Accuracy of pre-operative fistula diagnostics in anorectal malformations

Abstract

Background
Surgical safety during posterior sagittal anorectal plasty (PSARP) for anorectal malformations (ARM) depends on accurate pre-operative fistula localization. This study aimed to evaluate accuracy of pre-operative fistula diagnostics.

Methods
Ethical approval was obtained. Diagnostic accuracy of pre-PSARP symptoms (stool in urine, urine in passive ostomy, urinary tract infection) and examination modalities (voiding cystourethrogram (VCUG), high-pressure colostogram, cystoscopy and ostomy endoscopy) were compared to final intra-operative ARM-type classification in all male neonates born with ARM without a perineal fistula treated at a tertiary pediatric surgery center during 2001–2020.

Results
The 38 included neonates underwent reconstruction surgery through PSARP with diverted ostomy. Thirty-one (82%) had a recto-urinary tract fistula and seven (18%) no fistula. Ostomy endoscopy yielded the highest diagnostic accuracy for fistula presence (22 correctly classified/24 examined cases; 92%), and pre-operative symptoms the lowest (21/38; 55%). For pre-operative fistula level determination, cystoscopy yielded the highest diagnostic accuracy (14/20; 70%), followed by colostogram (23/35; 66%), and VCUG (21/36; 58%). No modality proved to be statistically superior to any other.

Conclusions
Ostomy endoscopy has the highest diagnostic accuracy for fistula presence, and cystoscopy and high-pressure colostogram for fistula level determination. Correct pre-operative ARM-typing reached a maximum of 60–70%.

Full text

R E S E A R C H Open Access
Accuracy of pre-operative fistula
diagnostics in anorectal malformations
Louise Tofft
1,2*
, Martin Salö
1,2
, Einar Arnbjörnsson
1,2
and Pernilla Stenström
1,2
Abstract
Background: Surgical safety during posterior sagittal anorectal plasty (PSARP) for anorectal malformations (ARM)
depends on accurate pre-operative fistula localization. This study aimed to evaluate accuracy of pre-operative fistula
diagnostics.
Methods: Ethical approval was obtained. Diagnostic accuracy of pre-PSARP symptoms (stool in urine, urine in
passive ostomy, urinary tract infection) and examination modalities (voiding cystourethrogram (VCUG), high-
pressure colostogram, cystoscopy and ostomy endoscopy) were compared to final intra-operative ARM-type
classification in all male neonates born with ARM without a perineal fistula treated at a tertiary pediatric surgery
center during 2001–2020.
Results: The 38 included neonates underwent reconstruction surgery through PSARP with diverted ostomy. Thirty-
one (82%) had a recto-urinary tract fistula and seven (18%) no fistula. Ostomy endoscopy yielded the highest
diagnostic accuracy for fistula presence (22 correctly classified/24 examined cases; 92%), and pre-operative
symptoms the lowest (21/38; 55%). For pre-operative fistula level determination, cystoscopy yielded the highest
diagnostic accuracy (14/20; 70%), followed by colostogram (23/35; 66%), and VCUG (21/36; 58%). No modality
proved to be statistically superior to any other.
Conclusions: Ostomy endoscopy has the highest diagnostic accuracy for fistula presence, and cystoscopy and
high-pressure colostogram for fistula level determination. Correct pre-operative ARM-typing reached a maximum of
60–70%.
Keywords: Anorectal malformations, Fistula, Voiding cystourethrogram, Colostogram, Cystoscopy, Endoscopy
Background
Boys born with anorectal malformations (ARM) without
a perineal fistula are suspected to have a recto-urinary
tract fistula until proven otherwise [1]. These patients
are commonly given a neonatal diverted ostomy [2,3].
According to the Krickenbeck classification of ARM,
recto-urinary tract fistulas are sub-divided into recto-
bulbar, recto-prostatic, and recto-bladder neck fistulas
[4]. A minority of patients present with no fistula [1].
The Krickenbeck classification not only predicts long-
term outcome in ARM [5–7], but is also used for plan-
ning reconstructive surgery in detail. Accurate surgical
work up prior to posterior sagittal anorectal plasty
(PSARP) [8] is essential in order to plan surgery cor-
rectly and thereby increase surgical safety, minimize any
risk of unnecessary surgical trauma or injuries to the
urinary and genital tract, avoid the presence of remnants
of fistulas, and to make an accurate decision as to
whether or not to operate laparoscopically [9–12].
