDS).
development were abstracted,
on
375
.
California syndrome
.
postnatal patients
1 978,
February
Nursery at University of of respiratory distress
of Pediatrics, Elmwood
and .
.
structures. The clinical were reviewed to identify
patient (tables as described severity.
records of these the radiographic
1 and 2). Radiographic elsewhere [10], with
severity a scone of 1
418
CLARKE
TABLE
1 : Radiographic
Severity
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P.otient
of
Age (days) Were
when pseudocysts
First
Reached Max:mum
Seen
1
5
41
2
3
3
3
3
4
65
14
109
4
21
11
4
1
12
4
TABLE
absent, + = present; disappearance not
Patient
1
PIE-’PM,
PIE-’PM--.pseudocyst
3 5
PIE, PM-.pseudocysts PIE-’pseudocysts PIE-.PT, PM, PP-’pseudocyts
6
PIE-+PT,
7
PM, PT-.PIE--.pseudocysts PlE-.pseudocysts--.PT--.PM PlE-.PT---pseudocysts PIE, PT-.pseudocysts PlE-.pseudocysts-.PT PIE-+PT--’pseudocyst
8 9 10
1 12 1
Left
at Last
Radiograph
79 33
Severe Mild
iO
+
+
-
-
1
19
+
+
-
+
.
3-4
18
+
+
-
+
5 8
+
-
-
.
5 3
+
+
+
-
Mild
223 25
+
+
+
+
Mild
61
+
+
+
+
+
-
+
-
+
+
+
+
Uncertain
9 18
18 15
1
8
Moderate
3
4-5
18
i-2
3
4-5
7
+
-
+
-
3
3-4
9
+
-
+
-
Mild Mild
44
16
+
+
+
+
Severe
3i
distress
9 15
1
syndrome;
BPD
bronchopuimonary
-
10
51
dyspiasia.
of Air Leaks
2 4
Right
Chest
of Air Leaks
Sequence
No
Left
Age (days)
documented
Sequence
2: Radiographic
Radiographic BPD
2
4
respiratory
=
Pneumolhoras
4-5
2 2 ADS
Emphy-
5
6
5
=
1979
5
10
3
4
Note . Radiographic
September
8
7
3
sema
1e#{128}:Ti:) Right
Quantify
i2
6
21
4
87
Disappeared
4
4
interstitial
Pseudocysts
6
4
3
4
AJR:133,
EDWARDS
Findings
ADS ( 1 -5)
No
AND
PP-apseudocysts
...
pseudocyst-.PP-PM,
pseudocyst
.r
, .,,
,
V4.’
Noti
-PIE
I,
pulmonary
=
or,i,diastinum.
PP
interstitial
emphysema;
PT
ipsilateral
=
pneumothorax;
PM
pneumoporicardium
=
Fig.
Results All all
patients
male.
were
920-2,960).
were
clinically
diagnosed
The
average
birthweight
All
patients
weeks
(range,
age 28-36).
sisted
ventilation
were
timated
gestational
(range,
28.2
average
cm 3.4
cm
Comparison leaks
water
of time
dium) the
showed patients
attempts cases
Ten
for
to have
or were
patient
and
two
died.
cally
well,
was
transferred
One
the
rest
or
were
patient
(patient
of
survivors well from
were
from
3), who
hospital
old. Antenopostenior size. Lucencies
age cally
structures
supine.
Right
on left over
The
the
pearance
was
at age
age
was
283
The The
with
penihilar silhou-
cardiac
not
second
onset
of PIE
patient
had
(patient
patient time
on
examined
80
superim9) died at
from had
which
he
radiographi-
of death,
pathologic
all doing
at age
with
arrest
at the
are
1 ) died
fatality
Neither
factors
31 -944);
hospital
and
such
examination.
in this
study
are
pre-
1 . All patients had ADS that was radiographto very severe (average 3.8 on a scale
interstitial
clini-
(range,
that
at prep-
survivors
the
(patient
identified
severity
9 days
days
a cardiopulmonary
radiographic
of
the
dysplasia
pseudocysts were
it is assumed
of
from
first
be resuscitated.
sented in table ically moderate increasing
follow-up;
discharged standpoint.
died.
apparent
In all as could aphos-
report
pneumonia.
not
to
average
bronchopulmonary
1 8 days
in not
this
who were a respiratory patients
posed
lost The
of severe
monary
discharged
to another
aration
of
pneumonia.
and
pital,
Three
subsequently survived.
could
ventilator
pseudocysts. as rapidly bacteriologically
the
was also
days
pneumopericar-
pressure
and he
Two
with
pressure.
relationship.
lowered
clinically
staphylococcal survived
for
2.4 hr aver-
appearance of air pseudocysts,
emphysema;
this
pressures
No
patients
temporal
es-
of 31 .4 and as-
pressure,
and
,
and
1 , 6 days at maximum
g (range,
average
expiratory
in a negative
interstitial
ventilator
peared
positive
of peak pressure with interstitial emphysema,
treated
tolerated.
with
end
positive
placed
to resolve
specifically
be
Maximum intermittent
no consistent were
1 731
examination intubation
pneumomediastinum
pneumothorax,
RDS,
having
at an average of ventilator pressures
initiated
water
(pulmonary
premature,
by physical Endotracheal
to 1 2 hr.)
birth
aged
were
as
was
1 -Patient
psudocysts (arrows) ette are artifactual.
from
was
5).
emphysema
pseudocysts
cystic
1 to
(PIE)
in the
at 1 .0 days structures
All
patients
that
affected
(range, on
initial
exhibited
preceded lung.
birth chest
The
the
ap-
average
to 4 days). film,
of pul-
and
No no
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AJR:133.
