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\begin{document}

\title{Urethral diverticula in a 26-year-old gelding: a unique case report}



\author[1]{Lisa-Marie Hermans}%
\author[1]{Laura Borde-Doré}%
\author[1]{Bianca Drumond}%
\author[2]{Jean-Luc Cadoré}%
\affil[1]{VetAgro Sup}%
\affil[2]{Université de Lyon, VetAgro Sup, Campus Vétérinaire de Lyon}%


\vspace{-1em}



  
  \date{May 26, 2024}


\begingroup
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\selectlanguage{english}
\begin{abstract}
Urethral diverticula have been described in other species. Congenital
anomalies of the urinary tract in horses are rare. A 26-year-old gelding
presented with complaints of abdominal discomfort and dysuria. Urine
dribbling was reported since several years. General clinical examination
was within normal limits. Rectal palpation showed a distended bladder,
which was emptied spontaneously after manipulation. Urine cytology
showed an increased white blood cell count and presence of bacteria.
Bacterial culture however remained sterile. Cystoscopy identified two
symmetrical urethral diverticula dorsal to the entrance of the bladder,
lined with normal epithelium. Mucosa of the bladder, urethra and two
diverticula were hyperemic with evidence of urine accumulation in both
diverticula. Ultrasound, abdominal and transrectal, did not identify the
diverticula. Treatment included trimethoprim-sulfamides and meloxicam.
After 2 weeks, the owner reported resolution of clinical signs. It is
unclear if the diverticula in this case were congenital or acquired. In
humans it is described that urethral diverticula can be asymptomatic and
incidental findings. Surgical intervention has been described in small
animal and human medicine with complete resolution of clinical signs,
however in this case was not possible due to financial restraints.%
\end{abstract}%



\sloppy


Urethral diverticula in a 26-year-old gelding: a unique case report

Urethral diverticula

L.M. Hermans, L. Borde-Dore, B. Drumond, J.L. Cador\selectlanguage{ngerman}é

\emph{Equine Department, VetAgro Sup, University of Lyon, Marcy l'Etoile
69280, France.}

\emph{lisa-marie.hermans@vetagro-sup.fr}

\textbf{Keywords: horse ; urethral diverticula ; congenital ; urinary
incontinence ; colic}

\textbf{Summary}

Urethral diverticula have been described in other species. Congenital
anomalies of the urinary tract in horses are rare.

A 26-year-old gelding presented with complaints of abdominal discomfort
and dysuria. Urine dribbling was reported since several years. General
clinical examination was within normal limits. Rectal palpation showed a
distended bladder, which was emptied spontaneously after manipulation.
Urine cytology showed an increased white blood cell count and presence
of bacteria. Bacterial culture however remained sterile. Cystoscopy
identified two symmetrical urethral diverticula dorsal to the entrance
of the bladder, lined with normal epithelium. Mucosa of the bladder,
urethra and two diverticula were hyperemic with evidence of urine
accumulation in both diverticula. Ultrasound, abdominal and transrectal,
did not identify the diverticula. Treatment included
trimethoprim-sulfamides and meloxicam. After 2 weeks, the owner reported
resolution of clinical signs.

It is unclear if the diverticula in this case were congenital or
acquired. In humans it is described that urethral diverticula can be
asymptomatic and incidental findings. Surgical intervention has been
described in small animal and human medicine with complete resolution of
clinical signs, however in this case was not possible due to financial
restraints.

\textbf{Main text}

\textbf{Introduction:}

Urethral diverticula have been described in other species, and to our
knowledge there is only one report of a urethral diverticulum in a
breeding stallion (Diel de Amorim \emph{et al.} 2014).

In human medicine, urethral diverticula are described as epithelized
outpouchings of the urethral lumen into surrounding periurethral fascia
(Crescenze \emph{et al.} 2015; El-Nashar \emph{et al.} 2014;
Reeves\emph{et al.} 2014). It is a rare condition that affects mainly
adult women with a reported incidence between 1 and 6\% (Crescenze
\emph{et al.} 2015; El-Nashar \emph{et al.} 2014). There are only few
reports of diverticula in males (Thakur \emph{et al.} 2016; Cinman
\emph{et al.}2012; Popoola \emph{et al.} 2009; El Ammari \emph{et al.}
2012; Mohanty\emph{et al.} 2014). Diverticula are congenital or acquired
and the latter is the most frequent. Rupture of periurethral glands into
the urethral lumen, secondary to repeated obstruction and infection is
thought to be the main etiology for acquired cases (Huffman 1948) but
trauma may also play an important role (Reeves \emph{et al.} 2014). Most
of the diverticula reported in human medicine are located in the
mid-to-distal third of the urethra. This location reinforces the theory
of ruptured periurethral glands, since these are draining in the distal
one-third of the urethra (Lang et al.; MacKinnon et al.).

