Asymptomatic hypospadias with tight meatal stricture in the father of twelve children.

Hypospadias is a highly prevalent congenital anomaly. English articles, indexed in PubMed, published the long-term sexual and reproductive outcome following hypospadias repair. Almost all repairable cases of hypospadias are operated in childhood. Although distal hypospadias does not interfere with fertility, it is worthy reporting a case of an elderly man, who fathers 12 children and has no urologic complaint, presented with unrepaired coronal hypospadias and severely tight external urethral meatus.

CASE REPORT

A 60-year-old man was presented to an orthopedician in the operating theatre as a case of difficult urinary catheterization. The patient was going for a lengthy orthopedic operation that required a drained urinary bladder beforehand. The urologist was called and he found the patient anesthetized, having circumcised penis and a pin-hole external urethral meatus (EUM) at the corona penis level. The tight meatus was negotiated first with a lubricated 4-french (Fr) bougie urethral dilator followed by subsequent sized dilators up to 14- Fr caliber when a proper Foley catheter was introduced easily into the bladder. Post-operatively, the catheter was removed. The patient was carefully interviewed to reveal that circumcision was performed neontally as a religious rite, and he has no history of urologic diseases or trouble voiding, as this was the first time for him to meet a urologist. The patient, who is unaware of his condition, is a father of 12 children and never underwent urinary catheterization.

DISCUSSION

Hypospadias is a highly prevalent congenital anomaly of the male genitalia. In comprehensive review, Sorensen (1953) credited Rennes in 1831 with reporting a prevalence of hypospadias of 1 in 300 “recruits” and also reported the same figure for live male births in Denmark. There is a plethora of hypospadias cases of different degrees of severity repaired in childhood that resulted in different psychological, social, and sexual outcomes. Psychosexual effects of hypospadias repair are endured in adulthood although affected men maintain satisfaction in their sexual life [1]. This patient represents an unusual presentation of a common congenital anomaly. It is unusual in the sense that the elderly man should have had his hypospadias deformity repaired decades ago in order to pass urine freely and to impregnate his spouse with a wider, and properly located meatus.

EUM stenosis is part of the hypospadias anomaly complex. It results in obstructive urinary symptoms in many instances. The tight meatus in this case was severe, hardly admitting a lubricated 4-Fr bougie dilator. Litvak et al., reported that the meatus in children younger than one year accepts a lubricated 5-Fr feeding tube. They also reported that, in children aging 1-6 years, an 8-Fr feeding tube could pass without difficulty [2].

The social and sexual life of adults operated for hypospadias during childhood has been studied by a few authors. Aho et al., compared those who underwent hypospadias repair and circumcision. There was no significant difference in sexual and social life [3]. A few publications reported patient and partner dissatisfaction with the appearance of genitalia, as sexual dissatisfaction is often attributed to penile size and curvature. Genital perception and libido are mostly unaffected, especially in those with distal defects. Bubanj et al. noted that self-reported strength of libido was slightly better for controls compared to patients with hypospadias, but without a statistically significant difference. Moriya et al. noted that only about 10% of both patients and controls reported that their libido was low [4]. Ejaculatory disturbances range between 6% and 37% of operated individuals. Problems reported include weak or dribbling ejaculation, having to milk out ejaculate after orgasm, quantity of semen passing after intercourse, anejaculation with or without orgasm [5]. Liu et al. observed that the rates of ejaculation problems in the distal and proximal groups were 19.5% (8/41) and 48.6% (17/30), respectively [5, 6]. The erectile problems in repaired hypospadias may be attributed to surgically correctable and non-correctable causes. More commonly encountered correctable causes include persistent chordee, penile torsion, fistula formation, acquired meatal stenosis, and inadequate cosmetic outcome. The commonest surgically uncorrectable cause is the size of the penis. Achieving a straight penis is one of the objectives of hypospadias correction. There is no convincing evidence for impaired fertility in repaired cases [5]. Similarly, in unrepaired cases, as the EUM is distal enough to deposit the seminal fluid high in the posterior vagina with no difficulty, fertility is supposed to be preserved. Furthermore, Aho et al. found that men who had hypospadias during childhood were less likely to live with a partner, and that they had fewer children (0.8 vs. 1.1). The difference was not statistically significant [3]. Figure 1

Figure 1

Extremely tight external meatus in a fairly asymptomatic 60 yrs, old man with distal hypospadias.

The literature is scant about the urinary, social, sexual, and fertility aspects of unrepaired hypospadias cases. To our knowledge, no other case has been reported for a circumcised uncorrected hypospadias with extremely tight EUM in a “well”, fertile, elderly man who never experienced voiding troubles.

CONCLUSIONS

The finding in this treatise supports the previously published data surrounding the unaffected sexual and fertility aspects of patients having distal hypospadias, and limits repair to cosmetic reasons. Sufferers, although psychologically affected, maintain satisfaction with their sexual life.