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Abstract
Dear Sir,
Surgical interventions for vesicoureteral reflux (VUR) include open or laparoscopic
ureterocystoneostomy (UCN) and endoscopic injection. Historically, open UCN has been
the gold standard in surgical treatment of VUR with high success and low complication
rates. [1] Although open UCN causes bleeding from the incised bladder wall or dissected
trigone, the bleeding is usually not severe unless a bleeding disorder is present.
We report a case of bilateral VUR that developed severe persistent hematuria and prevesical
hematoma after open UCN.
A 7-year-old boy was referred for management of febrile urinary tract infection (UTI).
He had a history of recurrent UTIs during infancy. Voiding cystourethrography showed
bilateral grade 4 VUR. Dimercaptosuccinic acid (DMSA) renal scintigraphy revealed
bilateral renal scarring. Subsequently, bilateral UCN by the Cohen technique was performed.
The postoperative course was uneventful till postoperative day (POD) 5. He developed
fever and severe gross hematuria after removal of the urethral catheter. Ultrasonography
showed a hematoma in the prevesical space. Even after the urethral catheter was reinserted,
severe hematuria persisted for 4 days and the prevesical hematoma further increased
in size. We performed further coagulation testing because preoperative coagulation
testing showed slightly prolonged activated partial thromboplastin time (APTT) of
45 seconds (normal range, 30–40 seconds). The factor VIII activity was 22% (normal
range, 62–145%), while von Willebrand activity/antigen were normal. The result led
to a diagnosis of mild hemophilia A. On further interview, his parents recalled that
he had developed persistent bleeding after tooth extraction. Additionally, when his
little brother (3 years old) underwent inguinal herniorrhaphy, preoperative coagulation
testing also showed a slightly prolonged APTT but he did not develop any would bleeding
or hematoma. The little brother was also diagnosed with mild hemophilia A afterward.
Gross hematuria subsided spontaneously, and the urethral catheter was removed on POD
20. After removal of the urethral catheter, gross hematuria did not recur and the
prevesical hematoma did not increase in size. He was discharged on POD 25. Ultrasonography
performed at 3-month after discharge showed complete resolution of prevesical hematoma.
An abnormal APTT or history of abnormal bleeding may lead to preoperative diagnosis
of congenital bleeding disorders. On the other hand, a normal APTT with no family
or personal history cannot always exclude the potential of congenital bleeding disorders
including hemophilia. According to Teruya’s report, an APTT was normal even when factor
VIII activity was 30%. When APTT was prolonged to more than the normal upper limit,
factor VIII activity was 13%. He concluded that a normal APTT did not guarantee adequate
coagulation factor levels. [2] Hemophilia A is graded into varying levels of severity
depending on factor VIII activity (severe, less than 1%; moderate, 1–5%; mild, 5–40%).
The factor VIII activity of our patient was 22%. It led to the diagnosis of “mild”
hemophilia A. Patients with mild hemophilia may not bleed excessively until they experience
trauma or surgery. [3] A more meticulous inquiry regarding bleeding tendency¬ is suggested
in case of even slight abnormality of clotting profile or history of prolonged bleeding
to preempt postoperative bleeding complications.
Footnotes
Source of Support: Nil
Conflict of Interest: None declared
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