Reported clinical signs in humans are non-specific and include dysuria,
dyspareunia, incontinency and/or discharge (Romanzi \emph{et al.} 2000).
They have also been reported to be incidental findings
(Crescenze\emph{et al.} 2015; El-Nashar \emph{et al.} 2014One-third of
the patients have recurrent urinary tract infections (Ganabathi \emph{et
al.} 1994; Davis and Robinson 1970). To our knowledge only 4 cases have
been described in small animals and clinical signs included: perineal
swelling, dysuria, stranguria, urinary incontinence and urinary tract
infections (Henry \emph{et al.} 2018; Foster \emph{et al.} 1999; Atilla
2018; Watanabe \emph{et al.} 2015). Distal/penile urethral diverticula
have been reported in goat kids and calfs and are frequently associated
with hypospadias (Simon \emph{et al.} 2010; Temi\selectlanguage{english}zsoylu
2005\textbf{;}Bokhari 2013; Sylla \emph{et al.} 2019; Abd-El-Hady 2014;
Maiti \emph{et al.} 2018). The only case described in a stallion by Diel
de Amorin et al. showed blood dripping from the penis after a breeding
attempt. The stallion had a history of a previous history of breeding
injury with a surgical repair of the urethra.

Diagnosis can be made in other species using cystourethroscopy, double
balloon positive pressure urethrography, voiding cystourethrogram,
Magnetic Resonance Imaging, computed tomography scan and transperineal
and transvaginal ultrasound (Crescenze \emph{et al.} 2015; Stav \emph{et
al.} 2008; Gugliotta \emph{et al.} 2015; Romanzi \emph{et al.} 2000;
Kim\emph{et al.} 1993; Ljungqvist \emph{et al.} 2017; Ockrim \emph{et
al.}2009; Pathi \emph{et al.} 2013; Dwarkasing \emph{et al.} 2011;
Blander\emph{et al.} 2001). Various surgical techniques have been
described in human medicine (Greiman \emph{et al.} 2019) and successful
surgical therapy has been reported in other species (Henry \emph{et al.}
2018; Foster \emph{et al.} 1999; Atilla 2018; Watanabe \emph{et al.}
2015; Simon \emph{et al.} 2010; Temi\selectlanguage{english}zsoylu 2005\textbf{;} Bokhari 2013;
Sylla\emph{et al.} 2019; Abd-El-Hady 2014; Maiti \emph{et al.} 2018).
Conservative therapy has also been described (Lee and Fines 2005; Ellik
1957; Mizrahi and Bitterman 1988). This case report describes a unique
case of 2 urethral diverticula in a 26-year-old gelding.

\textbf{Case history:}

A 26-year-old gelding presented signs of dysuria and a few hours later,
signs of abdominal pain, not responding to the administration of
metamizole. The gelding had a history of urinary incontinence in the
last 6 years and also had a history of urolithiasis. There was no
previous history of colic. He was referred to the hospital for further
investigation of the colic with associated dysuria.

\textbf{Clinical findings:}

Upon presentation at the clin\selectlanguage{ngerman}équine of the Vetagro Sup Lyon, the gelding
was bright and alert, weighed 518 kg and his body condition score was
estimated at 6/9. The mucous membranes were slightly icteric and the
capillary refill time was slightly elevated at 3 seconds. The
gastrointestinal borborygmi were decreased in the upper left part of the
abdomen. His rectal temperature was within normal limits (37,3°C). His
heart rate and respiratory rate were within normal limits (48 beats per
minute and 20 breaths per minute) and auscultation was normal. He showed
signs of dysuria and a urinary catheter was placed. A distal obstruction
with sediment was present and urine was taken by catheterization for
bacteriology and cytology. Urine cytology showed an increased white
blood cell count (20-30 per chamber) and the presence of multiple
bacteria and carbonates. Bacterial culture however remained sterile. On
rectal palpation, a distended bladder was present that could be voided
by manipulation. Abdominal ultrasound showed no abnormalities. A
complete blood count revealed a mild neutrophilic leukocytosis (WBC:
12,83 x 10\^{}9/L; neutrophiles: 9,39 x 10\^{}9/L). Biochemistry
revealed no abnormalities. The venous blood gas revealed a slight
hypokalemia (2,7 mmol/L).