September
PULMONARY
1979
Fig.
2.-Patient
3, 4 days
old.
PSEUDOCYSTS
Anteroposterior
(A)
and
lateral
WITH
(B).
419
RDS
Pseudocysts
and
(arrows)
pneumomediaslinum.
.-
‘.{
&
o
.
,‘f, ; :
..
.,
I’.
,_.w.
Fig. 3.-Patient medial
6, 1 day old. Solitary
pseudocyst(arrow)
contralateral This
patient had pleural effusions staphylococcal pneumonia. In 1 1 cases, the pseudocysts thorax. In 1 0 of these 1 2) with a pseudocyst situated 1 -3).
with
pneumothorax.
primarily It was
not
consolidation appeared
in the
perihilar from
and the
in the
infrahilar
right
hem-
(patient were
regions
radiographs
were subpleural these structures
resolution
suggesting
cases, and in the one case on the left, the pseudocysts
apparent
not the pseudocysts patient (patient 4),
or
(figs.
whether
in location. appeared to
be
Fig. 4.-Patient is only case
or
In one clus-
began.
documented days (range, ing
25
5 had days;
and
asymptomatic
the pseudocysts
vanished
cysts
and
pneumothorax. In addition, (which, as described above,
2.4
day of life) reappeared pneumomediastinum,
in
table to
1 are
count
shadows of interstitial The average age days (range, at an average
approximate,
because
of
emphysema at appearance
birth to 6 days). The of 4.0 days (range
since overlapping
they
were margins
and other air leaks. of pseudocysts was
often
pseudocysts were largest 1 -1 4), after which time
mediastinum Ipsilateral
at right lung base in location.
resolution
was
were
on his last on his
last
available. radiograph
follow-up
visit
days, but no further radiographs were taken. of the pseudocysts appeared gradual (over at days) in 1 0 cases. However, in patients 6 and
1 1
difficult
perihilar
radiographs
pseudocysts
tered near the lung base (fig. 4). As the figures show, the pseudocysts were roughly spherical, and apparent radiographic diameter was 3-19 mm. The numbers of pseudo-
appearance PIE and
not
radiographic
no further
persisting he was
age 302 The shrinking least several at
Complete
(arrows)
in 1 0 cases, and occurred at an average of 7.1 3-18). Patient 3 leftthe hospital with diminish-
pseudocysts,
Patient at
4, 6 days old. Pseudocysts in which these structures were
abruptly
of, respectively: a pneumopericardium
on the PIE
in patient disappeared at
and same
preceded
with
the simultaneous
(patient 6) markedly increased and (patient 1 1 ) a large 6, the pseudocysts initially on the first
age 1 0 days together disappeared with the
with pneumo-
a
day. the
formation
of pseudocysts
in
420
CLARKE
all cases, precede
and in three cases pseudocyst formation.
PIE
was the only air leak to In the other patients the
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sequence of air leaks was more complex The average duration of radiographic days nary
(range, dysplasia
8-223). were
with
multiple
pulmonary
dysplasia
was
not
AND
(table 2). follow-up
Changes suggesting seen in eight patients. infiltrates,
and
was
49
bronchopulmoPatient 9 died
bronchopulmonary
EDWARDS
such
AJR:133,
pseudocysts
mate resolution structures were peared
limited
no
mise
described by Campbell [1 , by other authors [2, 3, 7-9]. tures have also been noted
that appear in the lungs of with other air leaks were first and were subsequently Radiographically similar in adults in association
mechanically assisted ventilation referred to these structures as
‘
[1 1 , 1 2]. ‘ pseudocysts,
noted strucwith
Campbell [11 ‘ ‘ suggesting
that they were formed by air from pulmonary interstitial emphysema (PIE) that collected beneath the visceral pleura. This is in keeping with the experimental work of MackIm and MackIm from
[1 3,
1 4],
ruptured
who
monary interstitium a pneumomediastinum One
of this
should experience
radiographically docyst air
along
dissection spaces
thesis
is that
prior the
demonstrated
ipsilateral
However,
(pneumothorax
and
into
of such
development of all 1 2 patients
of our
mediastinum)
prior
to pseu-
cases, also
before or simultaneously with the appearance cysts. This is not unexpected if one postulates of air leaks occurring at about the same time. Another implication that subpleural blebs common phenomenon
pul-
to rupture
PIE
in eight
of air in the
formation
be preceded by the supports this since
appearance.
leaks
the
perivascular
to the mediastinum, or pneumothorax.
implication
pseudocysts PIE; our
demonstrated
alveoli
other
of pseudoa multiplicity
seem
integral
that
momediastinum docysts
in most
that do not radiographs. This
of
speculation [3]:
to In two
our
of the extensive
massive ipsilateral other, ipsilateral dently mation.
form of
disappearance of
are
patients long
Weller
pseudocyst
pearance
such
persist
latter
finding
tinum.
pseudocysts of the sequence and pneumothorax,
part
a relative
are
evanescent
enough
the
to
be
is supported apparent
leak
or and
with ipsilateral is of prognostic
on
sporadic by a of a
because
7].
