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 Table of Contents  
ORIGINAL ARTICLE
Year : 2015  |  Volume : 5  |  Issue : 2  |  Page : 88-95

Nonfluoroscopic well-tempered pressure augmented distal colostogram in high-type anorectal malformation: Our experience


1 Department of Paediatric Surgery, Sawai Man Singh Medical College, Jaipur, India
2 Department of Paediatric Surgery, National Institute of Medical Sciences, Jaipur, India
3 Department of Paediatric Surgery, Adesh Medical College, Bhatinda, Punjab, India

Date of Web Publication16-Jun-2015

Correspondence Address:
Dr. R Gupta
202 A, A3 block, Kamal Apartment - 2, Banipark, Jaipur - 302 004, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2278-9596.158823

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  Abstract 

Background: Accurate imaging of rectourethral fistula is essential for optimal surgical management of infants with high-type anorectal malformation (ARM). The aim of this study was to study the role of nonfluoroscopic well-tempered pressure-augmented distal colostogram in high-type ARM in demonstrating rectourinary fistula, bladder anomalies, and for the presence of vesicoureteral reflux (VUR).
Patients and Methods: We present a prospective study of 10 male infants who underwent a diverting colostomy in the neonatal period from January 2011 to December 2012. Our method of performing a colostogram is similar to conventional colostogram, but involves (a) meticulous bowel preparation preoperatively, ensuring that while doing the last distal colostomy wash before colostogram there is passage of clear saline from the urethral meatus to establish the presence of rectourinary fistula; (b) nonfluoroscopic study (c) performed with gentle and sustained pressure of contrast media, maintained over the period of the investigation; (d) bladder is completely filled with contrast media through rectourinary fistula and it is confirmed clinically; and (e) radiographic films in addition are taken in the micturating phase.
Results: Rectourinary fistula was demonstrated in all (100%) cases. Rectoprostatic urethral fistula was present in three (30%) infants, rectobulbar urethral fistula in six (60%) patients, and one (10%) infant had type 4 congenital pouch colon (CPC) with colovesical fistula. Also three (30%) patients demonstrated unilateral primary vesicoureteral reflux (VUR), which was previously undiagnosed in all of them, as micturating cystourethrogram (MCU) was not attempted.
Conclusions: Our study confirmed the level of anomaly and accurately demonstrated the type of fistula in all patients. It also detected the undiagnosed VUR in three (30%) cases. It obviated the need for a MCU.

Keywords: Pressure-augmented, distal colostogram, nonfluoroscopic, rectourethral fistula, vesicoureteral reflux, well-tempered


How to cite this article:
Gupta R, Sharma S B, Dagla R. Nonfluoroscopic well-tempered pressure augmented distal colostogram in high-type anorectal malformation: Our experience. Arch Int Surg 2015;5:88-95

How to cite this URL:
Gupta R, Sharma S B, Dagla R. Nonfluoroscopic well-tempered pressure augmented distal colostogram in high-type anorectal malformation: Our experience. Arch Int Surg [serial online] 2015 [cited 2019 Sep 23];5:88-95. Available from: http://www.archintsurg.org/text.asp?2015/5/2/88/158823


  Introduction Top


Anorectal malformations (ARM) are a complex group of malformations diagnosed because of the absence of an anus or presence of an ectopic anus at birth. Its incidence is approximately 1:5,000 live births. [1] A high percentage of male neonates with ARM have rectourinary fistula, while ARM without fistula occurs in approximately 5% of cases. [2] A newborn male with ARM, a trace of meconium at the urethral meatus or presence of meconium cells in the urine, strongly suggest the presence of rectourinary fistula, while meconuria is confirmatory. [3] Optimal surgical management of an infant with ARM is directed according to the level of anomaly as well as the presence or absence of a fistula between the rectal pouch and urinary tract. [1],[2],[3],[4],[5] Radiological diagnostic modalities like micturating cystourethrogram (MCU), retrograde urethrography (RGU), and ultrasonography (USG) have been used to identify and delineate the fistula. [3],[6] Distal colostogram is the most valuable investigation as films provide great deal of information regarding the preoperative planning, type of ARM, presence of fistulae between the blind rectum and the bladder, urethra, perineum, and vagina. [1],[2],[3],[4],[5],[6] Accurate imaging of rectourethral fistula can be challenging, especially in centers that do not have access to fluoroscopy.