A standard method to estimate fistula presence is pre-
operative registration of symptoms: stool-colored urine,
urinary tract infection (UTI), and urine in diverted os-
tomy. Pre-operative radiologic examinations for fistula
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* Correspondence: louise.tofft@med.lu.se
1
Department of Pediatric Surgery, Skåne University Hospital, Lasarettsgatan
48, S-221 85, Lund, Sweden
2
Department of Clinical Sciences, Pediatrics, Lund University, Lasarettsgatan
48, S-221 85, Lund, Sweden
Tofft et al. BMC Pediatrics (2021) 21:283
https://doi.org/10.1186/s12887-021-02761-6
Content courtesy of Springer Nature, terms of use apply. Rights reserved.

level determination traditionally include high-pressure
colostogram [13], possibly combined with voiding
cystourethrogram (VCUG) including additional urinary
tract anomaly diagnostics [14,15]. Other methods to es-
tablish uro-genital and fistula anatomy include peri-
operative cystoscopy and ostomy endoscopy of the
atretic rectum [16].
The diagnostic accuracy of high-pressure colostogram
and VCUG vary from 52 to 100% according to the few
previous published studies with fistula diagnostic accur-
acy data [10,17,18]. A pre-operative fistula diagnostic
accuracy of 100% is unlikely according to our clinical ex-
perience. Establishing pre-operative fistula diagnostic ac-
curacy of conventional modalities compared to definite
ARM-subtyping during PSARP is not only important for
patient surgical safety but is also essential for further de-
velopment and assessment of upcoming modalities, such
as high-frequency ultrasound and high-Tesla magnetic
resonance imaging (MRI) [17–21].
The aim of this study was therefore to evaluate the
diagnostic accuracy of pre-operative clinical symptoms,
VCUG, high-pressure colostogram, and endoscopy of
the urinary tract and diverted ostomy, regarding pres-
ence and location of fistulae compared to peri-operative
findings in male neonates born with ARM.
Methods
Study design
This was a retrospective study of medical records of all
male neonates born with ARM without a perineal fistula,
treated at a tertiary center of pediatric surgery between
January 2001 and October 2020. In 2018 the center was
appointed as one of two national ARM-centers, thereby
evolving from a low- to a high-volume center, now serv-
ing 5 million inhabitants. Patients’medical records were
reviewed regarding pre-operative diagnostic observations
and examinations of fistula presence and location. All
patients underwent surgical reconstruction according to
the original PSARP-method [8] and they had annual
follow-ups according to the local and national ARM-
care programs.
Inclusion and exclusion criteria
All male neonates born with ARM without a perineal
fistula, treated with diverted ostomy and submitted to
surgical work-up including fistula diagnostics prior to
PSARP at the center, were included. Exclusion criteria
were primary PSARP without diverted ostomy and
PSARP performed elsewhere.
Methods
Medical charts were reviewed regarding pre-operative
clinical observations of stool-colored urine, urine in os-
tomy and UTI, X-ray reports of pre-operative VCUGs
and high-pressure colostograms, and peri-operative
examination findings of cystoscopies and endoscopies of
diverted ostomies. Final ARM-type classifications during
PSARP were noted.
Diagnostics
Radiologic- and endoscopic examinations were con-
ducted and the presence and location of a fistula was
noted. VCUGs and high-pressure colostograms were
performed according to standard clinical practice [14,
22] by five pediatric radiology specialists at an accredited
radiology department. Colostograms were performed by
a dynamic X-ray examination with a water-soluble con-
trast injection through a catheter with an inflated cuff
balloon at the orifice of the passive stoma, creating
intra-bowel pressure and a convex appearance of the
atretic rectum, enabling fistula visibility. VCUGs were
performed collecting evidence of vesico-urethral reflux,
and by retracting the catheter slowly in the urethra
under dynamic X-ray examination, enabling fistula visi-
bility. Cystoscopies and endoscopies of diverted ostomies
including fistula catheterization with a guide wire from
the atretic rectum to the urinary tract [16] were per-
formed during PSARP anesthesia by five pediatric sur-
geons or pediatric urologists.