However,
to expand
contributing
respiratory
to patient
demise
cases
PIE.
Of
the
clinically
pseudocyst exhibited
resolution pseudocysts
described,
with
were
not,
23
(64%)
radiographic
Unfortunately
is a suggestion
that
insufficient adequately; [9] lists
data
are
forwhen
patients
to test
series by of pathologic
who
this
hy-
Stocker and diagnosis,
would be later than radiographic diagnosis. that resolved spontaneously, Campbell [1
Of the observed
]
the appearance of pseudocysts in his five patients ‘ ‘before 72 hours of age’ ‘ ; pseudocysts appeared at 1 8 days of life in a patient of Fletcher et al. [2], at an average of 4 days in the series of Harris [7], and at an average of 2.4 days in our series. The average time of appearance was 1 9 days in patients who required surgical resection reported by Magilet
al.
In our
[8].
These
report
and
by examining
for
suggest
in others,
where
the
formation is available, the pseudocysts that and
data
a trend
pseudocysts being more apt more information is required.
the
curious
later-
and
association
pseudocysts
there resolve
toward
to persist
grow,
is found
appeared.
When
in-
is an impressive tendency for to be located in the right chest,
pseudocysts
that
persist
and
enlarge
to
be
situated on the left. This peculiar correspondence was examined by chi-square test, with results that were significant at levels between p < 0.01 and p < 0.001 , depending on
and oppose it in the left. bronchopulmonary dysplasia
pneumothorax importance
because cases
earlier in their did not clear.
available
the large only the time
in the right lung Radiographic
and in the of Harris [7] evi-
of (36%) clinical
than
the
developed pseudocysts patients whose pseudocysts
may be spurious, exist that favor
ap-
apparently
documentation
progression. between the patients whose pseubenign and those whose pseudo-
there
cleared than the
been
(92%). particu-
in most of these; 1 3 cases that persisted with adverse
exhibiting symptomatic In seeking differences docysts were clinically ultimately courses
has
was RDS, in 33 cases were observed as well,
how
case,
or necessitatthat
effects. This number is undoubtedly exaggerated self-limited cases are less apt to be reported
cysts
the
compro-
36 cases of pseudocyst formation in been described [1 -3, 7-9]. The most
larly
recovered
other
reports,
removal [8, 9], a complication diffuse unilateral PIE as well [4-6]. report, have
of
in other until
in and
pseudocysts.
experience
with prognosis tomic differences
in one
ob-
required,
of the
the
showed
abrupt
be
spontaneously;
therapy
pneumomediassimultaneous
elsewhere:
PIE and pneumopericardium, pneumothorax. A patient
behaved similarly, This observation
not
phenomena
seen
radiography
pseudocyst(s) air
and
to some extent decompression
a pneumothorax cases,
rarity
of events leading to pneuor possibly that pseu-
should
to resolve specific been
disease air leaks
appearing but clearly
5% in the series [1 ]. Therefore, it
patients
In our series, these in that their growth ap-
has
common underlying In all cases other
ner
The incidence was in that of Campbell
2,
,
continued
Including our newborn infants
which cases
of the MackIm mechanism might be or pseudocysts should be a fairly in newborns with air leaks. However,
[1
occurred,
with RDS (about 3%). of Harris [7], and 6% would
to die
well
ing surgical noted with
this is not true, at least in terms of radiographically visible pseudocysts. Whereas the incidence of PIE, pneumothorax and pneumomediastinum, may approach 40% [1 5], the incidence of pseudocysts in our series was 1 2 of 375 infants
an
appeared character
as
pothesis Madewell
occurred
tended
or other
pseudocysts cystic structures in association
they
surgery
benign
authors
Intrathoracic newborn infants
the
of the pseudocysts. relatively benign and
was
patient
This
Discussion
noted;
1979
served closely for development of more extensive air leaks. Of greater prognostic significance is the question of ulti-
no case
apparent.
are
September
the data
were
interpreted.
This
large
number
of our
patients;
eight
patients
(67%),
and
strate
this
disease
were
the not
association
of laterality
or perhaps indeed anaclearance of pseudocysts
the
appeared
diagnosis
patients radiographed
who
was did beyond
not
in a
made
in
demon1 8 days
AJR:133,
of
life,
so
dence RDS,
Downloaded from www.ajronline.org by Central Michigan Univ on 10/18/15 from IP address 141.209.100.60. Copyright ARRS. For personal use only; all rights reserved
September
the
incidence
is considerably even among
[1 0].
It is likely
who
usually
extensive than do
that suffer
exposure newborns emphysema
ing
in the
sia by Watts series tended scores,
have
ventilatory
with
the
could
be
even
greater.
those
higher than that usually treated with assisted
the
group
from
of patients
multiple
air
with
itself
development
require
severe of these
RDS, factors
bronchopulmonary
more
ventilation Pulmonary as a predisposdysplain our Apgar
and require early are associated dysplasia
[10].
