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 Table of Contents  
ORIGINAL CONTRIBUTION - CLINICS IN NUCLEAR MEDICINE
Year : 2017  |  Volume : 3  |  Issue : 4  |  Page : 188-195

Characterizing renal abnormalities in children with high anorectal malformation with renal scintigraphy: A single-centre experience


1 Department of Nuclear Medicine, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India
2 Department of Paediatric Surgery, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India

Date of Web Publication7-Jul-2017

Correspondence Address:
Ravinder S Sethi
Department of Nuclear Medicine, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi - 110029
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/astrocyte.astrocyte_36_17

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  Abstract 

Objective: The purpose of this study was to characterize the renal abnormalities with renal scintigraphy in patients with high anorectal malformation (HARM). Patients and Methods: We retrospectively reviewed 53 patients who had undergone corrective surgery for HARM from July 2011 to January 2017 and who were referred to this department for Technetium-99m dimercaptosuccinic acid (Tc99m-DMSA) scan for evaluation of renal cortical scarring. Scintigraphic evaluation of all 53 patients included both dynamic renography [Tc-99m diethylene triamine penta acetic acid (DTPA) and Tc-99m-L, L-ethylenedicysteine (L-EC) scan] and cortical scintigraphy. A total of 68 scans were done in these 53 patients in which there were 38 Tc-99m DMSA scans, 15 Tc-99m DTPA and 15 Tc-99m EC scans. Tc-99m DTPA scans were done in 15 patients (26 kidneys) to look for glomerular filtration rate and drainage pattern. Results: Mean age of presentation of these 53 patients (86 kidneys or renal units) with HARM was 28.9 months. Out of 53 patients, male comprised 62.26% of cases (n = 33) and female comprised 37.7% of cases (n = 20). Spectrum of urologic abnormalities noted in our patients with HARM included unilaterally non-visualized kidney in 37.7% cases, i.e., 20/53 patients; hydroureteronephrosis in 28.3% of cases, i.e., 15/53 patients; hydronephrosis in 16.98% cases, i.e., 9/53 patients. Scarring was noted in 28.3% cases, i.e., 15/53 patients. Small kidney was noted in 10 patients out of 53, i.e., 18.8% of cases. Ectopic kidney was seen in 5.6% cases, i.e., 3/53 patients, horseshoe kidney in 3.7% (2/53) and duplex kidney was found in 1 patient, i.e., 1.8% of cases. Conclusion: Children with HARM have a high incidence of renal anomalies and these are the most common cause of morbidity and mortality in these patients. It is important to document these renal anomalies early in life and initiate management as early as possible. Apart from routine investigations, such as ultrasound, renal scintigraphy performed with Tc-99m DTPA, Tc-99m EC and Tc-99m DMSA-III are non-invasive investigations that may provide important help in diagnosis, follow-up and management of renal anomalies in these patients.

Keywords: Cloaca, high anorectal malformation, renal scintigraphy


How to cite this article:
Namgyal PA, Sethi RS, Bagga D, Sehgal AK, Kumar D. Characterizing renal abnormalities in children with high anorectal malformation with renal scintigraphy: A single-centre experience. Astrocyte 2017;3:188-95

How to cite this URL:
Namgyal PA, Sethi RS, Bagga D, Sehgal AK, Kumar D. Characterizing renal abnormalities in children with high anorectal malformation with renal scintigraphy: A single-centre experience. Astrocyte [serial online] 2017 [cited 2017 Sep 20];3:188-95. Available from: http://www.astrocyte.in/text.asp?2017/3/4/188/209928


  Introduction Top


Anorectal malformations (ARMs) are a complex group of congenital anomalies involving the distal anus and rectum, characterized by an absent or ectopic anus. In a significant number of children, urinary and genital tracts are also involved. The prevalence is approximately one per 5000 live births, with a slight male preponderance.[1],[2]

ARMs are associated with other congenital anomalies in up to 70% of cases.[2],[3],[4],[5] Of the various systems involved, genitourinary, cardiovascular, gastrointestinal and vertebral systems are the ones most commonly involved. Long-term prognosis and quality of life of these children depends on the presence and extent of associated anomalies. Urological anomalies are the leading abnormalities associated with ARM with vesicoureteral reflux (VUR) and hydronephrosis (HDN) being the most common findings.[1],[6] Renal anomalies have been observed in 50–60% of patients with high or intermediate forms and 15–20% of low ARM.[7] Unlike genital anomalies, urologic anomalies are difficult to identify on clinical examination and form the primary cause of morbidity and mortality as renal failure is the most important cause of morbidity and mortality in these patients.[8] Rate of death from end-stage renal disease in children with ARM has been reported between 2.5 and 6%.[9]Therefore, the urinary tract anomalies associated with ARMs need to be managed in neonatal period so as to determine the immediate future of child and prevent long-term renal damage.[10],[11],[12],[13]