The technique of nonfluoroscopic well-tempered pressure-augmented distal colostogram in high-type ARM is proposed after imbibing ideas from the work of other authors. [2],[3],[4],[5],[6] It is similar to conventional distal colostogram as described by Cremin et al. [1],[2],[3],[4],[5] The difference being:

  1. Meticulous bowel preparation preoperatively to ensure the distal colon was completely emptied so that any residual fecal material does not block the rectourinary fistula, and while doing the last distal colostomy wash before colostogram there is passage of clear saline from the urethral meatus to establish the presence of fistula;
  2. Nonfluoroscopic study;
  3. Procedure is performed with gentle and sustained pressure of contrast media maintained over the period of investigation, à well-tempered pressure-augmented;
  4. Bladder is completely filled with contrast media through rectourinary fistula and it is confirmed clinically; and
  5. Radiographic films are taken at the onset of dribbling of contrast material out of the urethral meatus, and in the micturating phase. [1],[2],[3],[4],[5]


We herein present our experience with the nonfluoroscopic method of performing distal colostogram in 10 male infants with high-type ARM and study its role in demonstrating type of rectourinary fistula, bladder anomalies, and the presence of VUR.


  Patients and Methods Top


We present a prospective study performed from January 2011 to December 2012. The study included 10 male infants who underwent a diverting colostomy in the neonatal period for high-type ARM. Clearance was obtained from the ethical committee. Informed and written consent was taken from the parents of all the patients prior to the study. All patients had an abdominal USG to detect a renal anomaly. A nonfluoroscopic well-tempered pressure-augmented distal colostogram, using water-soluble contrast material was performed before the definitive procedure. Well-tempered is defined as well prepared distal colon without residual inspissated fecal matter (meticulous bowel preparation) until there is passage of clear saline from urethral meatus. All female cases and males with low ARM were excluded from the study.

Our study was guided by limited resources and involved following steps

  1. Meticulous bowel preparation as mentioned under:
    1. Distal colonic washes were performed with 0.9% normal saline.
    2. Distal colonic washes were started intraoperatively, starting at the time of formation of the colostomy to remove the sticky meconium. The distal stoma was made smaller than the proximal one to prevent its prolapse and also in addition to help the Foley's catheter to get snuggly fit, prevent leakage of the contrast and thereby enabling adequate pressure to be generated during study. It was repeated before discharging the patient from the hospital.
    3. Colostomy washes were performed twice a month in the follow-up visits and before the definitive surgery, ensuring the distal colon was completely emptied and any impacted fecaloma removed.
    4. End-point was clean watery efflux seen from the distal stoma and from the urethral meatus. A flow chart to explain the workup before performing nonfluoroscopic well-tempered pressure-augmented distal colostogram is presented in [Figure 1].
      Figure 1: Flow chart to explain the workup before performing nonfluoroscopic well-tempered pressure-augmented distal colostogram

      Click here to view
  2. Nonfluoroscopic well-tempered pressure-augmented distal colostogram, using diatrizoate meglumine (urograffin 76%) was planned after thorough bowel preparation, before the definitive procedure to evaluate the anoperineal height of the rectal or colonic pouch, demonstrate rectourinary fistula, and bladder anomalies.
  3. During the micturating phase, radiographs were taken in lateral positions for detecting VUR and also assessment of its grade.