In summary, the prognostic implications of such pseudocysts are: (1 ) they can apparently rupture, causing other intrathoracic air leaks, (2) they are associated with a high incidence of chronic lung disease (bronchopulmonary dysplasia), and (3) they can progressively expand to a lifethreatening
extent,
although
this
behavior
is very
uncom-
WITH
cation
mci-
seen with ventilation
Additionally, the patients and immature, have low
radiographically assistance; all of
implicated
This
of bronchopulmonary
et al. [1 6]. to be small
occurrence
was
PSEUDOCYSTS
pseudocysts,
leaks,
to oxygen and mechanical without these complications.
interstitial factor
PULMONARY
1979
of
assisted
Fletcher
1 0.
Edwards
OK,
penience
i i
1 2.
.
with
926, 14.
tial
BO, Outerbridge
emphysema
in the
interstitial disease.
intersti21:273-
279, i970 3. Weller MH: The roentgenographic course and complications of hyaline membrane disease. Pediatr Clin North Am 20:38i 406, 1973 4. Drew JH, Landau LI, Acton CM, Kent M, Campbell PE: Pulmonary interstitial emphysema requiring lobectomy: compli-
tation
i 5.
1 6.
WM,
Northway
WH
bronchopulmonany
Jr:
Twelve
years’
Pediatrics
dysplasia.
ex-
59:
in
sheaths Arch
of pulmonic Intern
Med
blood 64:9i 3-
i939 MT,
lungs
in many
emphysema: a comAJR i i 0:449-456,
EW, Dunbar JS: Pulmonary newborn. J Can Assoc Radio!
53:424-426,
i974
MackIm the
RE: Intrapulmonary of hyaline membrane
Dyer
CC: Transport of air along from alveoli to mediastinum.
MackIm
vessels
Campbell plication 1970 2. Fletcher
Child
839-846, 1977 Rohlfing BM, Webb WA, Schlobohm AM: Ventilator-related extra-alveolar air in adults. Radiology 1 2i :25-3i , 1976 Westcott JL, Cole SR: Interstitial pulmonary emphysema children and adults: roentgenographic features. Radiology iii:367-378,
.
Dis
60, Outerbnidge EW, Youssef 5, Bolande RP: Pulmonary interstitial emphysema in a newborn infant treated by lobectomy. Pediatrics 54:808-81 1 , 1974 6. Bauen CR, Brennan MJ, Doyle C, Poole CA: Surgical resection for pulmonary interstitial emphysema in the newborn infant. J Pediatr 93:656-661 , 1978 7. Harris H: Pulmonary pseudocysts in the newborn infant. Pediatrics 59:199-204, 1977 8. Magilner AD, Capitanio MA, Wertheimer I, Bunko H: Persistent localized intrapulmonary interstitial emphysema: an obsenvation in three infants. Radiology 1 1 i :379-384, i 974 9. Stocker JT, Madewell JE: Persistent interstitial pulmonary emphysema: another complication of the respiratory distress syndrome. Pediatrics 59:847-857, 1977 5.
1 3.
1
Arch
ventilation.
1978
mon.
REFERENCES
421
RDS
and
MackIm
respiratory of
the
CC:
mediastinum clinical
diseases literature
Malignant
interstitial
as an important
and other in
the
light
emphysema occult
of
complication
conditions: an interpreof laboratory expeni-
ment. Medicine 23:281 -358, 1944 Berg TJ, Pagtakhan RO, Reed MH, Langston C, Chernick V: Bronchopulmonary dysplasia and lung rupture in hyaline membrane disease: influence of continuous distending pressure. Pediatrics 55:5i-54, 1975 Watts JL, Aniagno AL, Brady JP: Chronic pulmonary disease in neonates after artificial ventilation: distribution of ventilation and pulmonary interstitial emphysema. Pediatrics 60:27328i,
i977
417
Pulmonary Pseudocysts Newborn Infants with Respiratory Distress Syndrome
Thomas and
A.
David
K
Twelve
2
Edwards1
.
neonates
are and described. all were
who developed
intrathoracic
cystlike
in
structures
(“pseudocysts”)
treated All infants with had assisted clinical ventilation. and radiographic Pulmonary respiratory interstitial distress emphysema syndromepre-
ceded the development of the pseudocysts in all cases. The pseudocysts appeared between 0 and 6 days of life (average, 2.4 days), and radiographically documented disappearance occurred in 10 patients at 3-18 days old (average, 7.0 days). Two patients had persisting pseudocysts at 8 and 25 days of age, and no subsequent radiographs. In no case did these structures persist symptomatically or require surgical intervention such as has been described with patients in other series. In two cases, abrupt disappearance of the pseudocysts was accompanied by increased extraalveolar gas in other locations. Eight patients developed radiographic bronchopulmonary dysplasia, which was severe in two of the cases.
Pulmonary respiratory
interstitial emphysema distress syndrome (ROS)
ventilation
[1 -3].
often
is the
and
An
are benign in that they reports these structures requiring
air
leaks,
extensive
[4-6].
of intrathoracic by Campbell [1
ultimately
identification
of other
because
compromise
formation scribed
radiographic
forerunner
mediastinum, tory
The
(PIE) is a well recognized in premature newborns who
]
regress did
surgical
because
can
it
and
pneumo-
cause
respira-
associated
with
PIE
is
or ‘ ‘ pseudocysts. ‘ ‘ As initially deby others [2, 7], such pseudocysts
and disappear not regress
resection
emphysema
phenomenon
structures observed
is important
as pneumothorax
interstitial
uncommon
cystlike and later
of PIE
such
complication of require assisted
but
spontaneously. However, instead continued to
or contributing
to the
patients’
in other enlarge,
demise
[8,
9]. This
disparity
amine Received vision
May
January
accepted
after
prompted
outcome
at our
describe
1 2 such
Subjects
and Methods
institution
patients
who
us to clinically who
were
developed
encountered
and
radiographically
pulmonary during
ex-
pseudocysts.
a 3 year
We
period.
re-
of Radiology,
Hospital, Address
University
225 Dickinson St. reprint requests
.
of Cali-
San Diego, to D. K.