Statistical analysis
Descriptive data analyses and group comparisons were
performed using Excel (Microsoft® Excel for Mac, ver-
sion 16.16.8, 2018) and SPSS® (IBM® SPSS® Statistics, ver-
sion 26, 2019). In group comparisons for dichotomous
data, Fisher’s exact test was used while Mann–Whitney
U-test was used for continuous parameters. Continuous
data were not normally distributed and were therefore
presented as median (min–max), and categoric data as
absolute numbers and percentages, n (%).
Contingency tables of true outcome (final ARM-type
classification during PSARP) and findings of symptoms
and examination modalities were devised. Diagnostic ac-
curacy (%) of each symptom and examination modality
regarding ability of correct differentiation between fistula
presence or absence was calculated by the proportion of
true positive and true negative cases in all evaluated
cases. Diagnostic accuracy (%) of examination modalities
regarding ability of correct fistula level determination
was calculated by the proportion of true positive and
true negative cases in all evaluated cases. To compare
the diagnostic ability regarding fistula presence or ab-
sence among symptoms and examination modalities, a
receiver-operating characteristic (ROC) curve analysis
was also used, with calculation of the area under the
curve (AUC) and its 95% confidence interval (95%CI). A
p-value of < 0.05 was considered significant.
Tofft et al. BMC Pediatrics (2021) 21:283 Page 2 of 6
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Ethics
This study was approved by the Regional Ethics Com-
mittee, Southern Region, Sweden (DNR 2017/191).
Results
Patients
Forty male neonates born with ARM without perineal
fistulas were identified in the hospital records. One was
excluded due to primary PSARP without diverted os-
tomy and one due to PSARP performed elsewhere.
Thirty-eight male neonates were thus included in the
study (Table 1). Median follow-up time post-PSARP was
8.2 (0.3–15.7) years. No remnant of any of the fistulas
was diagnosed during follow-up, while one patient
underwent re-operation for anal stenosis and another
patient for mucosal prolapse.
Diagnostic accuracy of fistula presence
Endoscopy of diverted ostomy had the highest diag-
nostic accuracy of fistula presence with 22 correctly
classified of 24 examined cases (92%). High-pressure
colostogram, cystoscopy and VCUG, showed falling
diagnostic accuracy (71, 70 and 64% respectively).
Symptoms of fistula presence had the lowest diagnos-
tic accuracy; only 21 cases of 38 (55%) were observed
correctly (Table 2). Correspondingly, in AUCs calcu-
lated from a ROC-curve, endoscopy of diverted bowel
showed the highest diagnostic ability of fistula pres-
ence, and VCUG and symptoms the lowest (Fig. 1;
Comparison of diagnostic ability of pre- and peri-
operative examinations of fistula presence in boys
born with anorectal malformations with final classifi-
cation during posterior sagittal anorectal plasty). None
of the diagnostic modalities showed any statistically
significant superiority.
None of the modalities delivered any false-positive
findings. No bowel perforations occurred during high-
pressure colostograms. No complications occurred dur-
ing distal ostomy endoscopy including during fistula
catheterization with a guide wire.
Diagnostic accuracy of fistula level determination
Cystoscopy had the highest diagnostic accuracy of fistula
level determination, correctly classifying 14 of 20
Table 1 Boys born with ARM reconstructed through PSARP and a divided colostomy
Recto-urinary tract fistula
b
n=31
No fistula
n=7
p-value
Prematurity
a
9 (29) 4 (57) 0.20
e
Birth weight (g) 3020 (1700–4280) 3100 (2450–3895) 0.73
f
Small for gestational age 1 (3) 0 1
e
Concomitant malformations
Total (at least one) 27 (87) 4 (57) 0.10
e
Vertebral 19 (61) 1 (14) 0.04
e
Sacral or coccygeal 17 (55) 0 0.01
e
Tethered spinal cord 9 (29) 0 0.16
e
Caudal regression 3 (10) 0 1
e
Urinary tract 11 (35) 0 0.08
e
Genital 5 (16) 0 0.56
e
0.56
e
0.56
e
Gastro-intestinal tract 5 (16) 0
Limb 5 (16) 0
Cardiac 4 (13) 2 (29) 0.30
e
Cranio-facial 2 (6) 1 (14) 0.47
e
VACTERL association 12 (39) 0 0.07
e
Genetic syndromes
Total 3 (10)
c
6 (86)
d
<0.01
e
Trisomy 21 0 5 (71) < 0.01
e
Values presented as the absolute number and percentage of patients, n (%), and as median (min–max)
ARM anorectal malformations, PSARP posterior sagittal anorectal plasty
a
Gestational week < 38 + 0
b
Recto-bulbar fistula n= 8, recto-prostatic fistula n= 17, and recto-bladder neck fistula n=6
c
Di Georges/CATCH 22, OEIS, and suspected syndrome but non-diagnosed
d
Beckwith- Wiedermann, and Trisomy 21
e
Fisher’s Exact test, two tailed
f
Mann–Whitney U-test, two tailed
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examined cases (70%), closely followed by high-pressure
colostogram where 23 of 35 examined cases (66%) were
classified correctly. VCUG had the lowest diagnostic ac-
curacy of fistula level determination; 21 of 36 examined
cases (58%) were classified correctly (Table 3).
