j o u r n a l o f s u r g i c a l r e s e a r c h 1 7 5 ( 2 0 1 2 ) e 4 3 ee 4 6
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Association for Academic Surgery
“Pulling the plug”dManagement of meconium plug
syndrome in neonates
Alex G. Cuenca, MD, Anam S. Ali, BS, David W. Kays, MD, and Saleem Islam, MD, MPH*
Department of Surgery, Division of Pediatric Surgery, University of Florida, Gainesville, Florida
article info abstract
Article history: Background: The significance of meconium plug syndrome (MPS) is unclear but has been asso-
Received 14 June 2011 ciated with Hirschsprung’s disease and magnesium tocolysis. We reviewed our experience to
Received in revised form attempt to identify any potential association with these conditions and to review our outcomes.
17 November 2011 Methods: Using the International Classification of Diseases, Ninth revision, code for meconium
Accepted 18 January 2012 obstruction, patient charts were identified during the 1998e2008 period. A total of 61 cases
Available online 27 March 2012 of MPS were found, after excluding 7 of meconium ileus. Data regarding the hospital course
and outcomes were collected and analyzed.
Keywords: Results: Approximately 30% of patients had spontaneous resolution of the meconium plug
Meconium plug syndrome without any treatment. Of those patients requiring treatment, contrast barium enema was
Meconium ileus used, with 97% success. Only 2 patients required surgical intervention owing to worsening
Left colon syndrome distension and subsequent peritonitis. When we stratified the patients according to
Hirchsprung’s disease gestational age of >36 and <36 wk, contrast barium enemas were performed 2.2 1.8
versus 8.6 7.8 d after birth (P ¼ 0.003), respectively, and the lower gestational age patients
had a longer length of stay. Contrast barium enema was still successful in 94% of patients
with a gestational age of <36 wk. Magnesium tocolysis was noted in 16% of the cases, and
Hirschsprung’s disease was only found in 3.2% of patients.
Conclusions: Patients with MPS have excellent outcomes, independent of gestational age.
Contrast barium enema remains the initial diagnostic and treatment of choice for patients
with MPS. Also, although previous reports have shown a link between magnesium tocol-
ysis and Hirschsprung’s disease with MPS, our experience suggests otherwise.
ª 2012 Elsevier Inc. All rights reserved.
1. Introduction a narrow caliber left colon to a normal or mildly dilated right/
transverse colon [2e4]. Management of this syndrome is
Meconium plug syndrome (MPS) is a relatively benign cause of largely supportive, because it typically improves with the
bowel obstruction in neonates in whom the passage of administration of the barium enema used to diagnose it.
meconium is delayed during the first 24e48 h of life [1]. Within Although no clear pathologic link to more serious forms of
this subset exists another entity known as small left colon bowel obstruction has been established, several reports have
syndrome, which presents in a similar fashion but is noted indicated an association with Hirschsprung’s disease (HD),
radiologically by the presence of a transition zone from citing an incidence as great as 38% [4e6]. MPS has also been
* Corresponding author. Department of Surgery, Division of Pediatric Surgery, University of Florida, 1600 SW Archer Road, PO Box 100119,
Gainesville, FL 32610, United States. Tel.: þ1 352 273 8800; fax: þ1 352 273 8772.
E-mail address: Saleem.islam@surgery.ufl.edu (S. Islam).
0022-4804/$ e see front matter ª 2012 Elsevier Inc. All rights reserved.
doi:10.1016/j.jss.2012.01.029
e44 j o u r n a l o f s u r g i c a l r e s e a r c h 1 7 5 ( 2 0 1 2 ) e 4 3 ee 4 6
associated with maternal hypermagnesemia from attempted
Table 1 e Patients diagnosed with meconium plug
tocolysis in pre-eclamptic women [7]. We believed our expe- syndrome, 1998e2008.
rience was different. Because of these observations, we
Variable GA 36 wk GA <36 wk
decided to perform a retrospective review and compare our
(n ¼ 38) (n ¼ 23)
institutional experience and outcomes with MPS with those
previously reported. Gender (n)
Male 17 7
Female 21 16
GA (wk) 38 1.6 31 3.5
2. Materials and methods Birth weight (g) 3,128 580 1,509 774
Presenting symptoms (%)
2.1. Patient selection Abdominal distension 58 91
Emesis 26 0
After approval from the institutional review board, we per- AD/E 16 4.5
Hematochezia 0 4.5
formed a retrospective review of all patients diagnosed with
Treatment
meconium obstruction (International Classification of Diseases, Resolved spontaneously 29 35
Ninth revision, code 777.1) during the 1998e2008 period. A total of Barium enema 71 65
68 patients were identified. Because the International Classification Successful 96 96
of Diseases, Ninth revision, code for meconium obstruction Complications 0 0
includes patients with MPS and those with meconium ileus, 7 of Age at enema (d) 2.2 1.8 8.6 7.8
Maternal tocolysis 13 22
these patients, noted to have meconium ileus and cystic fibrosis
HD 5.2 0
(CF), were subsequently excluded from the present study.
