The accumulation of fluid in the pleural space is
a common manifestation of a wide spectrum of diseases in the perinatal
period [1]. The incidence is low, ranging from 2.2 to 5.5 per 1000
births [2]. Most series addressing this issue have only reported
short-term outcome for the neonates born with pleural effusions. The aim
of this paper was to analyze the clinical features and the outcome of
neonatal pleural effusion, including mid-term follow-up, in a tertiary
neonatal care center in Shanghai, China.
Methods
This study was approved by the Ethic Committee of
Children’s Hospital of Fudan University. We retrieved records of
patients from the hospital computer database whose diagnoses were
pleural effusion, chylothorax, hydrothorax, or hydrops fetalis from
January 1, 2007 to December 31, 2011. All neonates admitted to the
neonatal intensive care unit (NICU) with any of the above diagnoses were
followed.
Pleural effusion was diagnosed on the basis of the
combination of clinical manifestations chest X-ray and/or pleural
fluid assessment. The etiology was subse-quently confirmed by a
neonatologist. Categorization of each patient as a congenital or
acquired pleural effusion was confirmed independently by the authors
based on prenatal and neonatal evaluation. The severity of the pleural
effusion was then categorized as per Petersen, et al. [3].
Statistical analysis was performed to investigate the
effect of case characteristics and prenatal intervention on survival
using chi-squared test for categorical variables and t test for
continuous variables. Multivariate logistic regression analysis was
performed to identify independent predictors for survival. A P
value <0.05 was considered statistically significant. Analysis was
performed with SPSS Version 17.0 software package.
Results
In the 5-year period, pleural effusion was diagnosed
in 38 (25 males) out of 15830 newborns admitted to NICU. Four children
died and 34 were discharged from the hospital. The median gestational
age was 34 (range 30-40) weeks. There were 23 full-term babies and 15
premature babies. The birth weight ranged from 1850 g to 4250 g (mean
2526 g). Cesarean section was the mode of delivery in 20 patients. The
pleural effusion was unilateral in 13 patients (6 in the right and 7 in
the left) and bilateral in 25 patients. Prenatal diagnosis was made in 6
patients: 4 had hydrops fetalis and 2 had other causes.
The etiology of neonates with pleural effusion of
each category is listed in Table I. Chylothorax was the
most common etiology in congenital pleural effusion (16) and congenital
heart disease for acquired pleural effusion (22). Four patients with
congenital chylothorax had a resolution of effusion after complete
cessation of enteral feedings and institution of total parental
nutrition (TPN) in combination with medium chain triglyceride- based
formula and/or chest tube placement. Three children with congenital
chylothorax did not respond to complete cessation of enteral feedings
and octreotide therapy.
TABLE I Underlying Causes of Neonatal Pleural Effusion (N=38)
Diagnostic category
|
Number (%) |
Congenital pleural effusion |
16 (42) |
Congenital chylothorax |
7 (18) |
Hydrops fetalis |
4 (9.5) |
Twin-twin transfusion (recipient) |
1 (2.5) |
Cystic hygroma
|
1 (2.5) |
Congenital diaphragmatic hemangioma |
1 (2.5) |
Intrapericardial teratoma |
1 (2.5) |
Trisomy 18 |
1 (2.5) |
Acquired pleural effusion
|
22 (58) |
Congenital heart disease
|
5 (15) |
Complication of thoracotomy |
3 (7.5) |
Congenital diaphragmatic hernia
repair |
2 (5) |
Patent ductus arteriosus ligation
|
2 (5) |
Oesophageal atresia repair |
2 (5) |
Leakage of total parenteral
nutrition |
2 (5) |
Congenital syphilis |
2 (5) |
Capillary leakage syndrome |
2 (5) |
Hypoproteinemia |
1 (2.5) |
Renal failure nephrotic syndrome |
1 (2.5) |
Four (10%) children died in this series. One patient
with hydrops fetalis, congenital anomaly, and congenital heart disease,
who received aggressive resuscitation immediately after delivery, died
12 hours later. Two other patients with hydrops fetalis died during the
course of hospitalization; they had received prenatal cordocentesis and
intra-uterine transfusions. One child who had extravasation of TPN from
the jugular vein catheter died of cardiac tamponade and multi-organ
dysfunction.
Univariate analyses showed that, compared with
neonates who were discharged from the hospital, neonates who died were
smaller, more immature and sicker in the period immediately after
delivery. Significantly poorer outcome was also observed with fetal
hydrops, preterm birth (<34 weeks), and associated defects (Table
II). Upon logistic regression analysis using only these
variables, clinical course remained as a significant independent
predictor of survival.
TABLE II Survival Rate According To Different Prognostic Factors
Factors
|
Survival rate (%) |
P value |
RR (95% CI) |
Location
|
Unilateral
|
13/13 (100) |
0.05 |
1.5 (0.3-11.9) |
Bilateral
|
18/21(84) |
|
|
Hydrops |
Yes
|
1/4 (25) |
0.001 |
13.5 (1.2-123.1) |
No
|
34/34 (100) |
|
|
Associated defects |
Yes
|
2/6 (33) |
0.002 |
9.6 (1.4-67.3) |
No
|
32/32 (100) |
|
|
GA at delivery |
<34 weeks
|
3/5 (60) |
0.003 |
17.5 (1.5-196.3) |
≥34
weeks
|
33/33 (100) |
|
|
Severity of pleural effusion |
Mild
|
11/11 (100) |
0.06 |
8.1 (1.1–-59.2) |
Moderate/severe
|
24/27 (88) |
|
|
In the one year follow-up, 2 of the 34 survivors were
lost to follow-up. We did not observe any recurrence of pleural effusion
in those 32 babies who completed the follow-up. Four children had
recurrent infections of the respiratory tract, and the remaining 28 had
a normal clinical course after one year of follow-up. The growth (weight
and length) and nutritional status assessment did not detect any
malnutrition or growth retardation.
Discussion
In this series of 38 neonates with pleural effusion,
16 patients (42%) had congenital and 22 patients (58%) had acquired
causes. A significantly poorer outcome was observed with fetal hydrops,
preterm birth <34 weeks, and associated defects. We did not observe any
recurrence of pleural effusion over one year follow-up.
The relatively small sample size was a limitation of
the study. Longer period of follow-up might give us more information
regarding these babies with variable underlying diseases.
Clinicians must be aware of the wide range of
disorders causing neonatal pleural effusion, the different types and
their clinical presentations, differential diagnosis, and their specific
treatment [4]. The prognosis ascribed to pleural effusion is dependent
upon patient characteristics, and may be modified by perinatal
intervention. Structured approach and multidisciplinary treatment plays
a key role in improving the prognosis [5].
1. Rocha G. Pleural effusions in the neonate. Curr
Opin Pulm Med. 2007;13:305-11.
2. Rocha G, Fernandes P, Rocha P, Quintas C, Martins
T, Proença E. Pleural effusions in the neonate. Acta Paediatr.
2006;95:791-8.
3. Petersen S, Kaur R, Thomas JT, Cincotta R,
Gardener G. The outcome of isolated primary fetal hydrothorax: a 10-year
review from a tertiary center. Fetal Diagn Ther. 2013; 34:69-76.
4. Caserío S, Gallego C, Martin P, Moral MT, Pallás
CR, Galindo A. Congenital chylothorax: from foetal life to adolescence.
Acta Paediatr. 2010;99:1571-7.
5. Rahman NM, Chapman SJ, Davies RJ. Pleural effusion: a structured
approach to care. Br Med Bull. 2005;72:31-47.