Antenatal diagnosis of an isolated ARM is rare and in most cases are being diagnosed in the early neonatal period.[14] Early diagnosis of ARMs with associated defects, increased understanding of anatomy and experience in management lead to better outcome. Few studies on this subject are available in India.[15],[16],[17] Imaging studies play a key role in the initial evaluation of ARMs and in taking a decision about colostomy. Most authorities recommend vesicoureterogram, voiding cystourethrogram and ultrasound to study the kidneys and bladder in all patients with ARM.[18],[19],[20],[21],[22],[23] An abdominal ultrasound (USG), generally the first line of investigation, is cheap and widely used to assess the kidneys and other urological anomalies. However, it is relatively non-specific investigation.[24] USG is relatively insensitive to the inflammatory changes of acute pyelonephritis and the residual cortical defects and scars. Cortical scintigraphy with Technetium-99m dimercaptosuccinic acid (Tc-99m DMSA) is significantly more sensitive than sonography and considered the gold standard for cortical scarring,[25] besides being of immense utility in locating and identifying functioning cortex of an ectopic kidney.[24] Renal scintigraphy with Tc-99m diethylene triamine penta acetic acid (DTPA) can be safely used in infants to differentiate between obstructive vs non-obstructive HDN.[24] Tc-99m DTPA can also be used to calculate glomerular filtration rate (GFR). Differential or split function calculation is particularly useful in identifying unilateral lesions, as estimating GFR and serum creatinine may not help in such cases. However, definite role of renal scintigraphy in patients with ARMs is not clearly defined in the literature.[24]


  Patients and Methods Top


In this retrospective study, 53 children [Table 1] were included who had undergone corrective surgery for high ARM (HARM) from July 2011 to January 2017. These children were referred to this department for Tc99m-DMSA scan for evaluation of renal cortical scarring/locating ectopic kidney, Tc99m-DTPA or Tc99m-L, L-ethylene dicysteine (L-EC) scans to look for renal function and drainage pattern.
Table 1: Depicting the age, sex distribution and various investigations in children with high anorectal malformation

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Tc-99m DMSA(III), Tc-99m DTPA and Tc-99m L, L-EC were prepared from a Board of Radiation Isotope Technology (BRIT), Bhabha Atomic Research Centre (BARC), Mumbai kits. All the children were injected with a weight adjusted dose of the radiopharmaceutical based on the maximal dose of 3mCi (111MBq). The minimum administered dose was 1mCi (37Mbq).

DMSA scan was acquired 3 h after Tc-99m DMSA (III) injection, in a supine position using a rotating large field-of-view, dual-head gamma camera (Precedence 16 SPECT-CT, Philips) fitted with low-energy, high-resolution collimator, set at 140 keV with a 20% energy window. Planar images were acquired in four projections, including anterior, posterior, right posterior oblique and left posterior oblique views of kidneys, for 500,000 counts/image in 256 × 256 matrix. Differential function in Tc-99m DMSA scan was acquired using geometric mean of both anterior and posterior images. A break in the cortical outline was labelled as scarring. Unilateral relative function of less than 30% as compared to the other kidney was considered as impaired function.

For Tc-99m DTPA and Tc-99m L, L-EC scans children were adequately hydrated with oral fluids and/or breast feeding as appropriate for their age. Perfusion images of 2 s per frame for 60 s were acquired followed by parenchymal images of 30 s per frame for 25 min. This was followed by a static prevoid image of 3 min. Then, postvoid and delayed images were acquired up to 4 h. Each static image was acquired after keeping the baby in upright position for at least 3 min to assist gravity-dependent drainage. Persistent retention of tracer in the renal pelvis till 4 h was labelled as obstructed urinary system. Processing the study involved, drawing regions of interest around kidneys and background, generating time activity curves for flow and dynamic phases and differential function and GFR by Gate's gamma camera method for individual kidneys were then determined. Diuretic furosemide was administered along with the radiotracer in all patients in a dose of 1 mg/kg body weight.


  Results Top


Age of these 53 children with HARM ranged between 12 days and 12 years with mean age of 28.9 months. Out of 53 children, 33 were male (62.26%) and 20 female (37.7%). Age at presentation in males was 12 days to 9 years 6 months (mean age 18.32 months) and in females 23 days to 12 years (mean age 48.8 months). Out of 53 children, 10 (18.8%) were less than 3 months old and 25 (47%) were less than 1 year old. Kidney function tests (KFT), (blood urea and serum creatinine) performed in these children were within the normal limits in 50 of 53 children. Three children had deranged KFTs. Ten out of 20 female children had common cloaca, age ranging from 1 month to 11 years.

