Fetal Diagn Ther 1998;13:325–333 Received: December 22, 1997

Accepted after revision: July 24, 1998

Yves Aubard a
Isabelle Derouineau a
Primary Fetal Hydrothorax:
Véronique Aubard a
Valerie Chalifour a
A Literature Review and Proposed
Pierre-Marie Preux b Antenatal Clinical Strategy
a Department of Obstetrics and Gynecology,
CHU Dupuytren and
b Laboratory of Biostatistics, Faculty of
Medicine, University of Limoges, France

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Key Words Abstract

Hydrothorax, fetal Objective: To investigate the prognostic factors for primitive fetal hydrotho-
Thoracocentesis rax (PFHT) and propose a clinical strategy based on systematic literature
Pleuroamniotic shunting review. Methods: We reviewed 64 articles describing 204 cases of PFHT. For
each case we focused on 11 criteria. We investigated prognostic factors in the
89 cases where no in utero treatment was undertaken. We also studied the
impact of different in utero treatments on the evolution of PFHT. Results:
We have found 4 factors correlated with the course of PFHT: the presence of
hydrops, gestational age at time of birth, the unilateral or bilateral nature of
the effusion, and the occurrence of spontaneous resolution. With multivariate
analysis, only hydrops remained determinant as a prognostic factor. Conclu-
sions: Studies such as ours, reviewing case reports or series, are subject to the
biases of literature underreporting of therapeutic failures or nonintervention.
However (with the best available data) we propose a clinical approach to
PFHT discovered in utero.
OOOOOOOOOOOOOOOOOOOOOO

Primary fetal hydrothorax (PFHT), also termed chylo- Methods

thorax, is an intrathoracic effusion arising in the fetus,
which may occur uni- or bilaterally. This anomaly is rare
occurring in 1 in 10,000–15,000 pregnancies [1, 2]. PFHT
can have a highly variable clinical course from sponta-
neous regression and survival of the infant without se-
quelae to fetal or neonatal death in some cases with com-
pressive features. Given this rarity and clinical variabili-
ty, the optimal treatment for this condition has not yet
been codified. We have conducted a literature review on
this subject with the goal of determining a logical clinical
approach from the time the discovery in utero of PFHT is
made (fig. 1).
Our research covered the cases of primary fetal hydrothorax pub-
lished in French or English between 1977 and 1996. From all
reported cases, we only retained those publications where the diagno-
sis of PFHT was certain, eliminating all cases of secondary hydrotho-
rax or where the cause of the hydrothorax was not certain. Cases
reported more than once by the same team in different publications
were counted only once. We verified that individually reported cases
were not recounted in the larger case series. To further avoid multiple
reporting of the same case, we excluded conference reports as a
source.
For each included case, we focused on the following criteria: tech-
nique of PFHT detection, gestational age at time of discovery, speci-
fication of uni- or bilaterality, presence of fetal hydrops, presence of
polyhydramnios, fetal gender, clinical course of the PFHT, descrip-
tion of interventions undertaken, gestational age at delivery, neona-
tal outcome, and information obtained through autopsy.

© 1999 S. Karger AG, Basel Yves Aubard

ABC 1015–3837/98/0136–0325$17.50/0 Service de gynécologie-obstétrique – CHU Dupuytren
Fax + 41 61 306 12 34 2, avenue Martin Luther King
E-Mail karger@karger.ch Accessible online at: F–87000 Limoges (France)
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www.karger.com http://BioMedNet.com/karger Tel. +33 5 55 05 61 17, Fax +33 5 55 05 66 67, E-Mail yaubard@compuserve.com
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 Fig. 1. Clinical algorithm in the event of discovery of a fetal hydrothorax. TS = Thoracocentesis.

