Encrusted Hairball in the Urethra: An Uncommon Complication of Hypospadias Repair

ABSTRACT

We present an uncommon case of an encrusted urethral hairball in an adult patient who had hypospadias repair during childhood. The management of such a case is discussed briefly and suggestions are made to overcome this issue.


Eng Hong Goh, Omar Syed, Boon Wei Teoh, Kah Ann Git

Submitted December 21, 2011 - Accepted for Publication January 23, 2012 


 KEYWORDS: Urethra, stone, hair

CORRESPONDENCE: Eng Hong Goh, Urology Unit, Department of Surgery, Universiti Kebangsaan Malaysia Medical Centre, Jalan Yaacob Latif, Bandar Tun Razak, Kuala Lumpur, 56000, Malaysia ().

CITATION: UroToday Int J. 2012 June;5(3):art 19. http://dx.doi.org/10.3834/uij.1944-5784.2012.06.06

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INTRODUCTION

In the case of hypospadiac repair, an encrusted hairball is not commonly seen. We present such a case and briefly discuss its management.

CASE REPORT

A 29-year-old man, with infertility and hypospadias, presented to another hospital with a scrotal mass along the midline, and with urethral fistulae he’d had for 10 years. The scrotal mass gradually increased in size but it was painless, and the patient mistook it as a testis. In addition, he was used to having a fistulous urinary stream for many years. He finally came to seek treatment for infertility as he was planning to start a family. The patient had multiple hypospadiac repair surgeries in various other health institutions when he was a child. He had been lost to follow-up after the last repair operation and was unable to recall details. On examination, both his tiny testes were barely palpable in the scrotum. The scrotal mass was situated along the midline, measured 2 x 2 cm, was hard in consistency, mobile, and its margin was well demarcated from the surrounding tissue. Also, there were a few fistula openings seen around the penoscrotal region. A computed tomography (CT) scan identified the mass as a stone.

The patient was referred to our hospital for further management. Surgical exploration revealed the presence of an encrusted hairball at the site of the neourethra constructed from the transpositioned scrotal skin. After removing the stone and trimming the hairs, the urethrotomy was primarily repaired. Postoperatively, the patient recovered uneventfully without any complications. Two months after the operation, he was still being followed up for the fistulae that were treated expectantly as well as the regrowth of hair in the urethra.

DISCUSSION

A urethral stone is an uncommon entity in urology practice, accounting for only 1% of all urinary stones [1]. The formation of encrusted hairballs or stones is also infrequently seen in hypospadiac surgery. Barbagli et al. reported an incidence of 1 in 60 patients with failed hypospadiac surgery had developed stones in the urethra [2]. A search yielded only a minute amount of literature, the chief of which were just case reports. The largest series was a collection of 5 cases by Hayashi et al. [3]. Among these 5 cases, 4 had previously undergone Thiersch-Duplay repair, and the type of repair was not known in another case. At presentation, the stones were numerous but collectively they measured about 2 cm in diameter and were located in the neourethra. How could a diagnosis be made? Given the suggestive history of hypospadiac repair and the easily palpable superficial location of the stone, we felt a clinical diagnosis could be confidently made. Otherwise, simple imaging studies such as a plain radiography or ultrasound should be sufficiently conclusive. The use of a CT scan as in our case was unnecessary unless the mass was deep within the scrotum or if diagnosis was questionable in the simpler imaging studies aforementioned.

