5/30/2018
SOUTH AFRICAN JOURNAL
OF RADIOLOGY, VOL 21, NO
1 (2017)
Abstract (#2)
Introduction (#3)
Methods (#4)
Results (#9)
Discussion (#10)
Conclusion (#12)
Acknowledgements (#13)
References (#16)
About the Author(s)
Monica S. Msomi
(mailto:monicasheilamsomi@gmail.com)
Department of Radiology, College of
Health Sciences, Nelson R. Mandela
School of Medicine, University of
KwaZulu-Natal, South Africa
Hansraj Mangray
Department of Paediatric Surgery,
College of Health Sciences, Nelson R.
Mandela School of Medicine, University
of KwaZulu-Natal, South Africa
Vicci du Plessis
SKG Radiology, Mandurah, Australia
Citation
Msomi MS. Mangray H. Du Plessis V. An
assessment of the accuracy of contrast
enema for the diagnosis of Hirschsprung
disease at a South African tertiary hospital.
S Afr J Rad. 2017;21(1), a1093.
https://doi.org/10.4102/sajr.v21i1.10
93
(https://doi.org/10.4102/sajr.v21i1.1093)
https://sajr.org.za/index.php/sajr/rt/printerFriendly/1093/1395
Msomi
Original Research
An assessment of the accuracy of contrast ene
tertiary hospital
Monica S. Msomi, Hansraj Mangray, Vicci du Plessis
Received: 16 Sept. 2016; Accepted: 17 Jan. 2017; Publi
Copyright: © 2017. The Author(s). Licensee: AOSIS.
This is an Open Access article distributed under the terms o
(http://creativecommons.org/licenses/by/2.0) , which per
original work is properly cited.
Abstract
Objectives: To compare radiological findings with the histo
enema as an initial screening and diagnostic tool. To correla
Materials and methods: Systematic searches were condu
Laboratory Service records for patients aged 0–12 years, w
performed between 01 January 2011 and 31 August 2015 in
accuracy levels were calculated by comparing radiological re
Results: Diagnostic accuracy of contrast enema was 78%,
(68.8%) and positive predictive values (63%) were conside
compared with the international reports of up to 30%.
Conclusion: Contrast enema remains useful as an initial sc
hospital were consistent with the best international results f
disease).
Introduction
Hirschsprung disease (HD), also known as colonic aganglion
caused by the lack of distal enteric ganglion cells with a rep
segments of bowel may be involved. It is associated with si
cautious to promptly identify or exclude this disease. There
because all the diagnostic modalities [contrast enema, anor
false-positive (FP) results.2 (#CIT0002_1093) Following critica
the 1980s, it was shown that radiologic studies alone are no
are also required for an accurate diagnosis.3 (#CIT0003_1093
standard for diagnosis.4 (#CIT0004_1093) However, the risks
related risks,5 (#CIT0005_1093) led to the development of the
previously, it can produce FP and FN results. Contrast enem
opinion forms part of the data used by clinicians to decide w
obstruction or abnormal bowel habits.3 (#CIT0003_1093) The
in many centres, including Grey’s Hospital, a tertiary referra
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literature regarding its usefulness in the work up of patients

enema in excluding HD is approximately 80% – 88%6 (#CIT
diagnostic accuracy of the contrast enema is, therefore, of c
assess how accurately radiologists are able to make the dia
identifying possible factors that may contribute to reduced d

Methods

Study sample and design

A retrospective audit was undertaken of all contrast enemas
and 31 August 2015 inclusive. Only children investigated wi
were included in the study.

Study description
A systematic search was conducted through the Picture Arc
referred for contrast enemas between 01 January 2011 and
for these patients were reviewed to identify the patients wh
or changed bowel habits). The total number of patients iden
National Health Laboratory Service (NHLS) website, and ult
and biopsy. The remaining 43 patients were excluded from
further for the purposes of our study.

Enemas were performed at our tertiary institution mostly by

predominantly examined by the performing trainee radiolog
examined by a radiology registrar alone. Only water-soluble
omnipaque or ultravist as contrast agents. For the procedur
inflated. Contrast is passed as far as possible, in most cases
mainly on the presence of a transition zone (TZ) or calibre c
delayed post-contrast evacuation of bowel, saw-tooth muco

Full-thickness rectal biopsies were performed under genera

posteriorly, and sent for haematoxylin and eosin (H&E) stai
available during the study period but has since been acquire
presence or absence of ganglion cells in Meissner’s submuco
neuronal hyperplasia.

Related symptoms such as constipation, abdominal distensi

each patient to ascertain any statistical significance.

A data sheet was used to collect the relevant data. The stud
Provincial Department of Health and the management of Gr

Ethical consideration
This is a blind, retrospective, descriptive study, focusing on
protected and no consent was necessary.