CA Ed-
wards.
Between
Present
address:
University
of
Rochester,
NY 14642.
Department
Rochester,
AJR 133:417-421, 0361-803X/79/1333-0417 © American Roentgen
601
September Ray
.
January
1 975 .
Cane .
2
of
patients
9. 1979.
Department fornia 92103.
3, 1 979;
the
diagnosis
1979
$00.00 Society
Ave.,
.
findings of
for
RDS
very
.
and
of events
mild
sequence initial
disease
chest
film
up to 5 for
with
each scored increasing
was
were .
admitted .
to the .
Intensive .
in San Otego with a primary admitting Of these patients, i 2 demonstrated the
.
.
of intrathoracic cystlike and the nadiographs
the
newborns .
Hospital (RDS).
development were abstracted,
on
375
.
California syndrome
.
postnatal patients
1 978,
February
Nursery at University of of respiratory distress
of Pediatrics, Elmwood
and .
.
structures. The clinical were reviewed to identify
patient (tables as described severity.
records of these the radiographic
1 and 2). Radiographic elsewhere [10], with
severity a scone of 1
418
CLARKE
TABLE
1 : Radiographic
Severity
Downloaded from www.ajronline.org by Central Michigan Univ on 10/18/15 from IP address 141.209.100.60. Copyright ARRS. For personal use only; all rights reserved
P.otient
of
Age (days) Were
when pseudocysts
First
Reached Max:mum
Seen
1
5
41
2
3
3
3
3
4
65
14
109
4
21
11
4
1
12
4
TABLE
absent, + = present; disappearance not
Patient
1
PIE-’PM,
PIE-’PM--.pseudocyst
3 5
PIE, PM-.pseudocysts PIE-’pseudocysts PIE-.PT, PM, PP-’pseudocyts
6
PIE-+PT,
7
PM, PT-.PIE--.pseudocysts PlE-.pseudocysts--.PT--.PM PlE-.PT---pseudocysts PIE, PT-.pseudocysts PlE-.pseudocysts-.PT PIE-+PT--’pseudocyst
8 9 10
1 12 1
Left
at Last
Radiograph
79 33
Severe Mild
iO
+
+
-
-
1
19
+
+
-
+
.
3-4
18
+
+
-
+
5 8
+
-
-
.
5 3
+
+
+
-
Mild
223 25
+
+
+
+
Mild
61
+
+
+
+
+
-
+
-
+
+
+
+
Uncertain
9 18
18 15
1
8
Moderate
3
4-5
18
i-2
3
4-5
7
+
-
+
-
3
3-4
9
+
-
+
-
Mild Mild
44
16
+
+
+
+
Severe
3i
distress
9 15
1
syndrome;
BPD
bronchopuimonary
-
10
51
dyspiasia.
of Air Leaks
2 4
Right
Chest
of Air Leaks
Sequence
No
Left
Age (days)
documented
Sequence
2: Radiographic
Radiographic BPD
2
4
respiratory
=
Pneumolhoras
4-5
2 2 ADS
Emphy-
5
6
5
=
1979
5
10
3
4
Note . Radiographic
September
8
7
3
sema
1e#{128}:Ti:) Right
Quantify
i2
6
21
4
87
Disappeared
4
4
interstitial
Pseudocysts
6
4
3
4
AJR:133,
EDWARDS
Findings
ADS ( 1 -5)
No
AND
PP-apseudocysts
...
pseudocyst-.PP-PM,
pseudocyst
.r
, .,,
,
V4.’
Noti
-PIE
I,
pulmonary
=
or,i,diastinum.
PP
interstitial
emphysema;
PT
ipsilateral
=
pneumothorax;
PM
pneumoporicardium
=
Fig.
Results All all
patients
male.
were
920-2,960).
were
clinically
diagnosed
The
average
birthweight
All
patients
weeks
(range,
age 28-36).
sisted
ventilation
were
timated
gestational
(range,
28.2
average
cm 3.4
cm
Comparison leaks
water
of time
dium) the
showed patients
attempts cases
Ten
for
to have
or were
patient
and
two
died.
cally
well,
was
transferred
One
the
rest
or
were
patient
(patient
of
survivors well from
were
from
3), who
hospital
old. Antenopostenior size. Lucencies
age cally
structures
supine.