\textbf{Treatment and additional findings:}

The gelding was hospitalized for observation and during the night no
dysuria was observed. Rectal palpation was repeated next day and
revealed no abnormalities. A cystoscopy was performed which revealed the
presence of 2 urethral diverticula dorsal to the entrance of the
bladder, lined with normal epithelium (figure 1 and 2). The mucosa of
the bladder, the urethra and the two diverticula were hyperaemic with
evidence of urine accumulation in both diverticula. No ectopic ureter
was identified. There was normal voiding of urine from the ureters and
presence of sediment in the bladder. After cystoscopy, the rectal
palpation was repeated and revealed a gas distended structure dorsal to
the bladder. Transrectal ultrasound showed a gas distended structure and
a diffuse thickening of the bladder wall. The gas distended structure
was suspected to be the urethral diverticula filled with gas, however,
the extent of the diverticula could not be estimated. The complete blood
count was repeated and showed a moderate neutrophilic leucocytosis (WBC:
14,14 x 10\^{}9/L; neutrophiles: 11,2 x 10\^{}9/L) and the blood gas
analysis confirmed the slight hypokalaemia (2,8 mmol/L). Treatment was
initiated with broad spectrum antibiotics (trimethoprim-sulfamides, 25
mg/kg PO BID) and anti-inflammatories (meloxicam, 0,6 mg/kg PO SID) to
treat the present urinary infection. No further dysuria was observed and
next day the repeat transrectal palpation and transrectal ultrasound
didn't allow identification of the diverticula No abnormalities of both
kidneys were identified during transabdominal ultrasound. On day 2 of
hospitalisation, the cystoscopy was repeated. The mucosa of the bladder,
the urethra and the two diverticula were less hyperaemic (figure 3 and
4) but urine accumulation in the diverticula was still present. Sediment
in the bladder was also still present and a lavage with an isotonic
polyionic solution (lactated Ringer) was performed. Surgical exploration
of the diverticula was declined by the owner due to financial
restraints. One day later, the gelding was discharged from the hospital
with systemic treatment.

\textbf{Outcome:}

Follow-up examination was not performed but a telephone call 2 weeks
after discharge revealed a complete resolution of the clinical signs,
including resolution of the permanent urine dribbling, according to the
owner.

\textbf{Discussion:}

This case report describes a case of 2 urethral diverticula in a
26-year-old gelding. There is one other case that reports a urethral
diverticulum in a breeding stallion (Diel de Amorim \emph{et al.} 2014)
and one report that describes a vesical diverticulum in a 6-year-old
gelding (Odenkirchen \emph{et al.} 1994).

The origin of the diverticula in the present case could not been
determined but a congenital origin was strongly suspected. The horse was
owned since the age of 2 years by the owner and no previous
catheterization or trauma, except for the urine dribbling, were
reported. Also, the diverticula were symmetrical, which made an acquired
origin less likely.

However, in human medicine, in males, acquired diverticula are more
frequently reported than congenital (Cinman \emph{et al.} 2012;
Popoola\emph{et al.} 2009; El Ammari \emph{et al.} 2012; Mohanty
\emph{et al.}2014). The case described by Diel de Amorim \emph{et al} .
(2014) was suspected to have an acquired urethral diverticulum due to a
breeding injury.

Congenital anomalies of the urinary tract in horses are rare although
some are reported such as ureteral ectopia, ureteral defects, renal
dys-/hypoplasia, polycystic kidney disease and rectovaginal or
rectourethral fistula (Chaney 2007). If the lesion in this case was
congenital, the reason why the horse did not show clinical signs before
is unknown. In women however, it has also been described that a urethral
diverticulum can be asymptomatic and an incidental finding
(Crescenze\emph{et al.} 2015; El-Nashar \emph{et al.} 2014).

The main hypothesis in order to explain the permanent dribbling of the
urine is that, during voiding of the bladder, the diverticula fill up
with urine and that there is a slow emptying caused by gravity
afterwards. This was also a hypothesis in the case report of
Henry\emph{et al} . in a cat with urinary incontinence. The stagnation
of the urine in the diverticula was also thought to have caused the
urinary tract inflammation. Another hypothesis is that the urinary tract
inflammation was responsible for the permanent dribbling of the urine
but this theory seems less likely because the horse showed signs of
urinary incontinence for 6 years and the signs of urinary tract
infection were recent. Dysuria was probably caused by the urinary tract
infection, which could have also been responsible for the reported signs
of colic. Another hypothesis for the colic could have been pain, caused
by urine accumulation but other none identified causes could not be
excluded.

In human medicine, described techniques for diagnoses are
cystourethroscopy, voiding cystourethrogram, MRI, CT and transperineal
and transvaginal ultrasound.

Different reports in humans demonstrate a variable ability, 15-89\%, to
visualize the ostium with cystourethroscopy (Crescenze \emph{et
al.}2015; Romanzi \emph{et al.} 2000; Kim \emph{et al.} 1993;
Ljungqvist\emph{et al.} 2017). Diagnosis in this case was purely based
on cystourethroscopy, as in the described case of a breeding stallion
(Diel de Amorim \emph{et al} . 2014).

