Key-words:
Circumcision - complications - meatal stenosis - neonatal
Introduction
Circumcision is the surgical removal of all or part of the human male prepuce. It is estimated that one in three male individuals worldwide is circumcised, with almost universal coverage in some setting and low prevalence in others.[[1]] Circumcision is one of the oldest procedures which was originally used for religious purposes, and religious circumcision is still done by Muslims and Jews worldwide. This surgery is also common in Christian Americans and is performed for cultural, medical, and esthetic reasons among Africans and native Australians.[[2]] The procedure is typically done in the neonatal period for religious reasons but may also be done in older children and adults for the same reasons or to treat several medical conditions including balanoposthitis, phimosis, or paraphimosis.[[3]] Meatal stenosis (MS) is a condition that almost always is acquired and occurs following neonatal circumcision. The risk of MS in noncircumcised boys is 10–26 times lower than circumcised boys.[[4]]
In most cases, MS does not become apparent until after the child becomes toilet trained and rarely leads to obstructive uropathy. Commonly, the child presents with either a forceful, fine stream that has a great casting distance, dorsally deflected stream, or prolonged voiding times. There are two theories regarding the pathogenesis of MS; ischemic and inflammatory. In a boy with suspected MS, the meatus should be calibrated with a Bougie a boule or assessed with infant sounds. If the meatus is diminished in size or if the child has abnormal voiding symptoms, renal and bladder ultrasound examination is indicated. If the child has a history of urinary tract infection (UTI), a voiding cystourethrogram (VCUG) should be done also. However, MS rarely causes obstructive changes in the urinary tract. In a series of 280 children with MS, 5% of the patients had surgically significant lesions on VCUG, and only 1% had upper urinary tract abnormalities.[[5]] This study was designed to study the different clinical pictures, age at presentation, and management and outcome of postcircumcision MS in Libyan children.
Patients and Methods
This retrospective study was conducted at the Department of Urology, Hawari Center for Urology and Otolaryngology, Benghazi, Libya, during the period from January 2010 to December 2018. Eighty-six patients presented with MS were included in this study with their age range from 3 to 13 years. All patients had a detailed history and genitourinary examination. The medical records were reviewed retrospectively regarding age at presentation, different clinical pictures, management, and their outcomes. Relevant investigations such as complete blood count, blood urea, serum creatinine, bleeding profiles, and urine analysis were evaluated.
The diagnosis of MS was confirmed by the inability to insert an age-matched feeding tube into the urethral meatus and the distortion of the meatus from an ellipsoid to a pinpoint shape. No investigations such as ultrasonography or micturating cystourethrography were performed during the evaluation.
As a curative procedure and under general anesthesia, all of our patients underwent a standard meatotomy as day case procedure where meatotomy done with application of hemostat at the stenosed segment up to a few millimeters down from the meatus for 1–2 min followed by ventral midline incision along the crushed segment. Hemostatic sutures were applied to anchor the urethral mucosa at the meatal lips. Antibiotic dressing was done and postoperative analgesia was prescribed.
Statistical analysis
Statistical Package for Social Sciences (SPSS) version 15, (IBM Corp., New York, U.S.A.) was used for data presentation and analysis and the results are expressed as frequencies and percentages.
Results
The mean age of the patients was 5.14 years (range 3–14 years). The majority of patients (73 out of 86) (84.88%) were between 3 and 8 years of age [[Figure 1]].
Figure 1: Age groups of children with symptoms of meatal stenosis
The most common symptoms among our patients was pain (burning) during micturition which accounts for 53.4%; followed by thin casting stream (19.76%); dorsally deviated stream (15%); sudden urge to micturition (5.8%); frequency of micturition (3.38%); and prolonged time of micturition (2.32%) [[Figure 2]].
Figure 2: Frequency of symptoms of meatal stenosis
After the curative procedures, the patients were followed for a period up to 17 months. No recurrence of any symptoms was seen during the follow-up period.