Technique of nonfluoroscopic well-tempered pressure-augmented distal colostogram

Patients were hydrated with intravenous fluids before the contrast study. Preprocedure antibiotics were given. Serum electrolytes were measured. A 10 Fr Foley's catheter, was passed through the colostomy into the distal limb for 2 or 3 cm, its balloon inflated with 3-5 ml saline, so as to make it snugly fit and pulled back to act as a plug and avoid any spillage from the colostomy. Gentle and sustained traction was continued throughout the procedure. About 20 ml urograffin, was taken with an equal quantity of saline. It was then injected via the Foley's catheter with gentle and sustained pressure for several minutes, using a 50 ml syringe. Additional volume of contrast was used, when required. Maintaining maximal filling of the rectum for several minutes was done. Continued pressure was applied until there was dribbling of the contrast material out of the urethral meatus or the bladder was full, confirmed clinically or child demonstrated discomfort without dribbling (atresia without fistula/blocked fistula). The first film was taken at this point in anteroposterior position. In cases where the bladder fullness was not clearly appreciated, inspite of dribbling of the contrast material out of the urethral meatus, penis was gently gripped between fingers, and more contrast medium was introduced to gently distend the bladder. Gentle pressure was continued until the bladder fullness was confirmed clinically or child demonstrated uneasiness. Also bladder capacity in each child, as per weight and age was calculated before the procedure to estimate the amount of contrast injection and to prevent bladder perforation. The bladder capacity (volume) in ml = 7 × weight (kg), for age less than 2 years and bladder capacity (ounces) = age (years) + 2. [7],[8],[9] During the micturating phase, radiographs were taken in lateral positions. An extra steep oblique film was also taken in few of the cases.


  Results Top


The results of the study are summarized in [Table 1]. The radiographs of the patients are shown in [Figure 2], [Figure 3], [Figure 4], [Figure 5]. The age of the patients ranged from 5 to 12 months, with a mean of 7.9 months. Seven (70%) infants had sigmoid loop colostomy, while three (30%) had transverse loop Colostomy in the neonatal period.
Figure 2: Radiograph (AP view) in filling phase showing unilateral VUR grade 3 [Figure 2a]; radiograph (lateral view) in micturition phase showing rectoprostatic urethral fistula, complete bladder filling, and unilateral VUR grade 3 [Figure 2b]. R = Rectum, B = Bladder, S = Stoma, U = Urethra, F = Fistula, AP = Anteroposterior

Click here to view
Figure 3: Radiograph (AP view) in filling phase showing CPC type-IV [Figure 3a]; radiograph (lateral view) in micturition phase showing colovesical fistula, bladder filling, and associated VUR grade 1 [Figure 3b]. C = Colon, CPC = Congenital pouch colon, B: Bladder, S: Stoma, U: Urethra]

Click here to view
Figure 4: Radiograph (oblique view) in filling phase showing rectoprostatic urethral fistula, complete bladder filling, and absence of VUR [Figure 4a]; radiograph (lateral view) in micturition phase showing rectoprostatic urethral fistula, no VUR seen [Figure 4b]

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Figure 5: Radiograph (lateral view) in micturition phase showing rectobulbar urethral fistula, complete bladder filling, and unilateral VUR grade 3 [R: Rectum, B: Bladder, S: Stoma, U: Urethra, F: Fistula]

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Table 1: Clinical characteristics of infants


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Radiographs confirmed high-type ARM with rectourinary fistulas in all (100%) patients. Lateral films were diagnostic with delineation of both the level of rectal pouch and the fistula. Rectoprostatic urethral fistula was present in three (30%) infants, rectobulbar urethral fistula in six (60%) patients, and one (10%) infant had type 4 congenital pouch colon (CPC) with colovesical fistula. The diagnosis of CPC was previously unknown in this patient. Bladder anomalies were not detected. Unilateral VUR was demonstrated in three (30%) patients. VUR grade 3 was present on left side in one patient with rectoprostatic urethral fistula, VUR grade 3 on right side in the patient with rectobulbar urethral fistula, and one patient of CPC had grade 1 VUR on left side. VUR was previously undiagnosed in these patients as MCU was not attempted earlier. Procedure was not repeated in any of the patient. There were no procedural complications.