ICU length of stay (d) 6.3 4.8 50 63
However, their cases have been described in a previous publi-
cation [8]. Furthermore, the diagnosis and classification of GA ¼ gestational age; AD/E ¼ combined abdominal distention and
patients with MPS was based solely on the clinician documen- emesis; HD ¼ Hirschsprung’s disease; ICU ¼ intensive care unit.
tation of “meconium plug syndrome” and failure to pass meco-
nium within 24 h after birth. The patient demographics (gender,
was 2,538 g (Table 1). The most common presenting symptom
gestational age, birth weight, and other demographic data),
was abdominal distension, noted in 70% of cases. Emesis was
radiologic findings, efficacy and use of contrast barium enema,
also noted to be a common presenting feature.
histologic information, delivery records, presence of maternal
Management of the condition was primarily nonoperative.
complications (gestational diabetes), operative findings, and
Approximately 30% of patients had spontaneous resolution of
clinical course were collected for 61 patients. The outcomes were
the meconium plug without any specific treatment. Of those
noted from the follow-up as outpatients. The results of suction
patients requiring treatment, barium enema was used with 97%
rectal biopsies and studies to diagnose CF were also recorded.
success (Table 1), with 98% of patients requiring only one enema
and only 1 patient requiring two. Only 2 patients required
2.2. Patient classification
surgical intervention because of worsening distension and
subsequent peritonitis. We then stratified the cohort into those
A total of 61 patients were identified as having meconium
with a gestational age >36 or <36 wk to determine whether any
obstruction with written documentation in the patients’
differences were present with prematurity. We noted that
electronic or paper medical chart of MPS were categorized as
barium enemas were performed at a later age (2.2 1.8 versus 8.6
having “meconium plug syndrome,” not just a discharge
7.8 d; P ¼ 0.003), and these patients had a longer length of stay
diagnosis of MPS, as described in the previous section. Of
because of prematurity (6.3 4.8 versus 23 44 d). Contrast
these, 7 patients had specific radiologic documentation of
barium enema treatment was successful in 94% of premature
“small left colon syndrome” on enema that was accompanied
neonates (Table 1). The route of delivery was analyzed for all
by the description of a “small” or “significantly decreased left
patients with MPS, and no significant differences between the
colon caliber.” The patients with HD were identified by
groups, with cesarean delivery noted in approximately 50%.
a pathologic diagnosis after a suction rectal biopsy performed
Maternal tocolysis was used in 16% of the cases in our series.
for persisting symptoms.
For the patients with persistent symptoms or recurrent
distension, we considered the coexistence of HD or CF with MPS.
2.3. Statistical analysis
A total of 14 patients (23%) underwent suction rectal biopsy, and
HD was found in only 2 patients (3.2% of all patients). Addi-
Fisher’s exact test was used to determine the differences
tionally, 2 patients underwent sweat chloride testing for CF,
between the groups according to gestational age, with
with negative findings for both. Outpatient follow-up did not
a significant result considered at P < 0.05. Sigmaplot, version
reveal the development of any new cases of HD.
11 (Systat Software, San Jose, CA), was used for all analyses.
3. Results 4. Discussion
Of the 61 patients with MPS identified, 37 were females (Table 1). In the spectrum of neonatal bowel obstruction severity, MPS
The mean gestational age was 35 wk, and the mean birth weight represents a relatively benign entity. Clatworthy et al. [1]
j o u r n a l o f s u r g i c a l r e s e a r c h 1 7 5 ( 2 0 1 2 ) e 4 3 ee 4 6 e45
originally described this finding as colonic obstruction incidence as great as 38% [4]. During follow-up, we did not
because of inspissated meconium. Although the underlying note any cases with persistent issues that would be consistent
pathology of this disorder remains largely unknown, some with HD; with a follow-up period of 6e12 months, we would
reports have indicated that it might be associated with HD have expected to capture such cases.