Right
on left over
The
the
pearance
was
at age
age
was
283
The The
with
penihilar silhou-
cardiac
not
second
onset
of PIE
patient
had
(patient
patient time
on
examined
80
superim9) died at
from had
which
he
radiographi-
of death,
pathologic
all doing
at age
with
arrest
at the
are
1 ) died
fatality
Neither
factors
31 -944);
hospital
and
such
examination.
in this
study
are
pre-
1 . All patients had ADS that was radiographto very severe (average 3.8 on a scale
interstitial
clini-
(range,
that
at prep-
survivors
the
(patient
identified
severity
9 days
days
a cardiopulmonary
radiographic
of
the
dysplasia
pseudocysts were
it is assumed
of
from
first
be resuscitated.
sented in table ically moderate increasing
follow-up;
discharged standpoint.
died.
apparent
In all as could aphos-
report
pneumonia.
not
to
average
bronchopulmonary
1 8 days
in not
this
who were a respiratory patients
posed
lost The
of severe
monary
discharged
to another
aration
of
pneumonia.
and
pital,
Three
subsequently survived.
could
ventilator
pseudocysts. as rapidly bacteriologically
the
was also
days
pneumopericar-
pressure
and he
Two
with
pressure.
relationship.
lowered
clinically
staphylococcal survived
for
2.4 hr aver-
appearance of air pseudocysts,
emphysema;
this
pressures
No
patients
temporal
es-
of 31 .4 and as-
pressure,
and
,
and
1 , 6 days at maximum
g (range,
average
expiratory
in a negative
interstitial
ventilator
peared
positive
of peak pressure with interstitial emphysema,
treated
tolerated.
with
end
positive
placed
to resolve
specifically
be
Maximum intermittent
no consistent were
1 731
examination intubation
pneumomediastinum
pneumothorax,
RDS,
having
at an average of ventilator pressures
initiated
water
(pulmonary
premature,
by physical Endotracheal
to 1 2 hr.)
birth
aged
were
as
was
1 -Patient
psudocysts (arrows) ette are artifactual.
from
was
5).
emphysema
pseudocysts
cystic
1 to
(PIE)
in the
at 1 .0 days structures
All
patients
that
affected
(range, on
initial
exhibited
preceded lung.
birth chest
The
the
ap-
average
to 4 days). film,
of pul-
and
No no
Downloaded from www.ajronline.org by Central Michigan Univ on 10/18/15 from IP address 141.209.100.60. Copyright ARRS. For personal use only; all rights reserved
AJR:133.
September
PULMONARY
1979
Fig.
2.-Patient
3, 4 days
old.
PSEUDOCYSTS
Anteroposterior
(A)
and
lateral
WITH
(B).
419
RDS
Pseudocysts
and
(arrows)
pneumomediaslinum.
.-
‘.{
&
o
.
,‘f, ; :
..
.,
I’.
,_.w.
Fig. 3.-Patient medial
6, 1 day old. Solitary
pseudocyst(arrow)
contralateral This
patient had pleural effusions staphylococcal pneumonia. In 1 1 cases, the pseudocysts thorax. In 1 0 of these 1 2) with a pseudocyst situated 1 -3).
with
pneumothorax.
primarily It was
not
consolidation appeared
in the
perihilar from
and the
in the
infrahilar
right
hem-
(patient were
regions
radiographs
were subpleural these structures
resolution
suggesting
cases, and in the one case on the left, the pseudocysts
apparent
not the pseudocysts patient (patient 4),
or
(figs.
whether
in location. appeared to
be
Fig. 4.-Patient is only case
or
In one clus-
began.
documented days (range, ing
25
5 had days;
and
asymptomatic
the pseudocysts
vanished
cysts
and
pneumothorax. In addition, (which, as described above,
2.4
day of life) reappeared pneumomediastinum,
in
table to
1 are
count
shadows of interstitial The average age days (range, at an average
approximate,
because
of
emphysema at appearance
birth to 6 days). The of 4.0 days (range
since overlapping
they
were margins
and other air leaks. of pseudocysts was
often
pseudocysts were largest 1 -1 4), after which time
mediastinum Ipsilateral
at right lung base in location.
resolution
was
were
on his last on his
last
available. radiograph
follow-up
visit
days, but no further radiographs were taken. of the pseudocysts appeared gradual (over at days) in 1 0 cases. However, in patients 6 and
1 1
difficult
perihilar
radiographs
pseudocysts
tered near the lung base (fig. 4). As the figures show, the pseudocysts were roughly spherical, and apparent radiographic diameter was 3-19 mm. The numbers of pseudo-
appearance PIE and
not
radiographic
no further
persisting he was
age 302 The shrinking least several at
Complete
(arrows)
in 1 0 cases, and occurred at an average of 7.1 3-18). Patient 3 leftthe hospital with diminish-
pseudocysts,
Patient at
4, 6 days old. Pseudocysts in which these structures were
abruptly
of, respectively: a pneumopericardium
on the PIE
in patient disappeared at
and same
preceded
with
the simultaneous
(patient 6) markedly increased and (patient 1 1 ) a large 6, the pseudocysts initially on the first
age 1 0 days together disappeared with the
with pneumo-
a
day. the
formation
of pseudocysts
in
420
CLARKE
all cases, precede
and in three cases pseudocyst formation.