Statistical analysis
Data were collected and entered into a computerised datab
and formats was developed. Sensitivity and specificity rates
method of Wilson. The single inconclusive enema result was
were identified using Fisher’s exact test. Factors associated
model. Because of the relatively small number of positive ca
sided p-value of < 0.05 was considered statistically significa

Results
Of the 54 patients included in the study, 28 had positive con
1 (#T0001_1093) ). Seventeen patients who had positive e
patients with positive enemas, 10 patients turned out to ha
negative enemas, most (22) had concurrent negative histol
histology results. The single inconclusive enema result had
inadequate or non-representative samples. The inconclusive
other supporting features of HD.
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TABLE 1: (https://sajr.org.za/index.php/sajr/article/view

Table 2 (#T0002_1093) denotes the diagnostic evaluation

TABLE 2: (https://sajr.org.za/index.php/sajr/article/view
rectal biopsy as the gold standard.

In terms of risk factors for the disease, with adjustments fo

positive on histology for the disease than children over 1 m
testing positive than females. In terms of radiological featu
testing positive for the disease (OR 3.4, 95% CI 0.6–18.4, p
3 (#T0003_1093) ). Of the related symptoms, only vomitin
however, this was not statistically significant when adjusted

TABLE 3: (https://sajr.org.za/index.php/sajr/article/view

Discussion
Results of the Grey’s Hospital were comparable with the bes
88%) for excluding HD.6 (#CIT0006_1093) There is, however,
Lorijn et al. concluded that such a discrepancy may be due
the low number of included patients.2 (#CIT0002_1093) Neve
referring patients for biopsies, early diagnosis and managem

The high negative predictive value (NPV) of 95.7% was con

missed, which supports the usefulness of the contrast enem
international reports of up to 30%. This may be because th
other institutions. Seventeen out of the 54 studied patients
prevalence of the disease and may be related to the fact th
with cerebral palsy and may be related to regular rectal was
patients and have been shown to lead to FN results.2 (#CIT0
concerning for radiological overdiagnosis, which may lead to
aforementioned reasons.

A study by O’Donovan et al. assessed the validity of using lo

investigation of HD.3 (#CIT0003_1093) It found good agreeme
in neonates and infants. The RRSR and the TZ were the onl
was even more significantly associated with the disease tha
27.3% of children without the disease. The TZ was present
disease. This is consistent with most literature reports whic
accurate diagnostic sign for HD,2 (#CIT0002_1093) ,7 (#CIT000
(#CIT0003_1093)
which reported the RRSR as the most sensit
radiographic TZ is also known to be the most reliable radiol
surgical management.9 (#CIT0009_1093) Notwithstanding the
more difficult to demonstrate.4 (#CIT0004_1093) Furthermore
aganglionosis,2 (#CIT0002_1093) which means that the absen

(https://sajr.org.za/index.php/sajr/article/viewFile/1093

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(https://sajr.org.za/index.php/sajr/article/viewFile/1093

(https://sajr.org.za/index.php/sajr/article/viewFile/1093

In this study, we did not assess for any correlation between

The most common presenting symptoms for HD are delayed

vomiting (19% – 37%).1 (#CIT0001_1093) In older children, t
however showed that for all age groups, vomiting was the o
statistically significant when adjusted for age and sex, as af
one child, and while constipation and abdominal distension
established with these symptoms. Lewis et al. demonstrate
of life and have delayed passage of meconium, abdominal d
least three of these findings. Most of our patients with HD w
contrast enema. Our findings of neonatal age and male sex
cent of patients are diagnosed when they are newborns, an
(#CIT0001_1093)

Diamond et al. reported that on a univariate analysis, age b

statistically, at the 0.05 level, associated with an increased
meconium beyond the first day of life was associated with a
multivariate analyses, the absence of a TZ, female sex and
history of bilious emesis decreased the risk.

Our study showed that females are more than three times a
(OR 3.4, 95% CI 0.5–25, p = 0.2). This is consistent with t
significant. Absence of a TZ was found more often in FP (40
Vomiting was found more often in TP rather than in FP patie
were FP versus 25% of children with vomiting (OR 0.4, 95%
provides some support to the findings of the report. Our stu
enema result.

According to our study, age above 30 days is an indication o

There was only one reported case of delayed passage of me

Factors associated with a TP result in our study include age

(#T0004_1093) ). No statistical significance was demonstra
0.1; OR 3.4, 95% CI 0.5–25.3, p = 0.3 and OR 2.3, 95% C

TABLE 4: (https://sajr.org.za/index.php/sajr/article/view

Lastly, a note about ARM. This is a non-invasive diagnostic t

ideal screening tool.2 (#CIT0002_1093) A study by de Lorijn e
(93% vs. 97%) than contrast enemas.4 (#CIT0004_1093) Unf

Limitations
The study is limited to a certain age group. The correct hist
interpretation is dependent on the degree of experience of t
are in turn influenced by the degree of experience of the pe
as lack of co-operation as can be seen with younger patient

Conclusion
The contrast enema remains useful as a screening test for H
international results for sensitivity of the contrast enema (a

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biopsy remains the gold standard for diagnosis of HD.

Acknowledgements
The authors would like to thank Catherine Connolly for her
analysis and suggested revisions for this article.

Competing interests
The authors declare that they have no financial or personal

Authors’ contributions
M.S.M. performed literature review, prepared research prop
primary write-up of the research manuscript. H.M. was the
helped with manuscript editing. V.d.P. supervised the resear

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