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 The study of prognostic factors was limited to the group of
patients who did not undergo treatment in utero. The comparison of
means was performed by the unpaired t test for quantitative values
(gestational age at time of discovery, gestational age at birth), and the
¯2 test was utilized for comparison of qualitative values (uni- or bilat-
eral appearance, presence of hydrops or polyhydramnios, fetal gen-
der). The odds ratio was determined by a logistic model using uni-
variable and multivariable step-down regression. The degree of sig-
nificance was 0.05 for each of the results.
Results of Literature Review
Sixty-four publications describing a total of 204 cases
of PFHT met the above criteria and were analyzed [1–64].
The therapeutic approach varied considerably among
cases: in 89 cases, no intrauterine intervention was per-
formed, in 78 cases a pleuroamniotic shunt was placed, in
29 cases one or more thoracocenteses were performed, in
1 case a pleurocutaneous shunt was placed, and in 6 cases
there was a medical termination of the pregnancy. We
were particularly interested in the 198 cases where the
pregnancy was not terminated.
Discovery of PFHT is consistently through echogra-
phy. In the 108 cases where the reason for performing
echography was stated, 63 were performed for a workup
of polyhydramnios (58%), 40 were systematic (37%), and
5 (5%) were performed for another pathology. While
some cases of PFHT were discovered as early as the 13th
week of gestation [23, 56], three quarters of PFHT cases
were discovered during the third trimester of pregnancy.
The mean time when PFHT was discovered was at 27.3
weeks of gestation (SD = 5.6).
PFHT most often occurred bilaterally (74%); when
unilateral, there did not appear to be a right (11%) or left
(14%) predominance. In 3 cases, the side of a unilateral
effusion was not specified. In 72% cases (101/140), PFHT
was associated with polyhydramnios at the time of discov-
ery. Over half the time, PFHT was associated with
hydrops (99/174). Prevalence did not appear to be signifi-
cantly different between males (57%) and females (43%)
in cases where fetal sex was specified.
Overall, the clinical course of PFHT is unpredictible
(table 1). In 22% of reported cases spontaneous regression
occurred; this occurred even when the initial effusion was
very large [2, 20, 33] and in 2 cases with hydrops [2]. One
can nevertheless try to define the characteristics of PFHT
cases which spontaneously resolve: diagnosis is generally
made early in the second trimester (67%), they are more
often unilateral (65%), they are not associated with poly-
hydramnios (69%), there is no hydrops (90%).
Primary Fetal Hydrothorax: A Review
Table 1. Summary of fetal outcomes re-
ported in the literature
Clinical outcome %
Death after delivery 26
Death in utero 9
Favorable after treatment 43
Favorable without treatment 22
If regression is possible, worsening is, however, more
frequently reported in the literature. This worsening is
characterized by an increase in the volume of the effusion
and by bilateralization. If the PFHT becomes compres-
sive, complications may appear at this point. Esophageal
compression by a large thoracic effusion can impede fetal
swallowing – the principal source of amniotic fluid resorp-
tion beginning in the second trimester [9, 13, 65]. Polyhy-
dramnios most often develops when the PFHT is bilateral
(79%), but may sometimes occur in cases that are unilat-
eral. A very large volume PFHT also can generate signifi-
cant elevation of intrathoracic pressure which produces
diaphragmatic inversion and cardiac plus caval compres-
sion. The cardiac insufficiency which results leads to
hydrops [13, 18, 65]. In 57% of the literature-reported
cases hydrops was involved; in these cases, the PFHT was
always bilateral and most often associated with polyhy-
dramnios (86%). Fetal distress may be engendered by tho-
racic compression, and manifested by an alteration in
fetal cardiac rhythm [31, 48]. Fetal pulmonary hypoplasia
is another complication of PFHT which is correlated with
size, early onset [26] and persistence [66] of the effusion.
Overall fetal mortality for PFHT was 34.8% (69/198) of
which one quarter were deaths in utero and three quarters
occurred postnatally.
Investigation of Prognostic Factors
There were 89 cases where no in utero treatment was
performed; we studied their course to try to define prog-
nostic factors for PFHT. The overall mortality in this
group was 39% (35 cases). Only four criteria were found
to have an adverse prognostic value (table 2): the bilater-
ality of the effusion, the presence of hydrops, the absence
of spontaneous regression, and premature delivery. Fetal
gender and gestational age at the time of diagnosis did not
have a prognostic value. The presence of polyhydramnios
Fetal Diagn Ther 1998;13:325–333 327
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 Table 2. Prognostic factors in
untreated PFHT Perinatal Neonatal p
death survival

Mean gestational age at diagnosis, weeks 28.25 (4.79) 27.02 (7.63) NS

Hydrops, % 76 24
No hydrops, % 25 75 !0.001
Spontaneous resolution, % 0 100
No spontaneous resolution, % 51 49 !0.001
Bilateral, % 47 53
Unilateral, % 23 77 !0.05
Mean gestational age at time of birth, weeks 31.97 (4.04) 34.78 (2.56) !0.001

Figures in parentheses represent SD.