All patients underwent definitive surgery after 3-7 days following distal colostogram. Posterior sagittal anorectoplasty (PSARP) was performed in all patients with rectobulbar urethral fistula and rectoprostatic urethral fistula. Abdominoperineal pull-through was done in case of CPC with colovesical fistula. Fistula could be delineated intraoperatively in all the cases. There was no urological injury in our series.


  Discussion Top


Planning of surgery in patients with high-type ARM is critical to prevent inadvertent injury to urinary tract, pelvic sling, and persistence of fistula. Even though a new trend of primary repair without colostomy in these patients has been reported, [10],[11],[12],[13] most pediatric surgeons prefer a protective colostomy before the definitive surgery. [14] Colostomy itself helps to investigate the malformation, as level of anomaly and rectourinary fistulas can be visualized by contrast enema through distal colostomy stoma. [2],[3],[4],[5],[6],[14] Keiller advised a colostogram by performing washouts of the distal colon and removal of the accumulated meconium before injection of contrast. He injected barium sulfate to visualize the distal blind end. [3] Until 1971, colostogram was performed with simple gastrografin enema. Cremin established the procedure in 1972. He insisted that the injection of contrast should be pressure-augmented. [5] Gross also illustrated the importance of the augmented pressure technique, where continued pressure is to be applied during injection for several minutes to ensure that the fistula is clearly visualized. [2]

We believe, colostogram being the most important investigative tool in ARM surgery, should be performed only after thorough washouts of the distal colon and removal of the accumulated fecalomas, ensuring the distal colon is completely emptied and clear watery efflux is present. The muscles of the pelvic floor are normally contracted, and thus the distal rectum is compressed. With sustained, gentle pressure which is continued for several minutes, the contrast material would fill and distend the rectal pouch, followed by the fistulous tract, urethra, and urinary bladder. Bladder fullness was confirmed clinically in all our cases and then films were taken in the micturating phase for VUR. These films were the most valuable.

Although, fluoroscopy is considered to be ideal for the purpose of colostogram, nonfluoroscopic distal colostogram using water-soluble contrast enema is a cheap and equally informative method in resource-limited setting as an imaging options for the evaluation of ARM. [6] In our set-up, where patient turnover is high, resources are limited, and 24 h radiology facility for fluoroscopy is unavailable, a nonfluoroscopic study is preferred. Increased radiation exposure is expected to be less than with conventional fluoroscopy.

ARM patients were classified radiologically into high and low type using two imaginary lines, the pubococcygeal (PC) line and M line. [6] Rectal pouch terminating above the M line are high-type, while those below it, are low-type anomaly. [6] Female with ARM were not part of our study due to rarity of rectourinary fistula, owing to their short urethra. [4],[6] Males with low anomaly were also not included because of single stage surgical correction in the form of posterior sagittal anoplasty in the newborn period. The preferred method of neonatal colostomy is sigmoid, [4] which is now our institutional protocol. It should be placed at the beginning of the sigmoid colon. Advantages with sigmoid colostomy include: Smaller portion of distal colon is defunctionalized, less chances of colostomy prolapse, low rates of urinary tract infections, and hyperchloremic acidosis, enough length for distal limb to reach the perineum, and easy to carry out colonic washes and colostogram. [4] However, in 30% of our cases, transverse colostomy was performed, owing to the surgeon's preference and because there was type 4 CPC in one, and with suspicion of wide bladder neck fistula (presence of both meconuria and pneumaturia) in other two cases. With sigmoid colostomy in such cases, it is sometimes very difficult to mobilize rectum and gain adequate length during definitive surgery. Also, blood supply of pulled through rectum may be damaged leading to ischemia of rectum.