Discussion
In this study, ostomy endoscopy and high-pressure
colostogram had the highest diagnostic accuracy for fis-
tula presence in ARM, while cystoscopy and high-
pressure colostogram had the highest accuracy for fistula
level determination. Correct pre-operative ARM-typing
only reached a maximum of 60–70% and no modality
was proven statistically superior to any others.
Even though this study revealed ostomy endoscopy to
be reliable with 92% diagnostic accuracy of fistula pres-
ence, pre-operative information solely regarding fistula
presence is not enough for the responsible pediatric sur-
geon. In planning a safe PSARP, an accurate pre-
operative predictive anatomic model of each malforma-
tion is desirable, preferably visualizing anatomic details
and possible potential obstacles [3,9,20,23].
This study from one ARM-center, with quite low pa-
tient volumes until 2018 when it was appointed as a na-
tional center, revealed fairly poor individual diagnostic
accuracy of all analyzed modalities of fistula level
determination.
According to the literature, high-pressure colosto-
gram should be the gold standard radiologic method
to determine both fistula presence and level in ARM
[13,15]. Our results confirm colostogram to be a ro-
bust modality for determining fistula presence but
weaker than expected for fistula level determination.
To improve pre-operative fistula diagnostics, it is im-
portant to ensure that high-pressure colostogram is
performed according to the literature-described cor-
rect method [14]. High-pressure colostogram has ap-
parent limitations as a diagnostic method due to its
operator-dependent outcome which is compromised
in low-volume centers; in addition it is a source of
radiation and there is the risk of bowel perforation
[24,25].
VCUG has been highlighted as a safe method with
high accuracy for fistula level determination, although it
is also subjected to method limitations including
operator-dependent outcome [14,15,26,27]. VCUG is
easier to perform in younger immobile infants compared
to older children. To optimize fistula visualization, it is
imperative to use VUCG contrast catheters with only
one end-opening and not several side-openings.
Catheterization may be difficult due to urethral ana-
tomic alterations in ARM with recto-urethral fistulas.
According to the clinical experience of pediatric radiolo-
gists in our department, a synergetic effect when per-
forming VCUG and colostogram simultaneously may
improve fistula diagnostic accuracy. Such simultaneous
examinations of colostogram and VCUG were not im-
plemented fully in our department until a couple of
years ago, and corresponding data were therefore not
analyzed in the present study due to there only being a
few cases.
Recent reports of ultrasound- and MRI-examinations
have revealed advantages in pre-operative diagnostics
in ARM by reducing radiation, improving accuracy of
fistula level determination and enabling simultaneous
diagnostics of concomitant malformations of the spine
and sacrum, spinal cord, genitalia and pelvic floor
muscle complex [15,17–21]. MRI method limitations
are need for anesthesia and current limited imaging
resolution in infants, and ultrasound is operator
dependent. To enable accurate visualization and subse-
quent pre-operative anatomic models of fistulas and
pelvic floor anatomy, method development, assessment
and proved safety in children of high-Tesla MRI and
validation of high-frequency 3D/4D ultrasound are
needed. Printed 3D-anatomic models might contribute
to better pre-operative planning and understanding of
the complex malformations.