PIE
was the only air leak to In the other patients the
Downloaded from www.ajronline.org by Central Michigan Univ on 10/18/15 from IP address 141.209.100.60. Copyright ARRS. For personal use only; all rights reserved
sequence of air leaks was more complex The average duration of radiographic days nary
(range, dysplasia
8-223). were
with
multiple
pulmonary
dysplasia
was
not
AND
(table 2). follow-up
Changes suggesting seen in eight patients. infiltrates,
and
was
49
bronchopulmoPatient 9 died
bronchopulmonary
EDWARDS
such
AJR:133,
pseudocysts
mate resolution structures were peared
limited
no
mise
described by Campbell [1 , by other authors [2, 3, 7-9]. tures have also been noted
that appear in the lungs of with other air leaks were first and were subsequently Radiographically similar in adults in association
mechanically assisted ventilation referred to these structures as
‘
[1 1 , 1 2]. ‘ pseudocysts,
noted strucwith
Campbell [11 ‘ ‘ suggesting
that they were formed by air from pulmonary interstitial emphysema (PIE) that collected beneath the visceral pleura. This is in keeping with the experimental work of MackIm and MackIm from
[1 3,
1 4],
ruptured
who
monary interstitium a pneumomediastinum One
of this
should experience
radiographically docyst air
along
dissection spaces
thesis
is that
prior the
demonstrated
ipsilateral
However,
(pneumothorax
and
into
of such
development of all 1 2 patients
of our
mediastinum)
prior
to pseu-
cases, also
before or simultaneously with the appearance cysts. This is not unexpected if one postulates of air leaks occurring at about the same time. Another implication that subpleural blebs common phenomenon
pul-
to rupture
PIE
in eight
of air in the
formation
be preceded by the supports this since
appearance.
leaks
the
perivascular
to the mediastinum, or pneumothorax.
implication
pseudocysts PIE; our
demonstrated
alveoli
other
of pseudoa multiplicity
seem
integral
that
momediastinum docysts
in most
that do not radiographs. This
of
speculation [3]:
to In two
our
of the extensive
massive ipsilateral other, ipsilateral dently mation.
form of
disappearance of
are
patients long
Weller
pseudocyst
pearance
such
persist
latter
finding
tinum.
pseudocysts of the sequence and pneumothorax,
part
a relative
are
evanescent
enough
the
to
be
is supported apparent
leak
or and
with ipsilateral is of prognostic
on
sporadic by a of a
because
7].
However,
to expand
contributing
respiratory
to patient
demise
cases
PIE.
Of
the
clinically
pseudocyst exhibited
resolution pseudocysts
described,
with
were
not,
23
(64%)
radiographic
Unfortunately
is a suggestion
that
insufficient adequately; [9] lists
data
are
forwhen
patients
to test
series by of pathologic
who
this
hy-
Stocker and diagnosis,
would be later than radiographic diagnosis. that resolved spontaneously, Campbell [1
Of the observed
]
the appearance of pseudocysts in his five patients ‘ ‘before 72 hours of age’ ‘ ; pseudocysts appeared at 1 8 days of life in a patient of Fletcher et al. [2], at an average of 4 days in the series of Harris [7], and at an average of 2.4 days in our series. The average time of appearance was 1 9 days in patients who required surgical resection reported by Magilet
al.
In our
[8].
These
report
and
by examining
for
suggest
in others,
where
the
formation is available, the pseudocysts that and
data
a trend
pseudocysts being more apt more information is required.
the
curious
later-
and
association
pseudocysts
there resolve
toward
to persist
grow,
is found
appeared.
When
in-
is an impressive tendency for to be located in the right chest,
pseudocysts
that
persist
and
enlarge
to
be
situated on the left. This peculiar correspondence was examined by chi-square test, with results that were significant at levels between p < 0.01 and p < 0.001 , depending on
and oppose it in the left. bronchopulmonary dysplasia
pneumothorax importance
because cases
earlier in their did not clear.
available
the large only the time
in the right lung Radiographic
and in the of Harris [7] evi-
of (36%) clinical
than
the
developed pseudocysts patients whose pseudocysts
may be spurious, exist that favor
ap-
apparently
documentation
progression. between the patients whose pseubenign and those whose pseudo-
there
cleared than the
been
(92%). particu-
in most of these; 1 3 cases that persisted with adverse
exhibiting symptomatic In seeking differences docysts were clinically ultimately courses
has
was RDS, in 33 cases were observed as well,
how
case,
or necessitatthat
effects. This number is undoubtedly exaggerated self-limited cases are less apt to be reported
cysts
the
compro-
36 cases of pseudocyst formation in been described [1 -3, 7-9]. The most
larly
recovered
other
reports,
removal [8, 9], a complication diffuse unilateral PIE as well [4-6]. report, have
of
in other until
in and
pseudocysts.
experience
with prognosis tomic differences
in one
ob-
required,
of the
the
showed
abrupt
be
spontaneously;
therapy
pneumomediassimultaneous
elsewhere:
PIE and pneumopericardium, pneumothorax. A patient
behaved similarly, This observation
not
phenomena
seen
radiography
pseudocyst(s) air
and
to some extent decompression
a pneumothorax cases,
rarity
of events leading to pneuor possibly that pseu-
should
to resolve specific been
disease air leaks
appearing but clearly
5% in the series [1 ]. Therefore, it
patients
In our series, these in that their growth ap-
has
common underlying In all cases other
ner
The incidence was in that of Campbell
2,
,
continued
Including our newborn infants
which cases
of the MackIm mechanism might be or pseudocysts should be a fairly in newborns with air leaks. However,
[1
occurred,
with RDS (about 3%). of Harris [7], and 6% would
to die
well
ing surgical noted with
this is not true, at least in terms of radiographically visible pseudocysts. Whereas the incidence of PIE, pneumothorax and pneumomediastinum, may approach 40% [1 5], the incidence of pseudocysts in our series was 1 2 of 375 infants
an
appeared character
as
pothesis Madewell
occurred
tended
or other
pseudocysts cystic structures in association
they
surgery
benign
authors
Intrathoracic newborn infants
the
of the pseudocysts. relatively benign and
was
patient
This
Discussion
noted;
1979
served closely for development of more extensive air leaks. Of greater prognostic significance is the question of ulti-
no case
apparent.
are
September
the data
were
interpreted.