Table 3. Clinical course according to

number of thoracocenteses performed Number of thoracocenteses
1 2 3 4 5 total

Regression of PFHT and favorable outcome 3 2 1 0 1 7

Recurrence of PFHT and favorable outcome 5 4 0 0 0 9
Worsening and death of fetus or infant 7 4 0 1 1 13
Total 15 10 1 1 2 29

did not reach a level of significance as an adverse factor,
perhaps because of the small sample size. In multivariate
analysis by stepwise logistic regression, the sole factor
remaining significant was the presence of hydrops, inde-
pendent of bilaterality and gestational age at the time of
delivery.
Attempted Treatments in utero
Thoracocentesis
Thoracocentesis was proposed for the first time as a
treatment of PFHT by Petres et al. [9] in 1982. This pro-
cedure is performed under echographic control under rig-
orous aseptic conditions. Many authors use maternal gen-
eral sedation and even fetal paralyzation. In our literature
review, we have retrieved 29 cases in 21 publications of
thoracocenteses performed between 17.5 and 37 weeks.
Table 3 shows the clinical course according to the number
of thoracocenteses performed.
It can be concluded that the clinical outcome after tho-
racocentesis is very variable, favorable in 16 out of 29
cases, with repeat thoracocenteses associated with two
positive results and two fetal deaths. Thoracocentesis may
also be performed with pleural fluid for diagnostic pur-
poses with more than 80% lymphocytosis considered
pathognomonic for this condition [7]. It may also be done
in the immediate prepartum period to help improve the
neonatal respiratory status. The principal drawback of
thoracocentesis is the rapid reaccumulation of the effu-
sion. 22 out of 29 fetuses (76%) had a rapid reaccumula-
tion of fluid, and 13 of them had a fatal outcome. In addi-
tion to the typical complications that may occur with
transamniotic puncture during pregnancy, there was a
case reported of fetal death by umbilical cord torsion after
the thoracocentesis [2]. Finally, there has been a warning
that repeated thoracocenteses produce hypoproteinemia
which could favor the development of hydrops [6, 7].

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 Table 4. Clinical course of infants who underwent pleuroamniotic shunting

Action undertaken
shunt only thoracocentesis shunt then repeated total
then shunt thoracocentesis shunting

Regression of PFHT and favorable outcome 37 6a 0 5 48

Recurrence of PFHT and favorable outcome 8 1 1 1 11
Worsening and death of fetus or infant 17 2 1 1 21
Total 62 9 2 7 80

a Of which 1 shunt was pleurocutaneous.

Pleuroamniotic Shunting
The catheters used for pleuroamniotic shunting are the
same type used for draining fetal urinary collections and
hydrocephalus. Use of this technique to drain PFHT was
first proposed by Seeds and Bowes [24] in 1986.
Presently, the most commonly used technique for pleu-
roamniotic shunting is that described by Rodeck et al.
[32]. A metal trocar with cannula is introduced through
the maternal wall through the fetal thorax as close as pos-
sible to the midaxillary line at the level of the base of the
scapula. A double pigtail catheter is introduced through
the trocar lumen. A short introducer rod is used to posi-
tion the distal catheter loop inside the fetal thorax. The
trocar is then gently withdrawn outside of the fetal thorax,
remaining in the amniotic cavity, where a long introducer
rod is then used to position the proximal catheter loop in
the amniotic cavity. Situated in this way, the catheter
creates a permanent communication between the pleural
space and the amniotic cavity.
The results of 80 pleuroamniotic shunts reported in the
literature are summarized in table 4. In the 43 cases where
this information was specified, a pleuroamniotic shunt
was placed on both sides in 24 cases and on one side only
in 19 cases.
In nearly half of cases, the placement of only one pleu-
roamniotic shunt permitted a total regression of the
PFHT and a favorable outcome. The outcome was unfa-
vorable in less than a quarter of the cases. We compared
the clinical outcome of shunts in large case series to those
of individual case reports; there were 20 shunt failures in
68 attempts reported in the large case series compared
with only 1 failure among the 12 single case reports.
Although the difference did not reach the threshold for
significance, it is likely that authors of individual cases
were more willing to report a case with therapeutic success
than one ending in failure.
Catheter migration occurred 10 times (out of a total of
80), but this did not contraindicate the placement of a
new shunt. These catheter migrations have frequently
been reported to be in the direction of the amniotic cavity
[2, 24, 41, 61], but can also occur toward the fetal thorax
[2, 32]. There was even 1 reported case where the catheter
migrated into the maternal peritoneal cavity [41]. Shunt
obstruction is also possible, as reported by Weiner et al.
[67], but in this case, the catheter used was of a finer cali-
ber than that recommended by Rodeck et al. [32]. Shunt
reversal, where amniotic fluid drains into the fetal thorac-
ic cavity, has also been described [32]. Finally, a case of
maternal ascites has been reported after shunt placement
[45].
Pleurocutaneous Drainage
A single case of pleurocutaneous drainage was reported
in the literature by Roberts et al. [23]. When pleural fluid
reaccumulated 4 days after a thoracocentesis was per-
formed at 24 weeks, the authors placed a pleurocutaneous
drain at the 25th week. The drain permitted the reexpan-
sion of the lung to the thoracic wall, it was enveloped after
7 days and the infant was born at 37 weeks without diffi-
culty.
Results of Interventions in Function with
Presence of Fetal Hydrops
Noting that hydrops emerged as the principal prognos-
tic factor, we were interested in analyzing the results of
different treatments in function with the presence or