Another modality often used in human medicine is ultrasound. Some
studies however have shown that the sensitivity is depending on the
skills of the sonographer, some studies even report that the sensitivity
is lower than 50\% when evaluating cases with confirmed urethral
diverticulum (Crescenze \emph{et al.} 2015; Reeves \emph{et al.} 2014).
Other studies showed a sensitivity of transvaginal and transperineal
ultrasound of 95-100\% (Crescenze \emph{et al.} 2015; Stav \emph{et
al.}2008; Gugliotta \emph{et al.} 2015). In the here reported case, we
were however unable to identify the urethral diverticula with
transrectal and transabdominal ultrasound. Visualization of the urinary
tract by transperineal ultrasound in horses is not possible due to the
pronounced muscle mass in this region. In one of the previous described
cases in a kitten, transabdominal ultrasound did not reveal any
abnormalities and also in this case, no transvaginal or transperineal
ultrasound were performed (Henry et al.). In the stallion described by
Diel de Amorim\emph{et al.} (2014), the authors were able to identify
the diverticulum with penile ultrasound, in our case the diverticula
were localized too high for this technique

Voiding cystourethrogram is another described imaging modality with a
reported sensitivity of 67-95\% (Crescenze \emph{et al.} 2015;
Ockrim\emph{et al.} 2009). This technique and retrograde contrast
cystourethrogram were used successfully in small animals (Henry \emph{et
al.} 2018; Foster \emph{et al.} 1999; Atilla 2018; Watanabe \emph{et
al.} 2015). In this case, the use of a contrast cystourethrogram could
have helped to determine the extent of the diverticula. However, the use
of abdominal radiographs in adult horses is limited to the ventral part
of the abdomen to identify sand impaction and enteroliths.

The ``gold standard'' for diagnosis of urethral diverticula in humans is
MRI (Pathi \emph{et al.} 2013; Dwarkasing \emph{et al.} 2011;
Blander\emph{et al.} 2001). In none of the described cases in small
animals, this technique was used and neither in this case. In one case
of a dog, CT was successfully used to identify the diverticulum.

In this case, due to financial restraint and the age of the horse, a
medical treatment with large spectrum antibiotics and anti-inflammatory
drugs was attempted with resolution of the clinical signs according to
the owner.

Treatment in human medicine consists mainly of surgical excision and
reconstruction in symptomatic patients with high success rate of 84\% to
98\%. Re-operation rate has been reported to be 2\% to 13\%
(Crescenze\emph{et al.} 2015; Reeves \emph{et al.} 2014; Nickles
\emph{et al.}2014; Stav \emph{et al.} 2008).

In asymptomatic patients, conservative treatment with prophylactic
antibiotics can be attempted (Greimann \emph{et al.} 2019). Surgical
treatment was successfully performed in the 4 described cases in small
animals (Henry \emph{et al.} 2018; Foster \emph{et al.} 1999; Atilla
2018; Watanabe \emph{et al.} 2015Diverticulectomy has also been
described to be successful in goats and calves (Temi\selectlanguage{english}zsoylu
2005\textbf{;} Bokhari 2013; Sylla \emph{et al.} 2019; Abd-El-Hady 2014;
Maiti \emph{et al.} 2018). Surgical treatment by perineal urethrostomy
and distal urethrostomy over the diverticulum was performed in the
stallion described by Diel de Amorim \emph{et al.} (2014).

To conclude, urethral diverticula are a rare condition in equids and
should be considered in cases of colic with urinary incontinence and
urinary tract infection. We were unable to identify the origin in this
case but a congenital defect was strongly suspected.

\textbf{Author's declaration of interest}

No conflicts of interest to declare.

\textbf{Ethical animal research}

Not applicable.

\textbf{Source of funding}

None.

\textbf{Authorship}

L.M. Hermans, L. Borde-Dore and J.L. Cador\selectlanguage{ngerman}é performed diagnostics, case
evaluation and contributed to the preparation of the manuscript. B.
Drumond performed the diagnostic imaging and contributed to the
preparation of the manuscript.

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\textbf{Figure legends}

Figure 1: Urethral mucosa with view on the entrances of the bladder and
the 2 diverticula. The mucosa is hyperaemic and there is presence of
some swelling.

Figure 2: Normal epithelial lining on the inside of the diverticula. The
mucosa is hyperaemic.

Figure 3: Urethra with view on the entrances of the bladder and the 2
diverticula after treatment with anti-inflammatories. Reduction of
hyperaemia and swelling.

Figure 4: Reduction of mucosal hyperaemia in one of the diverticula,
persistence of urine accumulation with sediment.\selectlanguage{english}
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