Surgical management of renal tract problems

SYMPOSIUM: SURGERY AND ORTHOPAEDICS

Surgical management of renal tract problems Lisa C Steven Imran Mushtaq

Abstract The majority of renal tract problems in childhood are congenital in origin with an overall incidence of 2e4 per 1000 live births. Some anomalies may occur in isolation e.g. posterior urethral valves; whilst others may be part of a more complex spectrum of anomalies e.g. duplex or horseshoe kidney in VACTERL or VATER association. Prenatal screening has highlighted an increasing number of children with renal tract anomalies which may require paediatric specialist involvement in the immediate postnatal period. Most congenital renal tract problems are managed by paediatric urologists and paediatric surgeons with a special interest in urology. The aim of this review is to highlight the most common renal tract disorders of childhood and to discuss their aetiology, clinical presentation, investigation and surgical management.

Keywords duplex kidney; horseshoe kidney; multi-cystic dysplastic kidney; pelvi-ureteric junction obstruction; posterior urethral valves; vesico-ureteric obstruction; vesico-ureteric reflux

Figure 1 The mesonephric duct lies laterally and connects to the primitive cloaca. The ureteric bud develops from distal mesonephric duct and interacts with the metanephros which ascends on the posterior abdominal wall as the caudal embryo grows. Descend of the gonad occurs simultaneously.

Embryology

signalling molecules. The bladder develops from the cranial portion of the urogenital sinus with the distal mesonephric ducts, including the ureteric bud, forming the trigone. Ascent of the kidneys occurs between weeks 6e10 with the metanephros deriving an arterial supply initially from the common iliac artery then from the abdominal aorta. Urine production begins from week 10e12 of gestation.

The majority of the genito-urinary tract is derived from the intermediate mesoderm. Within this mesoderm the paired urogenital ridges develop into the nephrogenic cord and gonadal ridge. At week 4, the mesonephros appears and functions briefly as an excretory unit with the mesonephric tubules connected to the mesonephric ducts which lie laterally and open into the cloaca. The mesonephros degenerates by the end of the 12th week by which stage the metanephros has formed and begun its excretory function. The metanephros begins differentiation at week 5 and is induced by the penetration of the metanephros by the ureteric bud which arises from the distal end of the mesonephric duct (Figure 1). The metanephros continues to give rise to new generations of nephrons until 36 weeks gestation. The ureteric bud gives rise to the ureter, the renal pelvis and calyces and collecting ducts. This interaction between the ureteric bud and metanephros is key to normal renal parenchymal development and is controlled by several transcription factors and

Investigation of renal tract problems Most common paediatric renal tract problems (Table 1) can be investigated using four primary diagnostic studies.

Topics covered in this review Embryology Investigation of renal tract problems Pelvi-ureteric junction obstruction Duplex anomlies Multi-cystic dysplastic kidney Horseshoe kidney Vesico-ureteric reflux Vesico-ureteric junction obstruction Posterior urethral valves

Lisa C Steven MBChB FRCS (Paed Surg) is Specialist Registrar in Paediatric Urology at the Great Ormond Street Hospital for Children NHS Trust, London, UK. Conflicts of interest: none. Imran Mushtaq MBChB MD FRCS (Glasg) FRCS (Paed) is Consultant Paediatric Urologist at the Great Ormond Street Hospital for Children NHS Trust, London, UK. Conflicts of interest: none.

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Table 1

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Renal ultrasound scan A renal tract ultrasound scan (USS) evaluates the following: renal size and growth, structural anomalies, significant parenchymal thinning or scars, hydronephrosis, bladder capacity and post-void residual volume. The anterioreposterior (AP) diameter of the renal pelvis represents the distance between the renal cortex in the transverse plane and is reported in the context of hydronephrosis.

difficult to quantify but approximately 1:1000e1500 children will undergo an operation for this condition. The majority of patients are asymptomatic and are diagnosed after prenatal scanning has demonstrated hydronephrosis which is confirmed postnatally. Older children may present with intermittent loin pain, infection or haematuria. Renal USS and MAG-3 renogram aid the diagnosis and guide the definitive management. For children without symptoms, with moderate hydronephrosis (less than 20 mm) and absent or minimal calyceal dilatation, and a differential function more than 40% on MAG-3 study, it is acceptable to manage these cases conservatively. A significant proportion will show improving hydronephrosis, will maintain good function and will not require surgical intervention.

Micturating cysto-urethrogram A micturating cysto-urethrogram (MCUG) requires the insertion of a urethral catheter to allow the bladder to be filled with contrast. A MCUG demonstrates the size and shape of the bladder, presence of vesico-ureteric reflux (VUR), appearance of the bladder neck and must always include catheter-out voiding urethral views in boys.

Indications for surgery for PUJ obstruction In patients with a significant degree of hydronephrosis ( more than30 mm) or increasing hydronephrosis, where differential functional is less than 40% or in children with symptoms, then surgical intervention may be indicated.

DMSA scan A di-mercapto-succinic acid scan (DMSA) is a nuclear medicine study which evaluates renal tubular extraction. The radio-isotope is given intravenously and images are taken 2 h later. A DMSA demonstrates differential function, relative renal size and focal cortical thinning.

Pyeloplasty for PUJ obstruction The Anderson-Hynes pyeloplasty is the procedure of choice for children of all ages. The operation can be performed in an open manner or laparoscopically. The stenotic PUJ is excised and the distal ureter spatulated to allow a wide anastomosis to be performed between the renal pelvis and proximal ureter (Figure 2). The use of a trans-anastomotic stent varies considerably amongst clinicians. Complications are rare and include bleeding, infection, anastomotic leak and anastomotic stenosis. The procedure has an overall success rate of 95%. Repeat renal USS after 3e6 months typically shows a reduction in the degree of hydronephrosis but it usually takes several years for this to fall within normal limits. A repeat MAG-3 is performed 12 months after surgery to demonstrate improved drainage and preserved function of the kidney.

MAG3 renogram A di-mercapto-acetyltriglycine renogram (MAG3) nuclear medicine study evaluates renal tubular secretion. The radio-isotope is given intravenously and images are taken immediately afterwards. A MAG3 demonstrates differential function and drainage of the kidney. Studies performed in the context of renal pelvis dilatation require an assessment of the effect of change in posture and micturition on drainage. In children who are toilet trained and will void on demand, a MAG-3 indirect cystogram can assess for VUR without the need for catheterization.

Pelvi-ureteric junction obstruction Obstruction at the level of the pelvi-ureteric junction (PUJ) may be due to intrinsic narrowing of the PUJ, high insertion of the PUJ into the renal pelvis or due to extrinsic compression from accessory lower pole vessels or a combination of the above. The obstruction causes a reduction in urine flow and progressive renal dilatation, culminating in irreversible loss of nephrons. The incidence is

Duplex anomalies Duplex renal anomalies affect approximately 1% of the population and 30e40% of patients will have bilateral findings. For incomplete duplex kidneys the ureteric bud bifurcates and therefore there are two ureters at the level of the kidney but only

Figure 2 (a) MAG3 demonstrating equal split function, dilated left kidney and delayed drainage curve (red) of left kidney. (b) Intra-operative view of the narrow PUJ, dilated renal pelvis and normal calibre ureter.

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one ureteric orifice correctly sited within the bladder. Complete duplex anomalies occur less frequently and arise when two ureteric buds develop from the distal mesonephric duct and interact independently with the metanephros forming a kidney with an upper and a lower pole. The ureter relating to the lower pole has an abnormal lateral position within the bladder and is associated with vesico-ureteric reflux. The ureter of the upper pole may have an associated ureterocele (cystic dilatation of the intravesical ureter) which can cause obstructive symptoms. In some girls the upper pole ureter may be ectopic, inserting into either the urethra or vagina. Most cases are detected prenatally with hydronephrosis resulting from the ureterocele or reflux. Older children may present with recurrent UTI or dribbling urinary incontinence. Routine investigations will demonstrate the abnormality in most cases. A MCUG is recommended in all cases where a ureterocele is detected in order to demonstrate the relationship of the ureterocele to the bladder neck and the presence of lower pole VUR. Ectopic ureters can be very difficult to identify cystoscopically and in such cases magnetic resonance urogram (MRU) may demonstrate the abnormality.

incidence of MCDK is 1:2000e4000 live births. Most cases are sporadic and there may be associated contra-lateral VUR in up to 40% of patients. A MCDK is usually diagnosed prenatally with the finding of multiple cysts in the area of the renal bed with thin, abnormal parenchyma. Older children may be diagnosed following investigation for UTI, abdominal pain or a palpable abdominal mass. A MCDK has a characteristic USS appearance with no discernable function on DMSA. A MCUG can be performed to document contra-lateral VUR but this is recommended only in the context of contra-lateral ureteric dilatation. Management of multicystic dysplastic kidney The management of MCDK remains controversial. A significant proportion of MCDKs will involute but over a variable timescale. Some centres advocate routine removal in all cases but this is not widely accepted practice. Systemic hypertension and malignant transformation are reported, recognized to be rare, and remain the source of much debate regarding the need for prophylactic nephrectomy. The authors practice is to recommend an annual blood pressure check and not to offer prophylactic nephrectomy. For those patients with pain, recurrent UTI or hypertension attributable to the MCDK, nephrectomy is recommended. This is performed laparoscopically, very well tolerated and children can be discharged home within 24 h.

Surgical management of duplex anomalies The majority of children with an incomplete duplex anomaly will be asymptomatic. However, for complete duplex anomalies the surgical strategy depends on the age of the child and whether the issues relate to the ureterocele, ectopic ureter or VUR. In infants with a ureterocele causing significant dilatation of the upper pole, the first line of intervention can be cystoscopic puncture of the ureterocele. This is a relatively straightforward procedure but does carry a small risk of infection. Good decompression is confirmed by renal USS and if the child remains asymptomatic it may be that no further intervention is required, even if there is minimal or no function in the upper moiety. Children with recurrent UTIs and significant hydronephrosis will benefit from open or laparoscopic upper pole hemi-nephroureterectomy. This is also the procedure of choice for girls with dribbling incontinence due to an ectopic upper moiety ureter. In children in whom investigations have revealed significant VUR into a poorly functioning lower pole there are three possible strategies for management. If the child is asymptomatic they can be managed conservatively with antibiotic prophylaxis. A second option is to perform endoscopic injection of DefluxÔ with an associated 50e60% success rate, significantly lower than that for single system VUR. The remaining option is to perform open or laparoscopic lower pole hemi-nephroureterectomy which is favoured in those cases with minimal function in the lower pole and recurrent UTIs. With open or laparoscopic hemi-nephroureterectomy there is a small but definite risk of some loss of function of the remaining moiety. Approximately 5% of children having this procedure will have future symptoms relating to the distal ureteric stump and may require a further procedure to remove the stump.

Horseshoe kidney Horseshoe kidney occurs in approximately 1:500 of the general population. Normal ascent of the kidneys from the pelvis to the abdomen is prevented by fusion of the inferior poles thus limiting ascent to the level of the inferior mesenteric artery. This fusion also prevents the medial rotation of the kidneys such that the ureters pass anterior to the isthmus which is the fibrotic tissue connecting the inferior poles. The majority of patients with a horseshoe kidney are asymptomatic. However, the diagnosis may be established after USS in children who have suffered a UTI or abdominal pain. USS frequently demonstrates a degree of mild hydronephrosis of each moiety and MAG-3 renogram shows differential function and drainage of each moiety. In symptomatic children with significantly delayed drainage in one or both moieties a modified pyeloplasty will improve drainage and relieve symptoms.

Vesico-ureteric reflux Vesico-ureteric reflux (VUR) is the abnormal retrograde flow of urine from the bladder to the kidney. The incidence of primary VUR is 1e2% in the childhood population and occurs due to a congenitally abnormal, short and incompetent distal ureter as it passes through the bladder wall. As a consequence, during passive bladder filling and/or with detrusor contraction, the vesicoureteric junction (VUJ) is not occluded and reflux occurs (Figure 3). Secondary VUR is associated with bladder outflow obstruction, neuropathic bladder and voiding dysfunction, where the incompetence of the VUJ is due to raised intra-vesical pressure. Low pressure reflux of sterile urine should not lead to renal injury. Reflux of infected urine poses the greatest risk of renal scarring and the eventual triad of reflux associated nephropathy; vesicoureteric

Multicystic dysplastic kidney A multicystic dysplastic kidney (MCDK) arises due to abnormal interaction of the ureteric bud with the developing metanephros resulting in failure to induce normal nephrogenesis. The

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the most common material used for the procedure in the UK. The technique is most useful for grade IIIeIV VUR and the aim is to change the configuration of the refluxing ureteric orifice from an open shape to that of a crescenteric or slit-like orifice. This is a day case procedure with minimal morbidity but complications can occur. These include: urinary tract infection, dysuria, haematuria, persistent VUR and very rarely, obstruction of the distal ureter. The success rate of the technique is 80% for grade III VUR and 70% for grade IV VUR. Resolution of symptoms and improved hydroureteronephrosis on ultrasound are good indicators of success and this is confirmed with a MAG3-indirect cystogram. Persistent VUR may be managed with a further DefluxÔ injection if indicated. Antibiotic prophylaxis can be discontinued once it is deemed the VUR has been successfully treated.

Figure 3 (a) Normal long, oblique ureter as it passes through the bladder wall. (b) Abnormal short, incompetent refluxing ureter as it passes through the bladder wall.

reflux, renal scarring and hypertension. One should bear in mind that most reflux nephropathy is actually congenital in origin and is a form of renal dysplasia associated with VUR. High grade reflux may be suspected during the prenatal period with the finding of hydroureteronephrosis on prenatal scanning. In older children, VUR may be detected in 20e40% of cases investigated following a urinary tract infection (UTI). The sibling of an affected child has a 30% risk of also having VUR thus prompting the investigation of these children. The inheritance pattern is believed to be autosomal dominant with variable penetrance. A renal USS gives useful baseline information about the upper tracts and bladder. A MCUG is the gold standard for diagnosis and must always include urethral views with the catheter removed in boys. The degree of VUR is described according to the International Reflux Society (Table 2). In children who are toilet trained and will void on demand, a MAG3 indirect cystogram (MAG3-IRC) will demonstrate VUR without the need for catheterization, although the ability to grade the reflux is more difficult. A DMSA scan is recommended to assess differential function and demonstrate renal scars.

Open surgical correction of VUR Ureteric re-implantation is an alternative in those patients with high grade VUR, failed endoscopic injection and VUR associated with obstruction at the vesico-ureteric junction. The Cohen crosstrigonal ureteric re-implantation technique has a high success rate with minimal morbidity. Through a pfannensteil incision the bladder is isolated and opened in the midline. The refluxing ureteric orifice is identified and mobilized from within the bladder such that sufficient length is obtained to allow a crosstrigonal submucosal tunnel to be created. This has the effect of compressing the distal ureter with detrusor contraction and preventing reflux. Bilateral re-implantation can also be performed by the same technique. A good outcome is judged by resolution of symptoms and improved appearance on USS and nuclear medicine imaging. In cases where VUR is associated with a poorly (less than 10%) or non-functioning kidney a nephrectomy is usually indicated.

Vesico-ureteric junction obstruction Non-surgical management of vesico-ureteric reflux It is of paramount importance that all children with VUR maintain a good fluid intake (1e1.5 litre/day) of non-carbonated and noncaffeinated fluids. Toilet trained children should be encouraged to void every 3 h during the day and co-existing constipation should be treated. It remains best practice in the United Kingdom to initiate antibiotic prophylaxis in children with proven VUR to ensure that the refluxing urine is kept sterile. However, children over 1 year of age with low grade VUR, have been shown to have no greater risk of renal scarring without antibiotic prophylaxis. Prompt assessment and urine culture in the presence of suspected infection is recommended. The anticipated rate of spontaneous resolution of reflux is shown in Table 2.

Vesico-ureteric junction (VUJ) obstruction occurs as a functional partial obstruction at the level of the VUJ and is also termed primary obstructive non-refluxing megaureter. The incidence is approximately 1:10,000 live births and is caused by an aperistaltic segment in the terminal ureter or stenosis of the ureteric orifice. The majority of patients with VUJ obstruction are detected prenatally with hydroureteronephrosis. Older children may be diagnosed following investigation for UTI, haematuria, failure to thrive or abdominal pain. A renal USS, MCUG and MAG-3 renogram are required to establish the diagnosis. A ureter more than 10 mm in diameter on USS and an excretion curve showing delayed drainage on MAG3 renogram with urine pooling in the dilated ureter, confirms the diagnosis.

Surgical management of primary VUR Surgical intervention for primary VUR is reserved for those children who suffer recurrent breakthrough infections, have compliance issues with prophylaxis and in those with new onset renal scaring. Circumcision is strongly recommended for infant boys with high grade VUR as it reduces the risk of UTI.

Surgical management of vesico-ureteric junction obstruction In the infant with no symptoms, stable or decreasing dilatation and no functional discrepancy (e.g. more than 40%), it is safe to observe these children as one third of patients will show spontaneous resolution. Surgical management is reserved for those with severe or increasing dilatation, function less than 40% or deterioration in function and/or symptoms. There are three main surgical options.

Endoscopic correction of VUR This procedure involves general anaesthesia and cystoscopy with submucosal injection of a safe implantable material. DefluxÔ, is

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Classification of VUR based on MCUG

Grade

I

II

III

IV

V

Frequency % Spontaneous resolution

Ureter only 7% 80%

Ureter and calyces, no dilatation 53% 60%

Ureter and calyces, mild dilatation 30% 50%

Moderate ureteric and calyceal dilatation 6% 10%

Gross calyceal dilatation, tortuous ureter 2% 0%

Table 2

Ureteric stenting of the VUJ This option is now offered to infants with significant VUJ obstruction. It is believed that the presence of a stent across the VUJ allows remodelling and removes the obstructive component. The procedure is performed under general anaesthetic either cystoscopically or by an open technique. Cystoscopicaly the obstructed ureteric orifice is visualized and using fluoroscopic screening, a guidewire is placed through the ureteric orifice and a double-J stent is placed. This requires an overnight stay and complications include UTI, dysuria, intermittent haematuria, bladder spasms secondary to the presence of the stent and migration of the stent. Rarely, the ureter can be perforated necessitating open repair. The silicone stent is left in place for up to 12 months and cystoscopy is required to remove the stent. For the open technique a limited pfannensteil incision allows access to the VUJ and the stent is inserted under direct vision. The technique of stenting is suitable for all age groups and has a success rate between 50 and 60%. Once the stent is removed renal USS and MAG-3 study are required to ensure improvement in the dilatation and drainage of the kidney.

Ureteric re-implantation Ureteric re-implantation is offered either as a primary procedure if the child is approaching 1 year of age or when ureteric stenting has failed to resolve the VUJ obstruction. The initial approach is as for the Cohen re-implantation but the ureter is mobilized in the extravesical plane. The ureter is traced distally to the bladder level where is it divided and ligated. The distal narrow segment of the ureter is excised and re-implantation performed in the usual manner. A very dilated distal ureter may require tapering or plication prior to re-implantation. The success of ureteric reimplantation for VUJ obstruction is 90e95% with postoperative renal USS showing improvement in the degree of hydroureteronephrosis and improved drainage on MAG3.

Posterior urethral valves Posterior urethral valves (PUV) occur as an obstructing membrane at the level of the veru montanum in the male urethra. The incidence is approximately 1:5000 male live births. Embryologically PUV arise due to anomalous insertion of the mesonephric ducts into the urogenital sinus which prevents normal migration of the ducts and anterior fusion. Chronic renal impairment secondary to posterior urethral valves is the most common indication for renal transplantation in the paediatric population. The diagnosis is suspected prenatally in two-thirds of patients. Prenatal findings include a distended, thick walled bladder, bilateral hydroureteronephrosis, oligohydramonios and echo-bright kidneys. Late presenting features include UTI, failure to thrive, urosepsis and/or a palpable bladder or kidneys. Older boys may present with eneuresis, haematuria and voiding dysfunction. In neonates with a prenatal suspicion of PUV the diagnosis must be established as soon as possible. For those babies born in good condition they can be managed in the special care baby

Formation of end ureterostomy and subsequent ureteric re-implantation This procedure is considered a first line intervention for those infants less than 6 months of age with severe hydro-ureter (more than 20 mm) and recurrent UTIs. The procedure involves a limited pfannensteil incision and identification of the distal ureter in the extra-vesical plane. The distal ureter is brought to the skin surface as a small stoma (ureterostomy) which drains into a normal nappy. This procedure carries very minimal morbidity and ureteric re-implantation is performed around the age of one year.

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Figure 4 (a) MCUG demonstrating dilated posterior urethra, irregular bladder and bilateral reflux. (b) Endoscopic view of PUV with the sites for valve incision marked.

unit. Renal function should be checked, a urethral catheter (6 fr.) passed and antibiotic prophylaxis commenced e.g. trimethoprim 2 mg/kg/day. The observation of a normal urine stream or easy catheterization does not exclude the diagnosis. Close attention to fluid and electrolyte imbalances is mandatory and advice from a paediatric nephrologist recommended, as these children may have a high urine output with significant sodium and bicarbonate losses. An early renal USS will assess bladder wall thickness and upper tract dilatation. A MCUG will confirm the diagnosis and must always include adequate urethral views (Figure 4). It will also demonstrate the presence of VUR which is seen in 50% of patients. In older children, a MCUG is generally not well tolerated and for this reason the diagnosis requires a cystoscopy under general anaesthetic (GA).

effectively. Follow-up of all PUV patients continues throughout childhood and these boys must transition into adolescent care. End stage renal disease is seen in 30% of PUV patients. Patients with a prenatal diagnosis, bilateral high grade VUR, serum creatinine more than 100 umol/l at 1 year of age, proteinuria and ‘valvebladder’ all carry a worse renal prognosis. A

FURTHER READING Carroll D, Chandran H, Joshi A, McCarthy L, Parashar K. Endoscopic placement of double-J ureteric stents in children as a primary treatment for primary obstructive megaureter. Urol Ann 2010; 2: 114e8. Dhillon H. Prenatally diagnosed hydronephrosis: the Great Ormond Street experience. Br J Urol 1998; 81(suppl 2): 39e44. Ichikawa I, Kuwayama F, Pope JC, Stephens D, Miyazaki Y. Paradigm shift from the classic anatomic theories to contemporary cell biological views. Kidney Int 2002; 61: 889e98. Jodal U, Smellie JM, Lax H, Hoyer PF. Ten-year results of randomised treatment of children with severe vesicoureteral reflux. Final report of the International Reflux Study in Children. Pedaitr Nephrol 2006; 21: 785e92.

Surgical management of posterior urethral valves In the neonatal period, once there has been stabilization of the renal function and electrolyte balance, the baby will proceed to have cystoscopy and primary valve ablation under GA. Close inspection of the posterior urethra reveals the typical valve leaflets and these are incised in the 12, 5 and 7 o’clock positions (Figure 4). A urethral catheter is left in-situ for 24 h then removed. Renal function is re-checked at regular intervals and a renal USS is repeated prior to discharge. A check cystoscopy and circumcision are performed 3 months later. Baseline DMSA at the age of 3 months is recommended with regular follow-up with paediatric nephrology and urology at 3e6 monthly intervals. In some cases, a rising creatinine or worsening upper tract dilatation after primary valve ablation require formation of a vesicostomy or ureterostomy for preservation of upper tract function. Despite successful primary valve ablation approximately 30% of patients will develop the ‘valve-bladder syndrome’. These boys show a delay in achieving continence and demonstrate underlying bladder dysfunction. This bladder dysfunction may manifest as an unstable detrusor with high voiding pressures or detrusor failure where the bladder is large and does not empty

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Practice points C C

C

C

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Congenital renal tract disorders are common in childhood. Prenatal screening has identified an increasing number of children with renal tract anomalies, many of whom would have remained asymptomatic. Renal tract ultrasound scan, MCUG, DMSA and MAG3 scans are the primary investigations used to diagnose and monitor the common renal tract anomalies. Chronic kidney disease due to posterior urethral valves and reflux associated nephropathy account for a significant number of children requiring renal transplantation.

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