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 Table 5. Fetal outcome in function
with presence of hydrops and treatment Surviving after Surviving Surviving p
undertaken thoracocentesis after shunt without treatment

Hydrops, % 10 (19) 66.6 (63) 23.5 (17) ! 0.01

No hydrops, % 60 (10) 100 (17) 21.3 (48) ! 0.05

Figures in parentheses represent number.

Table 6. Authors who reported more
than one case of pleuroamniotic shunting
Authors Cases of
pleuroamniotic
shunting
Nicolaides and Azar [41] 35
Mussat et al. [61] 18
Rodeck et al. [32] 7
Ayida et al. [58] 4
Dumez [31] 3 the literature [32, 41, 61]. In taking into account these dif-
Smoleniec and James [62] 2 ferent biases, we nevertheless still consider it possible to
absence of hydrops. Table 5 lists these findings; it under-
scores the poor outcome of thoracocentesis in the case of
hydrops (worse than therapeutic nonintervention) and the
good outcome of shunts with or without hydrops present.
Discussion
All the biases associated with a review of the literature
are present in this study. The outcome of primary hydro-
thorax reported in the literature is likely not an exact
reflection of the overall natural history of PFHT. It is
probable that many cases of PFHT which regressed spon-
taneously were not reported, and thus the 22% sponta-
neous regression rate derived from the literature review
may be an underestimation. It is also probable that some
unfavorable therapeutic results were not published. On
the other hand, it is much more interesting to publish all
the cases where an unusual treatment was proposed, most
notably pleuroamniotic shunting, especially if the out-
come is favorable. It is likely that not every case involving
thoracentesis was published and it is even less likely for
cases with noninterventional care (especially if the out-
come is fetal/neonatal death). One can generally presume
that the published cases reflect the most aggressive thera-
peutic approaches and the cases with good results.
Another bias in this type of review is that the case
authors often work in major tertiary hospitals and thus
have a level of experience far greater than would be possi-
ble for a practitioner working in a smaller community
hospital. Notably, as shown in table 6, three groups ac-
count for 75% of the pleuroamniotic shunts published in
propose a therapeutic approach toward PFHT based on
our review of the literature.
The therapeutic approach adopted first differs depend-
ing on whether there is an emergency situation or not. We
have observed that some cases of PFHT are revealed by
fetal distress, or that the fetal distress is found at the time
PFHT is discovered, manifested by fetal cardiac arrhyth-
mias. It is critical at such a point to decompress the fetal
mediastinum; the most appropriate step, in our opinion,
is thoracocentesis which alone may be sufficient to nor-
malize fetal cardiac rhythm. Thoracocentesis appears to
us to be more appropriate in this setting than the shunt
which is more difficult to place urgently on a fetus in acute
fetal distress. Once this first evacuation is accomplished
and the fetus is stabilized, the necessary full assessment of
PFHT can be completed. This assessment should begin
with confirmation of the presumed primary and isolated
nature of the hydrothorax by ruling out all secondary
hydrothorax etiologies and any associated fetal anomaly.
The maternal assessment serves as a starting point and
should include a review of past obstetrical and medical
history along with a current pregnancy evaluation. Mater-
nal serologies should be verified for all known congenital
infections (toxoplasmosis, rubeola, CMV, parvovirus
B19, adenovirus, syphilis, herpes). The fetal karyotype
should also be verified, keeping in mind that 6% of the
fetuses with hydrothorax are aneuploid [56]. Next, a fetal
ultrasound with particular attention given to the fetal tho-