Although divided and separated proximal colostomy is ideal and also recommended by Pena and other authors to achieve complete defunctioning. [4],[15] It helps in preventing passage of fecal matter between proximal and distal limb which may lead to fecal impaction, megarectum, and fecal contamination of urinary tract (due to rectourinary fistula). But in our institution and other centers, simple loop colostomy with adequate spur works quite well to relieve the obstruction, particularly in emergency. [15],[16]

In our series, distal bowel preparation was performed with similar technique in all the cases. Duration and volume of contrast injected was more in cases with transverse colostomy than those with sigmoid colostomy. Although subjective, extra pressure was not required in patients with transverse colostomy. To remove any bias, injection of contrast was performed each time by same individual. Pressure transducer studies were not carried out in our resource limited set-up and to the best of our knowledge, pressure studies have not been done in past, particularly in India. [1],[2],[3],[4],[5],[6]

The true extent of rectum and the presence of rectourinary fistula were revealed in all our cases. Fistula was also demonstrated with conventional fluoroscopy in other studies. [3],[6] Also our films clearly demonstrate that bladder always gets filled up by the contrast, on exerting gentle and sustained pressure. Even if fistula is long and narrow, contrast will pass through the fistula as meticulous bowel preparation will remove any impediment in the flow of contrast material. [3] Thus nonfluoroscopic study holds good in such cases. While performing the nonfluoroscopic study, any discomfort or uneasiness on part of the infant was carefully observed to prevent rectum or bladder perforation. Also no case of bladder or rectal perforation was observed in another study where penile clamp was used to prevent dribbling of urine and distend the bladder. [6]

The final outcome of the patient of ARM is dependent on the level of anomaly, presence of rectourinary fistula, and the associated anomalies. [4],[17] Higher anomalies are more commonly associated with high-type of fistula than low-type ARM. The probability of urinary incontinence and iatrogenic injury to urogenital tract is more commonly associated with high-type of fistula (colovesical fistula > rectobladder neck fistula > rectoprostatic urethral fistula > rectobulbar urethral fistula). The incidence of fecal incontinence is higher with high-type anomalies than low-type ARM. Also the incidence of associated anomalies, particularly the urogenital anomalies is higher in patients with high-type ARM than in those with low-type ARM. [17] The incidence of urogenital anomalies with high-type ARM is 50-60% and that in low-type ARM is less than 20%. [17] Studies recommended that all children with ARM should undergo an USG of the urinary tract in the neonatal period. [18]

Various studies have shown that the VUR is seen in one-third cases, ranging from 19 to 47% in patients with ARM. VUR is one of the most common genitourinary anomalies in ARM. [6],[19],[20],[21],[22],[23] In our study, there was 30% incidence of VUR in high-type ARM. One study reports, that, the incidence of VUR in ARM might have been higher if MCU had been performed for all patients, including those with normal renal ultrasound (USG) findings in addition to the ones with abnormal findings. [24] The role of MCU is clear when there is upper urinary tract dilatation on USG. [8] ARGUS (AnoRectal, GenitoUrinary, and Sacral anomalies) protocol recommends, that, in low-type ARM cases, MCU should be reserved for patients with dilated upper urinary tracts on USG. [25],[26] Urinary tract infections, lumbosacral-spinal abnormalities, and all patients of high-type ARM should undergo an MCU. [25],[26] Another group suggests that, all patients with imperforate anus, whether high or low, need to be screened for associated urologic anomalies with a MCU. [27]

We recommend and reinforce that, all patients with ARM, particularly high-type, should be investigated for VUR in the neonatal period even if there is no upper tract dilatation on USG, as morbidity associated with missed VUR and reflux nephropathy is significant and can be prevented by chemoprophylaxis.

In addition, for nonfluoroscopic well-tempered pressure-augmented distal colostogram to detect VUR in high ARM, following five criteria must be fulfilled:

  1. Thorough preparation of distal colonic loop.
  2. Injection of water-soluble contrast media with gentle and sustained pressure maintained over the period of investigation.
  3. Presence of recto (colon)-urinary fistula.
  4. Bladder must be full and clinically confirmed.
  5. Radiographs must be taken in micturating phase.