Discussion
Circumcision is one of the most common neonatal surgical procedures performed throughout the world, and most of them are performed for religious beliefs, not for medical reasons.[[6]],[[7]] MS is one of the late complications of circumcision that occurs in 5%–20% of circumcised cases.[[8]] However, higher rate may be seen in areas in which the procedure is done by nonqualified regional peoples.[[9]],[[10]] The incidence is significantly higher in neonatal circumcision compared to higher age groups.[[11]] The frequency may be as high as 20% after circumcision if the condition is defined as a meatal diameter of <5 Fr.[[9]],[[12]] It accounts for 26% of late complications.[[10]],[[13]] The wide variations in frequencies of adverse effect following circumcision are likely due to several factors such as age, training, and expertise of the provider.[[14]],[[15]] The pathophysiology of postcircumcision MS is debatable. Two causes are discussed: meatitis and meatal ischemia. Meatitis (nonspecific inflammation of the meatus) is the most common explanation reported in the literature; physiologically, there is eversion of the distal urethral mucosa through the urethral meatus (lips of the meatus) and excisions of the foreskin expose; this everted mucosa to constant forces of friction and trauma from children's underwear.[[16]],[[17]] Another explanation could be ischemia of the meatal mucosa secondary to damage of frenular artery as the frenular artery is possibly the vascular supply to the urethral meatus and perhaps to a larger area of the distal male urethra.[[16]],[[17]]
MS is divided into mild, moderate to severe, and severe. Mild types of MS do not require any treatment, while in moderate-to-severe types, treatment can be done using topical ointment or urethral dilatation. In severe types, surgery such as meatotomy should be performed as a curative procedure.[[2]],[[16]] A cohort study performed by Litvak et al. demonstrated that a boy younger than 10 years of age should have a meatus of at least 8 Fr, while boys 11–12 years of age should accommodate at least 10 Fr.[[18]]
Diagnosis of MS is usually based on a change in the elliptical shape of the meatal opening to a circular shape because of fibrosis or scarring, with visually apparent narrowing. There are, however, differences in meatal widths in different males, so a narrow meatus does not necessarily indicate functionally significant stenosis.[[19]]
Our study was a retrospective descriptive study where 86 boys of more than 3 years of age were included in the study. The most common presenting symptoms among our patients were pain (burning) during micturition and thin casting stream, which is consistence with the study done by Mahmoudi on 120 children with MS, where the most prevalent symptoms were dysuria and narrowing of the urinary stream or urinary deviation.[[20]] It is similar to results of study by Cartwright et al. where dysuria was the commonest presenting symptom.[[21]] In the study by Persad et al. done on 12 cases of MS following circumcision, the main symptoms were penile pain at the initiation of micturition (12 of 12) and narrow, high-speed stream (8 of 12).[[17]] In the study by Ghods et al., the most common symptoms among symptomatic patient group were discomfort on voiding or dysuria and decreased urinary caliber.[[13]] Upadhyay et al. reported their 12 years' experience with 50 cases and the most common presenting complaint among 34 (68%) symptomatic patients was decreased urinary caliber.[[22]]
Similar to our results, in the study performed by Joudi et al., 25 out 27 (92.6%) of symptomatic cases were presented with decreased urinary width and prolonged voiding times;[[12]] however, prolonged voiding times in our cases represents only 2.3% of all patients.
The differentiation between meatal narrowing and the urethral MS is really the crux of the problem. Stenosis implies disease while narrowing may be only an anatomic variant and visual inspection does not correlate with actual calibration. The clinical status of the patient and not the appearance of the meatus was the deciding factor.[[23]]
MS is one of the late complications of circumcision that is mainly seen 1–2 years later.[[6]] Regarding the age of presentation, it is closely related to the age of circumcision. MS is often missed because boys do not get long-term follow-up care after circumcision. Its symptoms often are mistaken for UTIs and may be treated empirically by antibiotics.[[24]] The symptoms of MS are usually ignored for many months until parents detect the child's voiding habits.[[17]] The mean age at presentation is related to the time of circumcision as we performed our evaluation from the toilet training till puberty (range 3–13 years) with a mean age of 5.14 years. In the study by Upadhyay et al., the mean age at presentation of symptomatic children was 4 years (range 3 months to 13 years);[[22]] whereas the mean age of the patients was 2.5 years (range 3 months to 6 years) in the study by Mahmoudi.[[20]]
Limitations
The limitations of this retrospective study are the small number of patients enrolled. Moreover, there was no control group. Furthermore, regarding obstructive uropathy secondary to MS, as it is of rare occurrence as notice by large studies, we reserve the use of auxiliary investigations such as ultrasound scan and retrograde urethrogram for persistence of symptoms during follow-up period after meatotomy.
Recommendations
Our results highlight the importance of long-term follow-up including genital examination, to detect possible meatal MS as its presentation can be very late.
Conclusion
Although MS is a common long-term complications following circumcision during infancy or nappy age, it continues to be unrecognized condition until toilet training and presentation can be very late. MS needs long-term follow-up, and it should be excluded by careful meatal examination in any child who is circumcised particularly at the neonatal or nappy period and presented with abnormal micturition or even lower abdominal pain.