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rax may permit detection of the numerous pulmonary
causes of secondary fetal hydrothorax (including cystic
malformation of the lung, extralobar fluid collection,
pleural tumor or diaphragmatic hernia). Congenital pul-
monary hypoplasia is very difficult to distinguish from
hypoplasia secondary to PFHT. Fetal cardiac assessment
includes ruling out morphological or rhythmogenic ab-
normalities. The remainder of the echographic exam
includes a systematic search for hydrops or polyhydram-
nios. Meticulous examination of fetal and placental struc-
tures may reveal an associated abnormality. Hence, after
an echographic exam, there are often strong clinical indi-
cations as to whether the hydrothorax is primary or sec-
ondary. In our review, the clinical features which emerged
in support of primary fetal hydrothorax are isolated uni-
lateral or asymmetric hydrothorax; if hydrops is present,
predominance of thoracic effusion over other effusion
sites; edema greater over superior aspect of body; signifi-
cant degree of polyhydramnios; no placental thickening,
and absence of other echographic anomaly.
Given the capricious course of PFHT, marked at times
by spontaneous regression, therapeutic caution is war-
ranted. Thus in the face of a unilateral well-tolerated
PFHT, without associated anasarca, it is acceptable to
proceed conservatively because of possible spontaneous
regression. Hence, a reasonable course of action, in the
absence of any sign of fetal distress, would be to repeat the
ultrasound 15 days after the discovery of the effusion. If
regression of the effusion is seen at the time of the second
examination, the literature suggests an excellent prognosis
(100% survival among the reviewed cases) without any
other therapeutic intervention. If the effusion is stable
and persists on subsequent ultrasounds, the experience of
several authors suggests that it is preferable to perform a
thoracocentesis immediately before delivery in order to
facilitate neonatal resuscitation [63].
If at the time of the follow-up the hydrothorax has
worsened or if it has poor prognostic features (particularly
presence of hydrops), the course to follow will depend
upon the term of the pregnancy. Before 32 weeks, the
results in the reported literature (see table 7) suggest that
the greatest fetal survival can be obtained by placing of a
pleuroamniotic shunt. The improved outcome following
such an intervention did not reach significance, which is
probably due to the very limited number of reported
shunt cases. However, based on the strong trend seen, we
recommend the initial placement of a shunt before 32
weeks if poor prognostic factors are present (in the
absence of fetal distress). In the case of shunt failure, the
data suggest that it is acceptable to try a new placement;
Primary Fetal Hydrothorax: A Review
Table 7. Favorable outcome by treatment method relating to
term of pregnancy
Before 32 weeks After 32 weeks
% %
Shunt 64 75
Thoracocentesis 37 78
No intervention 33 20
p NS NS
however, in the case of repeated shunt failure, the progno-
sis is very poor as the fetal extraction is hampered by high
mortality associated with prematurity and pulmonary hy-
poplasia. In our opinion, this situation is the sole instance
where termination of the pregnancy in the setting of
PFHT is medically indicated.
Between 32 and 37 weeks, we found that the risk of
death increases by a factor of 1.3 (1.1–1.6) for each week
of prematurity. The risk of death is 3.6-fold higher (1.3–
10.3) for an infant born anytime before 35 weeks relative
to an infant born after 35 weeks. Thus, premature fetal
extraction should be avoided (when possible). The results
in the literature (table 7) show that thoracocentesis pro-
vides the best results during this time period. Therapeutic
nonintervention is associated with an 80% fetal mortality.
While this finding did not reach statistical significance,