It should ideally be performed 1 month after colostomy, but in Indian patients and in developing nations, compliance is the limiting factor. We stress its importance as it may detect the undiagnosed VUR in high-type ARM, as ARM without fistula occurs only in a small percentage of patients. [2],[28] Also grade of VUR and time for evacuation of the upper tract may be assessed by taking further films. This may obviate the need for a MCU. We also believe that the factual incidence of ARM without fistula is lower than the reported (5% of patients, common in patients with Down's syndrome) due to improperly performed colostograms. [2],[28] Moreover, urological injury did not occur in any of our cases, owing to the preoperative knowledge of fistula. Importance of colostogram in preventing associated injuries has been reported by other authors also. [29]

VUR, particularly its higher grades are best assessed by taking AP films during MCU, but for visualization of bladder neck, posterior urethra, ectopic ureter, and low grades of VUR (particularly grade I); lateral films are useful. [30],[31] Also in our case, lateral films during micturition phase of colostogram were the most informative for the demonstration of level of rectal pouch, type of rectourinary fistula, bladder silhouette, and presence of low grades of VUR. On the other hand, the length of colon available from the mucus fistula down to the fistula and assessment of VUR in the filling phase were obtained by anteroposterior (AP) films. [6],[7]

The calculated radiation dose received by the infant per radiograph was 0.27 mGy/single exposure (DRS, General Electronics) in our study. In all the cases, usually two exposures were given and with a maximum of three exposures. Thus, maximum radiation dose received by the infant during the study was 0.81 mGy, which is extremely low when compared with a conventional fluoroscopic distal colostogram. Although X-ray exposure needed to produce one fluoroscopic image is low as compared to radiography, but during conventional fluoroscopy a continuous X-ray image with multiple exposures and series of images are carried out. Total radiation doses associated with fluoroscopic procedures are reported to be high as compared to radiography. [32],[33],[34] Radiation exposure due to MCU could be avoided in all our cases. Thus, a single study may serve dual purpose vis-a-vis low radiation exposure to the patients and obviating the need for a MCU.

[Figure 6] is proposed for the diagnosis of VUR in high-type ARM. Moreover thorough preoperative washouts and meticulous preparation of the distal colonic limb, done for colostogram in the study lead to clean operative field. This will also decrease the bacterial load in the distal loop which may further prevent morbidity of urinary system (due to the associated rectourinary fistula). No patient in our series suffered electrolyte imbalance with use of Urograffin due to preprocedural correction of hydration and serum electrolytes. This is our initial experience with 10 male ARM patients and to the best of our knowledge; we have not encountered any article about the value of distal colostogram in evaluating VUR. We are yet to formulate an institutional protocol for detecting VUR in ARM patients. A prospective study for comparing MCU with nonfluoroscopic well-tempered augmented-pressure distal colostogram in high-type ARM is being carried out and we are getting encouraging results.
Figure 6: Flow charts for the diagnosis of VUR in high-type anorectal malformation (ARM)

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  Conclusion Top


Nonfluoroscopic well-tempered pressure-augmented distal colostogram confirmed the level of anomaly and accurately demonstrated the type of fistula in all 10 patients in our study. We strongly advocate colostomy washouts before the procedure so that residual fecal material does not block the fistula. The study demonstrated undiagnosed VUR in three (30%) cases, which is one of the most common genitourinary anomalies in ARM. It obviated the need for a MCU, performed for detection of VUR. There was no urological injury in any of our cases, owing to the preoperative knowledge of fistula. In addition, it resulted in reduced radiation exposure to the patients. It is the most important investigative method and it is recommended prior to definitive surgical repair in high-type ARM. In centers with resource limited set-up, it may impact the management and planning of ARM patients.


  Acknowledgements Top


We sincerely acknowledge Dr Ram Babu Goyal, H. O. D. of Paediatric Surgery SMS Medical college, Jaipur for giving permission to carry out the study and Dr. Praveen Mathur, Professor, Paediatric Surgery for his constant motivation.

 
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
 
 
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  In this article
Abstract
Introduction
Patients and Methods
Results
Discussion
Conclusion
Acknowledgements
References
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