Scintigraphic evaluation of 53 patients included Tc-99m DTPA scans in 15 children (26 kidneys), Tc-99m L, L-EC scans in 15 children (22 kidneys) and Tc-99m DMSA scan in 38 children (61 kidneys). Eight patients underwent both DTPA and DMSA scans. Seven patients underwent both EC and DMSA scans.

The most common urologic abnormality [Table 2] noted in this study was unilaterally non-visualized kidney, i.e., 20 out of 53 patients (37.7% children). Out of these 20 children, 12 were diagnosed on DMSA scan, 5 on DTPA scan and 5 on EC scan. Contralateral kidney showed cortical scarring in 4 out of 20 children on Tc99m DMSA scan. Solitary kidney in these children showed good cortical function in 4 children and impaired function in 1 child on DTPA scan, while 4 children out of 5 showed good cortical function and 1 showed impaired cortical function on EC scan. Ten out of 12 children showed good cortical function on DMSA scan in the solitary kidney.
Table 2: Depicting various renal abnormalities in children with high anorectal malformation

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Hydroureteronephrosis (HDUN), the second most common renal abnormality in this study, was seen in 15 out of 53 children (28.3%) or 19 kidneys. Seven children underwent Tc99m DTPA scan, 8 children underwent Tc99m EC scan, 8 children underwent Tc99m DMSA scan, 4 children underwent both Tc99m DTPA and Tc99m DMSA, and 5 children underwent EC and DMSA. Eleven children showed unilateral HDUN, while 4 showed bilateral HDUN. Three children showed obstructive drainage in both the kidneys, 7 children showed unilateral obstructive drainage and 5 children revealed non-obstructive drainage. Good cortical function was noted in 5/14 kidneys on DTPA scan, 5/16 kidneys on EC scan and 5/16 kidneys on DMSA scan. Impaired function was noted in 3/14 kidneys on DTPA scan, 2/16 kidneys on EC scan and 3/16 kidneys on DMSA scan. Out of 19 HDUN kidneys, 12 (63.15%) showed obstructive drainage pattern. USG done in 14 children with HDUN revealed normal kidneys in 2 children, HDUN in only 5 children and HDN in 7 children. Micturating cysto-urethrogram was done in only 2 of these 15 children with HDUN and it showed VUR on the respective sides. Both children showed renal cortical scarring on the respective sides on DMSA scan. One child showed good cortical function and the other child revealed impaired function on EC scan.

Cortical scarring was the third common abnormality encountered in this study. It was seen in 15 out of 53 children (28.3%) or 19 kidneys. Out of these 15 children, 11 showed unilateral renal scarring and 4 showed bilateral scarring on DMSA scan. These scarred kidneys showed adequate cortical function in 9/19 kidneys and impaired cortical function in 10/19 kidneys on DMSA scan.

HDN was noted in 9 (16.98%) out of 53 children or 13 kidneys.

One child underwent DTPA scan, 4 children underwent EC scan, 4 children underwent DMSA and 1 child underwent both EC and DMSA. Five children showed unilateral and 4 showed bilateral HDN. Out of 5 children who underwent DTPA and EC scan, unilateral obstructive HDN was noted in 1 child, bilateral obstructive HDN in the 1 child and non-obstructive HDN was seen in 3 children. 3/13 HDN kidneys (23.07%) showed obstructive drainage pattern. Good cortical function was noted in 4 kidneys on EC scan and 2 kidneys on DMSA scan. Impaired cortical function was seen in 3 out of 8 kidneys on EC scan, 1 out of 2 kidneys on DTPA scan and 5 out of 10 kidneys on DMSA scan. USG done in these 9 children revealed bilateral HDN in 5 children, unilateral HDN in 1 child and bilateral HDUN in 3 children.

Small kidneys were seen in 10 out of 53 children (18.8%) or 10 kidneys. Small kidneys were seen in 4 children on DTPA scan, 4 on EC scan and 7 DMSA scan.

Out of 10 small kidneys, good cortical function was noted in 1 kidney on DTPA scan and 1 kidney on DMSA scan. Impaired cortical function was seen in 4 kidneys on EC scan, 3 kidneys on DTPA and 5 kidneys on DMSA scan.

Ectopic kidneys were seen in 3 out of 53 children (5.6%) or 4 kidneys. Out of 3 children, 2 showed crossed fused ectopic kidneys. One ectopic kidney was seen on DTPA scan and 4 ectopic kidneys on DMSA scan. Good cortical function was noted in 1 kidney on DTPA scan and 2 kidneys on DMSA scan. Impaired cortical function was noted in 2 kidneys on DMSA scan.

Horseshoe kidneys were noted in 2 out of 53 (3.7%) children. One horseshoe kidney was seen on EC scan and 1 on DMSA scan. One child with horseshoe kidney showed good cortical function with non-obstructive drainage in both renal moieties, while the other child revealed impaired function with obstructive drainage in one moiety and good function and non-obstructive drainage in other moiety.

Duplex kidney was noted in 1 child (1.9%) or 1 kidney. It was diagnosed on both EC and DMSA scans. Cortical function in this duplex kidney was impaired with obstructive drainage. Out of the 53 children, 10 female children had common cloaca [Table 3]. These children age ranged from 1 month to 11 years, with an average age of 3.7 years. Four out of these 10 children or 8/20 kidneys (40%) showed bilaterally normal functioning kidneys. Good cortical function was seen in 1 kidney on EC scan, 5 kidneys on DTPA scan and 10 kidneys on DMSA scan. Impaired function was seen in 3 kidneys on DTPA and in 1 kidney on DMSA scan. The most common renal abnormality observed in them was HDUN. HDUN was seen in 4/10 patients or 6/20 kidneys (40%). Urinary obstruction was noted in 5/6 of these HDUN kidneys. Next common abnormalities seen were single kidneys and small kidneys, which were noted in 2/10 patients or 2/20 kidneys (20%) each. GFR was measured in 4 children, ranging from 65.3 to 101.4 ml/min. Average GFR in these female children with common cloaca was 84.0 ml/min.
Table 3: Depicting the age, sex distribution and various investigations in children with common cloaca

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Three out of 53 (5.6%) patients had deranged KFT levels. In 3 patients with deranged KFT levels, 2 children had HDN. One child showed duplex kidney and scarring on one side with impaired cortical function and obstructive HDN in both the kidneys. Second child revealed impaired cortical function and scarring in both the kidneys with HDN on one side and HDUN in the other kidney. Third patient showed small sized kidney on one side with impaired cortical function and scarring in both the kidneys. GFR of this child was 33.6 ml/min.

DTPA scan done in 15 patients (26 kidneys) was to look for GFR and drainage pattern. GFR calculated by standardized Gates gamma camera method ranged from 33.6 ml/min to 117.6 ml/min in these 15 patients and mean GFR was 74.95 ml/min. Minimum GFR noted in our study was 33.6 ml/min, which was seen in a 9.5-year-old child with small and impaired functioning right kidney and scarring in both the kidneys. KFT of this child was deranged.


  Discussion Top


Of various anomalies associated with HARM patients, renal anomalies are the most common and may be present in up to 60% of cases (7). Renal failure is the most common cause of morbidity and mortality in these patients (8).

The most common urologic abnormality noted in this study was unilaterally non-visualized kidney seen in 37.7% children. Non-visualized kidney may be due to renal agenesis or dysplastic kidney, and it is reported up to 20% of patients with high anomalies.[26] Harisankar et al.[24]reported 37.5% cases of unilateral non-visualized kidney. Patients with a solitary functioning kidney are also at increased risk of developing future chronic renal disease. These patients, therefore, need regular follow-up as they are at risk of developing renal impairment due to glomerular hyperfiltration, thus gradually progressing to renal dysfunction.[27],[28]

HDUN was the second most common renal abnormality in this study seen in 28.3% cases (12 kidneys). Obstructive drainage pattern was noted in 63.15% of these renal units. Impaired function was noted in 3 out of 14 kidneys on DTPA scan and 2 out of 16 kidneys on EC scan.

In this study HDN was noted in 15.09% of cases (8/53 patients or 13 renal units). Of these 13 renal units, 23.07% showed obstructive subrenal drainage pattern. Impaired function was noted in the 3 out of 8 kidneys on EC scan and 1 out of 2 kidneys on Tc99m DTPA scan. Cortical scarring was noted in 5 out of 10 kidneys on Tc99m DMSA scan. The incidence of HDN varies greatly among the series reported in literature. HDN was the most common problem identified in the kidneys by Ratan et al.,[4],[29] Out of which 25% showed requirement of surgical reconstruction.[29] Renal scintigraphy with Tc-99m DTPA and TC-99m EC scan can play a vital role in evaluation of drainage pattern and deciding about surgical management to avoid potential impairment in renal function.[25]

Ectopic kidney and crossed fused renal ectopia was reported in 5.6% of patients of HARM in this study, which is comparable to McLorie et al.[30] and Metts et al.[31] who reported approximately 5% ectopic kidneys and 5% renal fusion. These patients may not need specific surgical management of renal anomalies; however, these do predispose to urinary tract infections and VUR, which needs to be considered. In this study no cortical scarring was noted in these kidneys.

Tc99m DMSA scan showed renal cortical scarring in 15/53 patients (28.3%) in this study. Cortical scarring was seen in 7/40 patients (17.5%) in a study by Harisankar et al.[24] Cortical scarring may develop due to VUR, however, exact incidence of VUR could be assessed in these children.

Duplex kidney was reported in 1.8% of patients of ARM in this study. Renal duplication, either partial or complete, was reported to be 2% Metts et al.[31] and 5% by Mollitt et al.[32] The management of these patients is the same as in those without HARM. An ectopic ureter associated with an upper pole moiety must be considered as a cause of incontinence in these children.[33]

Two out of 53 children showed horseshoe kidney (3.7%) in this study. One patient showed single moiety obstructive drainage. Warne et al.[34] reported one case of horseshoe kidney in their study. Since there is a high incidence of HDN in horseshoe kidneys as compared to normal kidneys (65 and 7%, respectively),[35] it is essential for precise diagnosis and effective workup.

Most high anomalies in girls are cloacae; a high anomaly with a rectovaginal fistula is exceedingly rare [16] and in the literature many such patients are misclassified. Cloacal malformations are more common than previously thought, most likely because patients were previously misdiagnosed as having a rectovaginal fistula.[36] In this study, 10 female children presented with cloacal malformation. Four out of these 10 patients showed bilaterally normal functioning kidneys. Most common renal abnormality observed in these children was HDUN, followed by small kidney and unilaterally non-visualized kidney. Average GFR in these female children with common cloaca was 84.0 ml/min. Braga performed renal USG, voiding cystouretrogram, renal scan and sacral radiograph in children with cloacal malformation to look for long-term renal function and continence status. Renal outcome was evaluated based on split renal function, GFR and age-adjusted serum creatinine values (μmol/L).[36] Evaluation of renal function showed that 75% children had an abnormal GFR (<80 ml/min) and had some degree of renal function impairment. As significant number of children with cloaca develop renal impairment, it has been recommend that all new cloaca patients have a baseline GFR measurement done at around 1 year of age.[37]

Tc-99m DTPA scan was done in 13 patients (26 kidneys) to look for GFR and drainage pattern. With mean age of 30.30 months, average GFR was 71.66 ml/min ranging from 33.6 ml/min to 101.4 ml/min. Minimum GFR noted in our study was 33.6 ml/min, which was seen in a 117-month-old child with HARM with small and impaired functioning right kidney and scarring in both the kidneys.

USG is an important non-invasive method for evaluation of urologic system; however, it is mainly a morphologic imaging modality providing little information about the functional status of the kidney. Its subjective nature may sometimes be cumbersome in follow-up of patients. In this study, USG of kidneys was done in all children.[24] Out of 53 children, 84.89% had some renal abnormality on USG, which may be due to referral bias as only symptomatic patients might have been referred. Intravenous urography (IVU) have been used to evaluate renal function; however, it is a difficult procedure in small children. It delivers a high radiation dose and has the risk of hypersensitivity besides iodinated contrast agents being contraindicated in patient with elevated serum creatinine. Renal diuretic scintigraphy with Tc99m DTPA and Tc99m EC offers advantage in comparison to IVU. Apart from being sensitive, it is safe to perform in infants. Cortical scintigraphy with Tc99m DMSA has the utility in locating ectopic kidney and may be considered to diagnose cortical scarring in children with urinary tract infection and suspected VUR.[24]

Warne et al.[33] reported that 25% of patients with a solitary kidney, 44% with renal dysplasia, 72% with VUR and 47% with renal scarring had some degree of renal function impairment. Therefore, it is important to document the renal anomalies in children with HARM early in life and manage these appropriately as per the need. Ultrasound scan cystogram and renal scintigraphy should, therefore, be performed in all HARM children at presentation to monitor their renal status.[38]


  Conclusion Top


Children with HARM have a high incidence of renal anomalies and these are the most common cause of morbidity and mortality in these patients.[24] It is important to document these renal anomalies early in life and initiate management as early as possible. Apart from routine investigations, such as ultrasound, renal scintigraphy performed with Tc-99m DTPA, Tc-99m EC and Tc-99m DMSA-III are non-invasive investigations that may provide important help in diagnosis, follow-up and management of renal anomalies in these patients.[38]

Financial support and sponsorship

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Conflicts of interest

There are no conflicts of interest.

 
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