pending a larger case pool, we feel it is reasonable to con-
sider initial management by thoracocentesis. In case of
rapid effusion reaccumulation, either shunt placement or
iterative thoracocenteses appear to be reasonable strate-
gies. Neonatal respiratory status appears to be significant-
ly improved by maximal pleural space drainage imme-
diately prior to delivery.
Finally, with respect to the single pleurocutaneous
attempt reported [23], in connection with just 1 case, it is
difficult to positively judge this technique which may lead
to serious infection.
To sum up, PFHT is a rare pathology in which antena-
tal therapeutic approaches range from simply watching
and waiting to performing highly invasive in utero inter-
ventions. The more accurate targetting of therapeutic
approaches to particular cases may allow us to significant-
ly ameliorate the somber overall prognosis of this condi-
tion, and make infant survival without sequelae more fea-
sible. This situation is rare in prenatal medicine but it
does nevertheless warrant special attention.
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References
1 Eddleman KA, Levine AB, Chitkara U, Berko-
witz RL: Reliability of pleural fluid lymphocyte
counts in the antenatal diagnosis of congenital
chylothorax. Obstet Gynecol 1991;78:530–
532.
2 Longaker MT, Laberge JM, Dansereau J, Lan-
ger JC, Cromblehome TM: Primary fetal hy-
drothorax: Natural history and management. J
Pediatr Surg 1989;24:573–576.
3 Carroll B: Pulmonary hypoplasia and pleural
effusions associated with fetal death in utero:
Ultrasonic findings. AJR 1977;129:749.
4 Defoort P, Thiey M: Antenatal diagnosis of
congenital chylothorax by gray scale sonogra-
phy. J Clin Ultrasound 1978;6:47.
5 Thomas DB, Anderson JC: Antenatal detection
of fetal pleural effusions and neonatal manage-
ment. Med J Aust 1979;2:435–436.
6 Bovicelli L, Rizzo N, Orsini LF, Calderoni P:
Ultrasonic real time diagnosis of fetal hydro-
thorax and lung hypoplasia. J Clin Ultrasound
1981;9:253–254.
7 Lange IR, Mannig FA: Antenatal diagnosis of
congenital pleural effusions. Am J Obstet Gy-
necol 1981;140:839–840.
8 Porembski M, Laughrin TJ, Brown G, Monthei
F: Ultrasonic antenatal diagnosis of pleural
effusion (chylothorax). Med Ultrasound 1981;
5:51–52.
9 Petres RE, Redwine FO, Crvikshank DP: Con-
genital bilateral chylothorax: Antepartum diag-
nosis and successful intrauterine surgical man-
agement. JAMA 1982;248:1360–1361.
10 Elser H, Borruto F, Schneider A, Schneider K:
Chylothorax in a twin pregnancy of 34 weeks
sonographically diagnosed. Eur J Obstet Gyne-
col Reprod Biol 1983;16:205–211.
11 Jouppila P, Kirkinen P, Herva R, Koivisto M:
Prenatal diagnosis of pleural effusions by ultra-
sound. J Clin Ultrasound 1983;5:73–76.
12 Benacerraf BR, Frigoletto FD: Midtrimester
fetal thoracocentesis. J Clin Ultrasound 1985;
13:302–304.
13 Dubos JP, Bouchez MC, Kacet N, Rouland V,
Morisot C, Bourgeot P: Anasarque non immu-
nologique et chylothorax congénital. Arch Fr
Pédiatr 1985;42:537–538.
14 Jaffa AJ, Barak S, Kaysar N, Peyser R: Antena-
tal diagnosis of bilateral congenital chylothorax
with pericardial effusion. Acta Obstet Gynecol
Scand 1985;64:455–456.
15 Kerr Wilson RHJ, Duncan A, Hume R, Bain
AD: Prenatal pleural effusion associated with
congenital pulmonary lymphangiectasia. Pre-
nat Diagn 1985;5:73–76.
16 Kurjak A, Rajhvajn B, Kogler A: Ultrasound
Diagnosis and Fetal Malformations of Surgical
Interest. Amsterdam, Excerpta Medica, 1985,
pp 243–271.
17 Peleg D, Golichowski AH, Ragan W: Fetal
hydrothorax and bilateral pulmonary hypopla-
sia. Acta Obstet Gynecol Scand 1985;64:451–
453.
332 Fetal Diagn Ther 1998;13:325–333
18 Schmidt W, Harms E, Wolf D: Successful pre-
natal treatment of non-immune hydrops fetalis
due to congenital chylothorax. Case report. Br J
Obstet Gynaecol 1985;92:685–687.
19 Benacerraf BR, Frigoletto FD, Wilson M: Suc-
cessful midtrimester thoracocentesis with anal-
ysis of the lymphocyte population in the pleural