[4e6], maternal tocolysis [9], and CF [10]. Regardless of these Previous reports have also suggested that magnesium
potential associations, nearly all cases of MPS in our study sulfate administration to pre-eclamptic women might depress
resolved with either rectal stimulation or barium enema, and the function of intestinal smooth muscle and thereby
few patients required surgical intervention. contribute to the finding of MPS in neonates [7,9]. Preclinical
Central to these associations with other disease patholo- studies in rats have contradicted these results and found no
gies is the diagnosis and/or classification of these patients as differences in meconium passage between pups whose
having meconium obstruction, which may be inclusive of mothers were administered magnesium sulfate and untreated
those patients diagnosed with MPS, meconium ileus, and/or rats [7]. We did find that 16% of the patients in our study had
small left colon syndrome. Clearly, meconium ileus is the received maternal tocolysis, although the significance of this
most severe of these clinical entities; however, because our finding is unclear. This result might be explained by the finding
goal was to limit our analyses to those with MPS, we excluded that tocolysis can further complicate the pregnancy and
the patients with meconium ileus. Although CF and MPS have predispose the neonate to septic challenge and/or additional
been found to be associated in some studies, the pathologic drug administration with unintended consequences. Regard-
link between the two entities is unclear and might have less, the reported percentage of neonates exposed to maternal
resulted from misdiagnosed or misclassified meconium ileus. tocolysis has been reported to be 10%e30% and the percentage
Some investigators have linked MPS and CF, reporting an of patients within our study was well within that range before
associated incidence with MPS in 24%e43% of patients [7,11]. and after stratification [12,13].
Other reports have not found any correlation between the two Another entity that is associated with, and thought to be
entities and have cited differences in the clinical definition [5]. a subset of, MPS is neonatal small left colon syndrome. This
Although only two of the patients in our cohort were tested clinical state also presents as meconium obstruction, similar
using a sweat chloride test for CF, neither test was positive. to MPS, and is typically associated with patients born to dia-
Additionally, no additional documentation relating to CF was betic mothers [3,4]. In our series, 7 of the 61 patients ultimately
found in our study. However, we did find several miscoded diagnosed with MPS were noted to have a radiologic diagnosis
reports of MPS that were found to be meconium ileus with CF. of small left colon syndrome on barium enema; however, only
Therefore, in our experience, as previously suggested by 1 mother of the 7 had documented gestational diabetes. These
Keckler et al. [5], these differences in institutional incidence patients were treated in the same manner as the other
could indeed be in part due to differences in definition. patients with MPS and, in our series, had 100% resolution of
In our cohort, 97% of the cases either spontaneously symptoms after the barium enema study. We also noted that
resolved with rectal stimulation or contrast barium enema the gestational age did not play any role in the outcomes, with
treatment. The diagnostic workup was typically initiated in rapid improvement in both patient groups.
our patients when the neonate failed to pass meconium after The present study had several weaknesses. Because the
24 h. The workup involved rectal stimulation, plain radiog- study was performed retrospectively, the ability to conclude
raphy, and, in cases that did not resolve with rectal stimula- a possible or lack of association between MPS and other clinical
tion and in the presence of radiographic evidence of a dilated entities was limited. For example, although the incidence of HD
bowel, contrast barium enema, which, as many have noted, is in our MPS cohort was less than what has been reported in
both diagnostic and, in most cases, therapeutic. Although in other studies, we could not definitively conclude that HD is not
some series, cases in which MPS resolved spontaneously associated with MPS, because the rate of suction rectal biopsy
would not have been included in the analysis, our such was relatively low (23%). In addition, as cited in the “Materials
patients were included to attempt to reflect both the relatively and methods” and “Results” sections, the identification of
benign nature of MPS and to capture all patients who might these patients is dependent on the correct coding at discharge,
have been concomitantly or later diagnosed with other and it is possible that we might have missed some patients.
conditions that have been associated with MPS, such as HD or A larger, multicenter prospective trial would not only
CF. Only 2 patients did not improve with nonoperative treat- address the potential association of MPS with HD, CF, or
ment and required surgical intervention. Both patients maternal tocolysis, but could also address other findings in
required enterotomy with plug evacuation and proceeded to our study such as whether the delay in enema use in the
do well postoperatively. No complications were noted in cohort <36-wk gestational age resulted from a difference in
either of these two cases and neither patient was subse- the timing of the initiation of oral feedings between the
quently diagnosed with HD by biopsy. younger and older groups. Such a prospective trial could also
A recent report by Keckler et al. [5] found, in a study of 77 be designed to further delineate and address the problematic
patients, that the incidence of HD was approximately 13%. and overlapping nomenclature that plagues MPS, such as
This was based on positive pathologic findings identified in 10 those infants who simply have delayed passage of meconium
suction rectal biopsies. However, just as in our cohort, only (patients with spontaneously resolution) versus those with
the few patients with no resolution of bowel function after the actual MPS (patients who require an enema for resolution) or
first or second enema, actually underwent suction rectal whether differences exists between those patients with MPS
biopsy. We found a 3.2% incidence of HD disease in our (clinical diagnosis) versus those with small left colon
patients with MPS, despite previous reports citing an syndrome (radiologic diagnosis).
e46 j o u r n a l o f s u r g i c a l r e s e a r c h 1 7 5 ( 2 0 1 2 ) e 4 3 ee 4 6
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