Strengths of this study include a broad inclusion
population from a national ARM-center with a
standardized program of pre-operative ARM-
Table 2 Diagnostic accuracy of pre- and peri-operative
examinations for fistula presence in boys born with ARM with
final classification during PSARP. n= numbers, (%) = percent
Fistula
a
n=31
No fistula
n=7
Diagnostic accuracy
b
Symptoms 14 (45) 0 21/38 (55)
Stool colored urine 8 (26) -
UTI 5 (16) -
Urine in colostomy 5 (16) -
VCUG 29 7 23/36 (64)
Visible fistula 16 (55) 0
Colostogram 28 7 25/35 (71)
Visible fistula 18 (64) 0
Cystoscopy 19 1 14/20 (70)
Visible fistula 13 (68) 0
Ostomy endoscopy 19 5 22/24 (92)
Visible fistula 17 (89) 0
Guide wire used 11 (58) -
Values presented as the absolute number and percentage of patients, n (%)
ARM anorectal malformations, PSARP posterior sagittal anorectal plasty, UTI
urinary tract infection, VCUG voiding cystourethrogram
a
Recto-bulbar fistula n= 8, recto-prostatic fistula n= 17, and recto-bladder neck
fistula n=6
b
Diagnostic accuracy (%) = (true positive cases + true negative cases) / all
evaluated cases
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diagnostics and long-term follow-up involving only
a handful of radiologists and pediatric surgeons.
Limitations are the retrospective study design with
no secondary review of X-ray reports and only a
few included patients meaning that it was not pos-
sible to show statistically proven differences in
diagnostic ability.
Conclusions
This study reveals that distal ostomy endoscopy has the high-
est diagnostic accuracy for fistula presence and cystoscopy
and high-pressure colostogram has the highest diagnostic ac-
curacy for fistula level determination. Correct pre-operative
ARM-typing only reached a maximum of 60–70% and no
modality was proven statistically superior to any others.
Table 3 Diagnostic accuracy of pre- and peri-operative examinations of fistula level determination in boys born with ARM with final
classification during PSARP. n= numbers, (%) = percent
Fistulae
n=31
No fistula
n=7
Diagnostic accuracy
a
Recto-bulbar
n=8
Recto-prostatic
n=17
Bladder neck
n=6
VCUG 8 15 6 7 21/36 (58)
Correct fistula level determination 5 (63) 8 (53) 1 (17) 7 (100)
Colostogram 8 15 5 7 23/35 (66)
Correct fistula level determination 5 (63) 8 (53) 3 (60) 7 (100)
Cystoscopy 4 12 3 1 14/20 (70)
Correct fistula level determination 2 (50) 9 (75) 2 (67) 1 (100)
Values presented as the absolute number and percentage of patients, n (%)
ARM anorectal malformations, PSARP posterior sagittal anorectal plasty, VCUG voiding cystourethrogram
a
Diagnostic accuracy (%) = (true positive cases + true negative cases) / all evaluated cases
Fig. 1 Comparison of diagnostic ability of pre- and peri-operative examinations of fistula presence in boys born with anorectal malformations
with final classification during posterior sagittal anorectal plasty
Tofft et al. BMC Pediatrics (2021) 21:283 Page 5 of 6
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Abbreviations
ARM: Anorectal malformations; PSARP: Posterior sagittal anorectal plasty;
UTI: Urinary tract infection; VCUG: Voiding cystourethrogram; MRI: Magnetic
resonance imaging
Acknowledgements
None.
Authors’contributions
All authors contributed to the study conception and design. Material
preparation and data collection were performed by L.T. Analysis was
performed by L.T., M.S. and P.S. The first draft of the manuscript was written
by L.T. All authors commented on subsequent versions of the manuscript. All
authors read and approved the final manuscript. All authors have agreed to
be personally accountable for both contribution to the work and accuracy
and integrity of the work.
Authors’information
Not applicable.
Funding
This research did not receive any specific grant from funding agencies in the
public, commercial, or not-for-profit sectors. Open Access funding provided
by Lund University.
Availability of data and materials
The datasets used during the current study are available from the
corresponding author on reasonable request.
Declarations
Ethics approval and consent to participate
The study was approved by the Regional Ethics Committee, Southern Region,
Sweden (DNR 2017/191), with waiver of informed consent. All methods were
carried out in accordance with relevant guidelines and regulations.
Consent for publication
Not applicable.
Competing interests
None.
Received: 18 March 2021 Accepted: 2 June 2021
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