effusion. Am J Obstet Gynecol 1986;155:398–
399.
20 Jaffe R, Di Segni E, Altaras M, Loebel R, Ben
Aderet N: Ultrasonic real time diagnosis of
transitory fetal pleural and pericardial effusion.
Diagn Imaging Clin Med 1986;55:373–375.
21 Lognon P, Bloc D, Saliba E, Gold F: Chylotho-
rax spontané néonatal: diagnostic anténatal.
Arch Fr Pédiatr 1986;43:751–752.
22 Meizner I, Carmi R, Bar-Ziv J: Congenital chy-
lothorax. Prenatal ultrasonic diagnosis and suc-
cessful postpartum management. Prenat Diagn
1986;6:217–221.
23 Roberts AB, Clarkson PM, Pattison NS, Jamie-
son MG, Mok PM: Fetal hydrothorax in the
second trimester of pregnancy: Successful in-
tra-uterine treatment at 24 weeks gestation. Fe-
tal Ther 1986;1:203–209.
24 Seeds JW, Bowes WA: Results of treatment of
severe fetal hydrothorax with bilateral pleu-
roamniotic catheters. Obstet Gynecol 1986;68:
577–580.
25 Booth P, Nicolaides KH, Greenough A, Gamsu
HR: Pleuroamniotic shunting for fetal chylo-
thorax. Early Hum Dev 1987;15:365–367.
26 Castillo RA, Devoe LD, Falls G, Holzman G,
Hadi HA, Fadel HE: Pleural effusions and pul-
monary hypoplasia. Am J Obstet Gynecol
1987;157:1252–1255.
27 Ladreyt JP, Bourlon JP, Maillet-Robert J, Ga-
mot M, Abeille A: Chylothorax néonatal: dia-
gnostic échographique anténatal et évolution
favorable. Pédiatrie 1987;42:535–536.
28 Reece EA, Lockwood CJ, Rizzo N, Pilu G,
Bovicelli L, Hobbins JC: Intrinsic intrathoracic
malformations of the fetus: Sonographic detec-
tion and clinical presentation. Obstet Gynecol
1987;70:627–632.
29 Adams H, Jones A, Hayward C: The sono-
graphic features and implications of fetal pleu-
ral effusions. Clin Radiol 1988;39:398–401.
30 Bruno M, Iskra L, Dolfin G, Farina D: Congen-
ital pleural effusion: Prenatal ultrasonic diag-
nosis and therapeutic management. Prenat
Diagn 1988;8:157–159.
31 Dumez Y: Anasarques et épanchements intra-
thoraciques. Problèmes diagnostiques et théra-
peutiques; in Medical S (ed): Les malforma-
tions congénitales diagnostic anténatal et deve-
nir. Montpellier, Couture, 1988.
32 Rodeck CH, Fisk NM, Fraser DI, Nicolini U:
Long-term in utero drainage of fetal hydrotho-
rax. N Engl J Med 1988;319:1135–1138.
33 Yaghoobian J, Comrie M: Transitory bilateral
isolated fetal pleural effusions. Ultrasound
Med 1988;7:231.
34 Carmant L, Le Guennec JC: Congenital chylo-
thorax and persistent pulmonary hypertension
of the neonate. Acta Pediatr Scand 1989;78:
789–792.
35 Pijpers L, Reuss A, Stewart PA, Wladimiroff
JW: Noninvasive management of isolated fetal
hydrothorax. Am J Obstet Gynecol 1989;161:
330–332.
36 Weaver E, Costello G, Innes M, Dye D,
O’Brien G: Second trimester detection of uni-
lateral hydrothorax: A case report of successful
pregnancy outcome with passive management.
Aust N Z J Obstet Gynaecol 1989;29:257–
258.
37 Zito L: Massive edema and pleural effusions in
a newborn. Ann Allergy 1989;63:277–280.
38 Faure R: Anasarque fœtoplacentaire non im-
munologique. J Gynecol Obstet Biol Reprod
(Paris) 1990;19:1023–1026.
39 Landy HJ, Daly V, Heyl PS, Khoury AN: Fetal
thoracocentesis with unsuccessful outcome. J
Clin Ultrasound 1990;18:50–53.
40 Lien JH, Colmorgen GHC, Gehret JF, Evan-
tash AB: Spontaneous resolution of fetal pleu-
ral effusion diagnosis during the second trimes-
ter. J Clin Ultrasound 1990;18:54–56.
41 Nicolaides KH, Azar GB: Thoracoamniotic
shunting. Fetal Diagn Ther 1990;5:153–164.
42 Philippe HJ, Paupe A, Dompeyre P, et al: Dia-
gnostic anténatal et prise en charge du chylo-
thorax congénital. Arch Fr Pédiatr 1990;47:
737–740.
43 King P, Gosh A, Tang M, Lam S: Recurrent
congenital chylothorax. Prenat Diagn 1991;11:
809–811.
44 Parker M, James D: Spontaneous variation in
fetal pleural effusions. Case report. Br J Obstet
Gynaecol 1991;98:403–405.
45 Ronderos-Dumit D, Nicolini U, Vaugnan J,
Fisk NM, Chamberlain PF, Rodeck CH: Uter-
ine-peritoneal amniotic fluid leakage: An un-
usual complication of intrauterine shunting.
Obstet Gynecol 1991;78:913–915.
46 Estroff J, Parad R, Frigoletto F, Benacerraf B:
The natural history of isolated fetal hydrotho-
rax. Ultrasound Obstet Gynecol 1992;2:162–
165.
47 Jernite M, Donato L, Favre R, Haddad J, Espo-
sito M, Messer J: Traitement médical de
l’épanchement chyleux du nouveau-né. A pro-
pos de trois cas. Arch Fr Pédiatr 1992;49:811–
814.
48 Mandelbrot L, Dommergues M, Aubry MC,
Mussat P, Dumez Y: Reversal of fetal distress
by emergency in uteri decompression of hydro-
thorax. Am J Obstet Gynecol 1992;167:1278–
1283.
49 Sherer DM, Abramowicz JS, Eggers PC,
Woods JR: Transient severe unilateral and sub-
sequent bilateral primary fetal hydrothorax
with spontaneous resolution at 34 weeks gesta-
tion associated with normal neonatal outcome.
Am J Obstet Gynecol 1992;166:169–170.
Aubard/Derouineau/Aubard/Chalifour/
Preux
198.143.32.65 - 8/13/2015 10:35:30 PM
University of Pittsburgh
Downloaded by:
50 Becker R, Arabin B, Nowak A, Entezami M,
Weitzel HK: Successful treatment of primary
fetal hydrothorax by long-time drainage from
week 23. Case report and review of the litera-
ture. Fetal Diagn Ther 1993;8:331–337.
51 Moerman P, Vanderberghe K, Van Hole C,
Fryns JP, Lauweryns J: Congenital pulmonary
lymphangiectasis with chylothorax: A heterog-
enous lymphatic vessel abnormality. Am J Med
Genet 1993;47:54–58.
52 Piney-Laniel F: Hydrothorax fœtaux d’origine
non immunologiques. Lyon, Université de mé-
decine de Lyon, 1993.
53 Geirsson RT, Palsson G: Prenatal thoracocen-
tesis may be life saving in congenital chylotho-
rax. J Perinat Med 1994;22:447–448.
54 Hartmann H, Samuels MP, Noyes JP, Gold-
straw P, Brookfield DS, Southall DP: A case of
congenital chylothorax treated by pleuroperi-
toneal drainage. J Perinatol 1994;14:313–315.
55 Meizner I, Levy A: A survey of noncardiac fetal
intra-thoracic malformations diagnosis by ul-
trasound. Arch Gynecol Obstet 1994;255:31–
36.
Primary Fetal Hydrothorax: A Review
56 Achiron R, Weissman A, Lipitz S, Mashiach S,
Goldman B: Fetal pleural effusion: The risk of
fetal trisomy. Gynecol Obstet Invest 1995;39:
153–156.
57 Aguirre OA, Finley BE, Ridgway LE, Bennett
TL, Cowles TA: Resolution of unilateral fetal
hydrothorax with associated non-immune hy-
drops after intrauterine thoracocentesis. Ultra-
sound Obstet Gynecol 1995;5:346–348.
58 Ayida GA, Soothill PW, Rodeck CH: Survival
in non-immune hydrops fetalis without malfor-
mation or chromosomal abnormalities after in-
vasive treatment. Fetal Diagn Ther 1995;10:
101–105.
59 Horibe S, Imai A, Kawabata I, Tamaya T: Fetal
blood sampling in the assessment of acute non-
immune hydrops fetalis. J Med 1995;26:185–
188.
60 Morrow RJ, Macphail S, Johnson JA, Ryan G,
Farine D, Knox Ritchie JW: Midtrimester tho-
racoamniotic shunting for treatment of fetal
hydrops. Fetal Diagn Ther 1995;10:92–94.
61 Mussat P, Dommergues M, Parat S, et al: Con-
genital chylothorax with hydrops: Postnatal
case and outcome following antenatal diagno-
sis. Acta Paediatr 1995;84:749–755.
Fetal Diagn Ther 1998;13:325–333
62 Smoleniec J, James D: Predictive value of pleu-
ral effusions in fetal hydrops. Fetal Diagn Ther
1995;10:95–100.
63 Cardwell MS: Aspiration of fetal pleural effu-
sions or ascites may improve neonatal resusci-
tation. South Med J 1996;89:177–178.
64 Watson WJ, Munson DP, Christensen MW:
Bilateral fetal chylothorax: Results of unilateral
in utero therapy. Am J Perinatol 1996;13:115–
117.
65 Murayama K, Jimbo T, Matsumoto Y, Mitsui-
shi C, Nishida H: Fetal pulmonary hypoplasia
with hydrothorax. Am J Obstet Gynecol 1987;
157:119–120.
66 Maeda H, Shimokawa H, Yamaguchi Y, Sue-
shi K, Nakano H: The influence of pleural effu-
sion on pulmonary growth in human fetus. J
Perinat Med 1989;17:231–236.
67 Weiner C, Varner M, Pringle K, Hein H, Wil-
liamson R, Smith WL: Antenatal diagnosis and
palliative treatment of non-immune hydrops
fetalis secondary to pulmonary extralobar se-
questration. Obstet Gynecol 1986;68:275–
280.
333
198.143.32.65 - 8/13/2015 10:35:30 PM
University of Pittsburgh
Downloaded by: