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444
a
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Expert comment Indications
for circumcision
The two ‘absolute’ medical
indications for circumcision are
BXO and recurrent balanoposthitis.
Clinicians should become familiar
with the appearance of the
former as it is under- recognized.
Balanoposthitis— oedema and
erythema of the prepuce and
glans— is likely overdiagnosed,
being confused for the frequent
and mild tip redness seen in many
phimotic boys.
Clinical tip BXO
Note the two classic features of
the prepuce affected by BXO.
The white ring of ‘scarred’ tissue
reflecting dermatological changes
and the ‘blunted’ appearance of
the prepuce when gently retracted
(e.g. no puckering is seen). The
prepuce is unable to be retracted
or protracted (Figure 46.3).
Challenging Concepts in Urological Surgery
Figure 46.1 Appearance of a physiological phimosis.
Learning point BXO
BXO, sometimes referred to as lichen sclerosis, is the hyperkeratinization of the epidermis, with
T- cell lymphocytic infiltration and loss of skin elasticity (Figures 46.2 and 46.3).
6,7
It presents with
tight white scarring of the foreskin, with white plaques visible on the prepuce and glans underneath.
In severe cases, the urethral meatus can be involved, as can the distal urethra. The most common
symptoms reported are phimosis, ballooning of the foreskin during micturition, dysuria, or recurrent
balanitis.7 BXO is rare in children <5 years old, with incidence being reported as 5– 6%.5 Typical age
of presentation is 9 years old.8 Many theories have been hypothesized for underlying aetiology,
none have proven correct. Symptoms such as preputial ballooning and dysuria can both be found in
physiological phimosis; the key for clinicians is to recognize from examination the differences between
the two conditions. In the former, symptoms generally improve as phimosis recedes, while in BXO, no
improvement can generally be expected.
Histology of BXO reveals hyperkeratosis, lamina propria thickening, and diffuse fibrosis with
lymphocytic infiltrate (Figure 46.2). Traditionally, diagnosis is clinical and confirmed on histology.
Circumcision has been the mainstay of treatment and is the best course to prevent recurrence.
Longer- term complications following circumcision for BXO include recurrence, meatal stenosis, and
urethral strictures.
Lymphocytic infiltrate of lamina propri
Figure 46.2 Histology of BXO.
445Case 46 Urethral pathology in children
https://t.me/med1917
Figure 46.3 Appearance of BXO.
In this patient, the symptom of dysuria persisted. Intermittently, he noted an
eschar forming at the end of the penis, which would dislodge when voiding. As
time progressed, the eschar would need to be peeled off before he could urinate.
He underwent an examination under anaesthesia, cystoscopy, meatoplasty, and biopsy 15 months post circumcision. Biopsy showed non- specific inflammation but
no BXO. Postoperatively, he was instructed to apply 0.1% Betnovate® to the meatus
and distal urethra using an 8- French (Fr) meatal dilator, which continued for 5
months.
The patient subsequently underwent four further cystoscopies and a urethral
meatus biopsy over 4 years as he developed terminal haematuria without resolution of his previous symptoms. Histology repeatedly showed chronic inflammation, without malignancy, infection, or BXO. At one cystoscopic examination at age
14, a bulbar urethral stricture was noted, which was dilated to accommodate a 14
Fr cystoscope from 9.5 Fr. He was also reviewed by a paediatric dermatologist who
prescribed fusidic acid ointment (Fucidin®) followed by Dermovate® with no change
in symptoms.
At age 15, the patient was referred for a second opinion. He described a pain made
worse when voiding frequently and he consequently developed a habitual tendency to
withhold urine. This behaviour had in fact predated the initial BXO diagnosis.
Expert comment Monitoring
for post- circumcision
complications
It has been debated how best to
monitor for post- circumcision
complications in patients with
BXO. Abnormal urinary stream can
be common in the early recovery
period in addition to the reality
that many of these patients have
contended with an abnormal
stream for many years prior to
surgery. A more sensitive marker is
the need for straining when voiding
which should prompt urgent
assessment.
446
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Learning point Meatal
stenosis
Narrowing of the urethral meatus,
meatal stenosis, is a cited long- term
complication of both circumcision
and BXO. The risk following
circumcision overall is low (0.7%)
but much greater in patients with
BXO, up to 20%.9 Typical symptoms
include initiation dysuria and
high- velocity, narrowed, upwards
diverging urinary stream.10
Treatment options include meatal
dilatation or meatoplasty.
11
Challenging Concepts in Urological Surgery
The patient underwent one further cystoscopy, which showed a 2 cm posterior urethral stricture. It is likely that high- pressure voiding due to meatal stenosis as well as
infrequent voiding resulted in an inflammatory reaction in the bulbar urethra, which
over time developed into a stricture. This would cause intermittent bleeding, creating
the appearance of an eschar as it coalesced at the meatus. Pain from the urethra would
be felt in the distal penile urethra. He will return in the near future for an anastomotic
urethroplasty using an inlay of buccal mucosa.
Learning point Causes and diagnosis of urethral strictures
Urethral strictures in children are rare and usually associated with trauma, specifically pelvic fracture
or straddle injuries.12 In children, the bladder and prostate sit higher than in adults and therefore are
more prone to disruption during trauma. Other causes include infection, post- hypospadias repair,
and meatal stenosis. Urethral strictures are rarely thought to be purely congenital in origin. One series
records a bimodal pattern of presentation, with those presenting under the age of 1 year typically
being investigated for bilateral antenatal hydronephrosis.
By contrast, in adults, the most common causes of strictures are iatrogenic, such as prolonged
catheterization during acute illness or idiopathic.14 This epidemiological picture has changed from
30 years ago, where a common cause included urethritis from sexually transmitted infection.
Diagnosis of stricture can be made on retrograde urethrography; however, incomplete filling or limited
studies due to discomfort can give spurious results.12 Direct visualization through cystourethroscopy
can also underestimate the degree of stenosis due to hydrostatic pressure stenting the urethral walls at
the time of procedure.
13
14
Learning point Management of urethral strictures
Management of strictures can be complex, with one series having 30% of patients with failed
interventions, and a further 11% requiring re- do repair following urethroplasty.
15
Surgical options for management include direct visual internal urethrotomy, excision and primary
anastomosis, or urethroplasty. Posterior urethral strictures, usually in the context of trauma, are
better suited to urethroplasty, rather than the minimally invasive urethrotomy, following a period of
suprapubic urinary diversion.
12
For anterior urethral strictures in adults, direct visual internal urethrotomy (DVIU) is a treatment
option, especially for short (<2 cm) strictures.16 With a success rate of 55%, the authors of that series
advocate one single attempt at DVIU; there is concern that this technique may exacerbate the
underlying scar formation thereby lengthening strictures.17 Similar outcomes with DVIU are noted in
13
children.
By comparison, excision and primary anastomosis has a success rate up to 98%.16 The exception
Expert comment Urethra
passage
It is important for clinicians not to
compartmentalize the urethra into
unconnected distal and proximal
components. The nature of the
urethra as a passage, is to transport
both physiological and pathological
fluids, discharging them distally
but also translating pressure from
obstructions proximally.
recurrence.
Conclusion
Concerns of the foreskin can cause much work for primary and secondary care alike;
the vast majority of concerns relate to physiological phimosis and reassurance is usually sufficient. Absolute indications for circumcision include pathological phimosis, of
which BXO is an uncommon cause in paediatrics. Strictures are rare in children and
lies in the subgroup of congenital strictures; these appear to respond well to DVIU without stricture
13
can be complex to resolve but it is important to recognize the linear nature of the urethra: what one sees at its distal end can be an indicator of proximal disease.
A final word from the expert
https://t.me/med1917
The penis and foreskin generate a great deal of anxiety for patients and doubly so for their
parents. This is reflected in the number of referrals for foreskin- related concerns to a paediatric
urology clinic, the vast majority of which will be diagnosed with physiological phimosis and
reassured. The success of said reassurance is to some extent dependent on expectations of
circumcision. We would generally advocate two things. The first is to steer families away from
comparing male siblings as the prepuce develops differently in each child and second, for
clinicians to avoid where possible medicalizing cases of physiological phimosis. This can be
harder said than done with the temptation to reschedule 6- monthly or yearly appointments to
monitor the situation. This can be a rather frustrating experience for families and clinicians as
the prepuce often shows little to no change between appointments, nudging clinicians towards
advocating for intervention. If possible, it may be better to set out expectations, describe the
symptoms of balanoposthitis, and warn parents of new- onset ballooning or dysuria where
previously not present as an indicator for BXO. In the absence of these, it may be better to
lengthen appointment intervals or aim to reschedule them nearer to puberty.
When concerning symptoms are present, such as in the case of this boy, we would encourage
clinicians to think of the urethra as a single unit, with signs of disease generally transmitted along
its length in either direction.
447Case 46 Urethral pathology in children
References
1. Yardley IE, Cosgrove C, Lambert AW. Paediatric preputial pathology: are we circumcising
enough? Ann R Coll Surg Engl. 2007;89(1):62– 65.
2. Gairdner D. The fate of the foreskin: a study of circumcision. BMJ. 1949:2(4642):1433– 1437.
3. Liu J, Yang J, Chen Y, et al. Is steroids therapy effective in treating phimosis? A metaanalysis. Int Urol Nephrol. 2016:48(3):335– 342.
4. Oster J. Further fate of the foreskin. Incidence of preputial adhesions, phimosis, and
smegma among Danish schoolboys. Arch Dis Child. 1968;43(228):200– 203.
5. Boksh K, Patwardha N. Balanitis xerotica obliterans: has its diagnostic accuracy improved
with time? JRSM Open. 2017;8(6):2054270417692731.
6. Jayakumar S, Antao B, Bevington O, et al. Balanitis xerotica obliterans in children and its
incidence under the age of 5 years. J Paediatr Urol. 2012;8(3):272– 275.
7. Celis S, Reed F, Murphy F, et al. Balanitis xerotica obliterans in children and adolescents: a
literature review and clinical series. J Paediatr Urol. 2014;10(1):34– 39.
8. Rickwood AMK, Hemalatha V, Batcup G, et al. Phimosis in boys. Br J Urol.
1980;52(2):147– 150.
9. Morris BJ, Krieger JN. Does circumcision increase meatal stenosis risk? A systematic review
and meta- analysis. Urology. 2017;110:16– 26.
10. Persad R, Sharma S, McTavish J, et al. Clinical presentation and pathophysiology of meatal
stenosis following circumcision. Br J Urol. 1995;75(1):91– 93.
11. Das S, Siva H, Tunuguntla GR. Balanitis xerotica obliterans— a review. World J Urol.
2000;18(6):382– 387.
12. Priyadarshi R, Mohd A, Manmeet S, et al. Post- traumatic urethral strictures in children: what have we learned over the years? J Paediatr Urol. 2012;8(3):234– 239.
13. Banks FC, Griffin SJ, Steinbrecher HA, et al. Aetiology and treatment of symptomatic idiopathic urethral strictures in children. J Pediatr Urol. 2009;5(3):215– 218.
14. Lumen N, Hoebeke P, Willemsen P, et al. Etiology of urethral stricture disease in the 21st
century. J Urol. 2009;182(3):983– 987.
448
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Challenging Concepts in Urological Surgery
15. Helmy TE, Sarhan O, Hafez AT, et al. Perineal anastomotic urethroplasty in a pediatric
cohort with posterior urethral strictures: critical analysis of outcomes in a contemporary
series. Urology. 2014;83(5):1145– 1148.
16. Hillary CJ, Osman NI, Chapple CR. Current trends in urethral stricture management. Asian
J Urol. 2014;1(1):46– 54.
17. Fenton AS, Morey AF, Aviles R, et al. Anterior urethral strictures: etiology and characteristics. Urology. 2005;65(6):1056– 1058.
47
(a) (b) (c)
(d)
(g)
CASE
https://t.me/med1917
Vesicoureteral reflux
in children
María S. Figueroa- Díaz and Alexander Cho
Expert commentary Imran Mushtaq
Case history
A male neonate was referred with an antenatal diagnosis at 31 weeks’ gestation of
bilateral hydronephrosis (right anterior– posterior diameter (APD) 21 mm and left APD
17 mm) with a distended bladder.
He was born at term via vaginal delivery with normal APGAR (Appearance, Pulse,
Grimace, Activity, and Respiration) scores. His initial serum creatinine level was elevated
at 102 μmol/ L. A urethral catheter was inserted soon after birth to aid bladder drainage.
The ultrasound scan (USS) on day 4 confirmed the right kidney had a stretched cortex
with bright echogenicity. The right APD was 12 mm with a tortuous ureter measuring
19 mm proximally and 17 mm distally. The left renal APD was 11 mm and the ureter
measured 9 mm. The bladder wall was noted to be thick- walled (Figure 47.1). The spine
USS was normal. A micturating cystourethrogram (MCUG) demonstrated a small- volume
trabeculated bladder, vesicoureteric reflux (VUR) into a grossly distended right kidney
(grade V), but no left- sided reflux. The urethra was noted to be normal (Figure 47.2).
Learning point Antenatal
hydronephrosis
Antenatal hydronephrosis (ANH) is
most commonly graded according
to the APD of the renal pelvis1:
● Mild: 4 to <7 mm (second
trimester); 7 to <9 mm (third
trimester).
● Moderate: 7 to ≤10 mm (second
trimester); 9 to ≤15 mm (third
trimester).
● Severe: >10 mm (second
trimester); >15 mm (third
trimester).
There is uniform agreement that an
APD >15 mm in the third trimester
represents severe hydronephrosis.
(e) (f)
Figure 47.1 USS of the kidneys, ureters, and bladder. (a) Right kidney, transverse; (b) right kidney,
longitudinal; (c) right distal ureter; (d) left kidney, transverse; (e) left kidney, longitudinal; (f ) left distal
ureter; (g) thick- walled bladder.
Clinical tip Initial
management of neonatal
bilateral hydronephrosis
In a male neonate with antenatally
diagnosed bilateral hydronephrosis
and distended bladder, the
condition of posterior urethral
valves (PUV) needs to be excluded.
Initial management, however, is the
insertion of a urethral catheter and
appropriate fluid management.
450
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Challenging Concepts in Urological Surgery
Figure 47.2 MCUG showing right- sided grade V VUR but a normal urethra.
Clinical tip Postnatal imaging
The first postnatal imaging study recommended is an USS. This should be performed after 48 hours
of life to compensate for the initial neonatal dehydration and postnatal oliguria. An early USS may
underestimate the severity of the hydronephrosis.
The distension of the urinary tract in the postnatal USS can be affected by the degree of bladder
fullness and hydration. It is recommended that in the presence of urinary tract dilation, the patient
should be rescanned after bladder emptying.
The Society for Fetal Urology grading system is commonly used for infant hydronephrosis and is based
on the appearance of the renal pelvis, calyces, and renal parenchyma rather than the size of the renal
pelvis (Table 47.1).
Table 47.1 Society for Fetal Urology grading of infant hydronephrosis
Pattern of renal
sinus. Splitting
SFU grade 0 No splitting
SFU grade 1 Urine in pelvis
barely splits sinus
SFU grade 2 Urine fills pelvis
with/without
major calyces
dilated.
Ultrasound variants
Table 47.1 Continued
https://t.me/med1917
451Case 47 Vesicoureteral reflux in children
Pattern of renal
sinus. Splitting
SFU grade 3 SFU grade 2 and
minor calyces
dilated and
parenchyma
preserved.
SFU grade 4 SFU grade 3 and
parenchyma thin.
Adapted from Nguyen et al. J Pediatr Urol. (2014).
Learning point Differential diagnosis
There are multiples aetiologies for ANH with the majority (50– 70%) due to transient or physiological
hydronephrosis with no clinical significance.1 In those fetuses with mild ANH, 88.1% will have transient
hydronephrosis. The incidence of any significant postnatal pathology is 11.9% for mild, 45.1% for
moderate, and 88.3% for severe ANH.2 The most frequent diagnoses are1:
● Transient/ physiological: incidence 50– 70%
● Ureteropelvic junction obstruction: 10– 30%
● VUR: 10– 40%
● Ureterovesical junction obstruction/ megaureter: 5– 15%
● Multicystic dysplastic kidney disease: 2– 5%
● Posterior urethral valves: 1– 5%
● Ureterocoele, ectopic ureter, duplex system, urethral atresia, Prune belly Syndrome, polycystic
kidney diseases: Uncommon (<1%).
Ultrasound variants
1
Learning point VUR
For an asymptomatic infant monitored for ANH, the estimated prevalence of VUR ranges from 10–
15% if postnatally there is absent or mild hydronephrosis,3 up to 40% if there are postnatal anomalies
detected on USS including hydronephrosis, renal cysts, or renal agenesis.4 A normal postnatal USS,
therefore, does not exclude VUR. The grading of VUR is shown in Figure 47.3.
MCUG is the gold standard in the diagnosis of VUR. Reflux can occur during filling or voiding.
Reflux during filling has been considered more severe because it occurs at low bladder pressures
and this may be a poor prognostic sign for VUR resolution. There is a significant risk of urinary tract
infections (UTIs) associated with the MCUG test and peri- investigation antibiotics are recommended.5
Radionuclide studies for detection of reflux offer a lower radiation exposure than MCUG but the
anatomical details demonstrated are inferior.
6
452
III III IV V
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Challenging Concepts in Urological Surgery
Figure 47.3 Classification of VUR based on the MCUG. (I) Reflux into a non- dilated ureter; (II) into
the renal pelvis and calyces without dilatation; (III) mild to moderate dilatation of the ureter, renal
pelvis, and calyces with minimal blunting of all the fornices; (IV) moderate ureteral tortuosity and
dilatation of the pelvis and calyces; (V) gross dilatation of the ureter, renal pelvis, and calyces; loss of
papillary impressions and ureteral tortuosity.
Adapted from Lebowitz RL et al. Pediatr Radiol. (1985).
7
Incomplete bladder emptying was detected during a bladder function assessment that combined nappy alarms with residual bladder volumes detected by USS.
Clean intermittent catheterization was initiated with oral trimethoprim prophylaxis. The patient’s serum creatinine improved to normal levels corrected for age
and weight.
Expert comment Primary versus secondary VUR
It is important to differentiate between the two types of VUR as their management differs
significantly. This is demonstrated in this case study where the bladder seemed abnormal.
Primary VUR is due to a congenital abnormality of the antireflux mechanism at the
vesicoureteric junction (VUJ) where the ratio between intramural ureter and its diameter
is <5:1.8 In secondary VUR, the VUJ is normal, but there is an abnormality of the bladder,
bladder outlet, or urethra. Any obstructive bladder process, whether functional or anatomical,
can produce high vesical pressures during storage and emptying that exceed the antireflux
mechanism resulting in VUR.
The most common anatomical obstruction of the bladder in male infants is PUV. VUR is present in
48– 70% of patients with PUV, but after PUV ablation, VUR resolution can reach 78%.9 Functional
causes of reflux must be excluded including a neurogenic bladder associated with spina bifida.
When evaluating the patient, direct questioning must be made about constipation (and faecal
incontinence if older), as well as an examination of the lumbosacral area. Spinal cord abnormalities
can be excluded by spinal ultrasonography in young infancy or by a magnetic resonance imaging
scan if older. The prevalence of bladder dysfunction in the VUR population varies and is seen in
18– 52% of non- invasive investigations.10 Abnormal micturition patterns without an indefinable
neurological abnormality are important to identify. There is a spectrum of functional disorders
known as ‘dysfunctional elimination syndrome’ with the most severe extreme labelled as ‘nonneurogenic neuropathic bladder’. It presents both lower and upper urinary tract deterioration, but
without evidence for neurological disease.
Expert comment High voiding detrusor pressure
https://t.me/med1917
Relevant to our case study, it has been reported that high voiding detrusor pressure in some infants
with VUR may be related to inadequate relaxation of the external urethral sphincter.11 In a normal
immature bladder at infancy, maximum voiding pressure is higher than later in life. Asynchronous
detrusor/ sphincter activity is often noted characterized by a low bladder capacity, high voiding
pressure, and overactivity during filling. The detrusor contraction with a closed urethral sphincter
results in high bladder pressure and may lead to VUR depending on the competence of the VUJ. This
dyscoordination is not considered a true neurogenic bladder because it is an immature bladder in
maturation12 and occurs more frequently in boys than girls.
Learning point Management of VUR
The primary goal of the treatment of VUR is prevention of febrile UTI and avoidance of renal
damage. The management of VUR includes conservative management or surgical intervention
which commonly comprises endoscopic correction or ureteric reimplantation.
Conservative management is based on the knowledge that VUR resolves spontaneously
mostly in the younger patients with low- grade reflux due to the enlargement of the
intravesical ureter and maturation of the antireflux mechanism. However, in high- grade reflux,
spontaneous resolution is <25%13 and when associated with bladder dysfunction this rate is further
reduced.
Evidence base Continuous antibiotic prophylaxis
Continuous antibiotic prophylaxis (CAP) in the context of children with VUR is also much debated.
A meta- analysis of randomized controlled trials concluded that CAP significantly reduced the risk of
febrile and symptomatic UTIs. However, CAP increased the risk of UTI secondary to antibiotic- resistant
bacteria and did not significantly impact the occurrence of new renal scarring.
Supported by the European Society of Paediatric Urology, a safe approach would be to use CAP in
most cases though decision- making would be influenced by risk factors for UTI (young age, high- grade
VUR, status of toilet training, lower urinary tract dysfunction, female sex, and circumcision status) and
parental opinion.
6
14
453Case 47 Vesicoureteral reflux in children
At 1 month of age, following a culture- proven UTI, the patient was also started
on overnight bladder drainage. A dimercaptosuccinic acid (DMSA) scan at 2 months
of age showed 14% right kidney differential function. Due to difficulties with clean
intermittent catheterization and overnight drainage, at 2 months of age, a diagnostic
cystoscopy was undertaken that demonstrated a normal urethra and excluded PUV.
A right- sided refluxing loop ureterostomy was formed and a circumcision was also
undertaken.
Learning point Endoscopic correction
This minimally invasive approach consists of a cystoscopic subureteral injection of biocompatible
bulking material to elevate the distal ureter and narrows the lumen which prevents VUR. The reflux
resolution rate depends on the grade of reflux: grades I and II are 78.5%, and 51% for grade V. The
success rate is lower in neurogenic bladder.15 Endoscopic injection is safe, but a serious complication
reported is VUJ obstruction in 0.1– 5% of cases. It can present acutely or years later, highlighting the
need for long- term follow- up.
Evidence base Circumcision
to reduce the risk of UTI
The health benefits of routine
newborn male circumcision remain
controversial.18 Less controversial
is the role of circumcision in
patients at high risk of UTI. The
number needed to treat to prevent
a UTI in a normal boy is 111 but
in those with high- grade reflux,
this number drops to 4.19 When
VUR is confirmed following ANH,
circumcision reduces the incidence
of UTI from 68% to 22%.
20
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