This
large
number
of our
patients;
eight
patients
(67%),
and
strate
this
disease
were
the not
association
of laterality
or perhaps indeed anaclearance of pseudocysts
the
appeared
diagnosis
patients radiographed
who
was did beyond
not
in a
made
in
demon1 8 days
AJR:133,
of
life,
so
dence RDS,
Downloaded from www.ajronline.org by Central Michigan Univ on 10/18/15 from IP address 141.209.100.60. Copyright ARRS. For personal use only; all rights reserved
September
the
incidence
is considerably even among
[1 0].
It is likely
who
usually
extensive than do
that suffer
exposure newborns emphysema
ing
in the
sia by Watts series tended scores,
have
ventilatory
with
the
could
be
even
greater.
those
higher than that usually treated with assisted
the
group
from
of patients
multiple
air
with
itself
development
require
severe of these
RDS, factors
bronchopulmonary
more
ventilation Pulmonary as a predisposdysplain our Apgar
and require early are associated dysplasia
[10].
In summary, the prognostic implications of such pseudocysts are: (1 ) they can apparently rupture, causing other intrathoracic air leaks, (2) they are associated with a high incidence of chronic lung disease (bronchopulmonary dysplasia), and (3) they can progressively expand to a lifethreatening
extent,
although
this
behavior
is very
uncom-
WITH
cation
mci-
seen with ventilation
Additionally, the patients and immature, have low
radiographically assistance; all of
implicated
This
of bronchopulmonary
et al. [1 6]. to be small
occurrence
was
PSEUDOCYSTS
pseudocysts,
leaks,
to oxygen and mechanical without these complications.
interstitial factor
PULMONARY
1979
of
assisted
Fletcher
1 0.
Edwards
OK,
penience
i i
1 2.
.
with
926, 14.
tial
BO, Outerbridge
emphysema
in the
interstitial disease.
intersti21:273-
279, i970 3. Weller MH: The roentgenographic course and complications of hyaline membrane disease. Pediatr Clin North Am 20:38i 406, 1973 4. Drew JH, Landau LI, Acton CM, Kent M, Campbell PE: Pulmonary interstitial emphysema requiring lobectomy: compli-
tation
i 5.
1 6.
WM,
Northway
WH
bronchopulmonany
Jr:
Twelve
years’
Pediatrics
dysplasia.
ex-
59:
in
sheaths Arch
of pulmonic Intern
Med
blood 64:9i 3-
i939 MT,
lungs
in many
emphysema: a comAJR i i 0:449-456,
EW, Dunbar JS: Pulmonary newborn. J Can Assoc Radio!
53:424-426,
i974
MackIm the
RE: Intrapulmonary of hyaline membrane
Dyer
CC: Transport of air along from alveoli to mediastinum.
MackIm
vessels
Campbell plication 1970 2. Fletcher
Child
839-846, 1977 Rohlfing BM, Webb WA, Schlobohm AM: Ventilator-related extra-alveolar air in adults. Radiology 1 2i :25-3i , 1976 Westcott JL, Cole SR: Interstitial pulmonary emphysema children and adults: roentgenographic features. Radiology iii:367-378,
.
Dis
60, Outerbnidge EW, Youssef 5, Bolande RP: Pulmonary interstitial emphysema in a newborn infant treated by lobectomy. Pediatrics 54:808-81 1 , 1974 6. Bauen CR, Brennan MJ, Doyle C, Poole CA: Surgical resection for pulmonary interstitial emphysema in the newborn infant. J Pediatr 93:656-661 , 1978 7. Harris H: Pulmonary pseudocysts in the newborn infant. Pediatrics 59:199-204, 1977 8. Magilner AD, Capitanio MA, Wertheimer I, Bunko H: Persistent localized intrapulmonary interstitial emphysema: an obsenvation in three infants. Radiology 1 1 i :379-384, i 974 9. Stocker JT, Madewell JE: Persistent interstitial pulmonary emphysema: another complication of the respiratory distress syndrome. Pediatrics 59:847-857, 1977 5.
1 3.
1
Arch
ventilation.
1978
mon.
REFERENCES
421
RDS
and
MackIm
respiratory of
the
CC:
mediastinum clinical
diseases literature
Malignant
interstitial
as an important
and other in
the
light
emphysema occult
of
complication
conditions: an interpreof laboratory expeni-
ment. Medicine 23:281 -358, 1944 Berg TJ, Pagtakhan RO, Reed MH, Langston C, Chernick V: Bronchopulmonary dysplasia and lung rupture in hyaline membrane disease: influence of continuous distending pressure. Pediatrics 55:5i-54, 1975 Watts JL, Aniagno AL, Brady JP: Chronic pulmonary disease in neonates after artificial ventilation: distribution of ventilation and pulmonary interstitial emphysema. Pediatrics 60:27328i,
i977