Various treatments had been suggested to overcome the issue of an encrusted hairball in these hypospadiac patients. Giordano et al. proposed the use of extracorporeal shock wave lithotripsy to blast the urethral stone, but it was necessary to consider the potential mechanical damage delivered to the testes due to the proximity of these organs [4]. Lasers had been utilized to remove hairs as well as the urethral stone on the neourethra [5]. Singh and Hemal advocated the chemical epilation of hair by using thioglycolate at a thrice-monthly interval [6]. A simple measure described by Hayashi et al. was the use of self-catheterization of the urethra and this seemed promising since there had been no recurrence among patients until a 7-year follow-up after reparative surgery [3]. Even if one decided on a surgical treatment, it was also vital to consider the access of choice. In the era of endoscopy, the open method was still a chosen route for many [3,5-7]. The reasons likely being the difficulty in identifying clearly the border of the stone from the neourethra as well as the density of the hair impairing vision during endoscopic surgery, compounded by bleeding that potentially damaged the urethra [3]. Additionally, a stone of substantial size certainly would take a much longer operating time if endoscopic method is employed. In fact, 2 out of 3 cases from Hiyashi et al. that underwent endoscopic lithotripsy developed recurrence [3]. The additional advantage of open surgery was clearly the opportunity to perform diverticulectomy, if any.

What could be the ideal in preventing the formation of hair and urethral stones in hypospadiac repair? Hiyashi et al. suggested an epilation of hair follicles at the donor skin site before urethroplasty [3]. Although the use of apparently non-hair-bearing donor skin for the neourethra seemed to be the solution, unfortunately it had proved to cause a hairy urethra as well [8]. It has been reported that a hairy urethra has a complication rate of 5% [9]. Other types of urethral mucosal substitutions such as buccal mucosa and tunica vaginalis also had their own share of limitations, albeit with different success rates. In a review of 60 patients, with a mean follow-up period of 33.8 months, Barbagli et al. concluded that buccal mucosa was superior to skin as a urethral substitution. The success rate of buccal mucosa was 82% regardless of the number of stages needed to repair the hypospadias. However, the success rate of skin was 82% in 1-stage repair but it dropped to 50% in multistage repair [2]. Saphenous vein grafts had been reportedly used as a neourethra. In their case report of an 8-year-old boy with hypospadias, Shaeer and El-Sadat claimed the superiority of the vein graft attributed to pre-tubularization, thus eliminating the longitudinal suture line and the risk of fistula, robustness as a graft due to the thick and multilayer nature of the vein, and the absence of hair. But the follow-up period of the patient was only 12 months and it was believed that a longer observation period was desirable [8].

Disregard of the type of tissue used, the alien nature of transplanted or transpositioned tissue, and the lack of vascularized spongiosum may all result in a neourethra that may not grow naturally or in tandem with the surrounding tissue [2]. It had been suggested that hypospadiac patients should be followed up until sexual maturation is attained [2]. Despite this, the complications of reparative surgery such as hairball formation could still occur later. Although most of the reported cases, including ours, occurred in the second or third decade of life, it had been reported in a patient in his sixth decade of life [3-7]. Therefore, we additionally felt that proper patient education is vital, and periodical self-massage of the neourethra may prevent or detect early formation of a stone. Early medical attention in this regard was pivotal since it served to prevent the breakdown of a successful yet painstakingly reconstructed urethral passage.

CONCLUSION

Although hairball formation after hypospadias repair using hair-bearing skin as a neourethra was an uncommon entity, it was still reasonable to avoid using hair-bearing skin as urethral substitution given the potential damage. However, in situations where the use of skin is inevitable, such as a surgeon’s familiarity with the technique or a lack of alternative substitution, a urethral hairball should be actively sought even many years after repair. In this regard, educating the patient in proper care would be beneficial in addition to long-term clinical follow-up.

FIGURES

Figure 1

Figure 2

REFERENCES

  1. Shyam, L., G.P. Shrivastava, V. Yedalwar, et al. (2010). “A Rare Case of Primary Giant Urethral Calculus.” J Surg Pakistan (Intl) 15(3):157-158.
  2. Barbagli, G., M. De Angelis, et al. (2006). “Failed hypospadias repair presenting in adults.” Eur Urol 49(5): 887-894; discussion 895. 
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  3. Hayashi, Y., T. Yasui, et al. (2007). “Management of urethral calculi associated with hairballs after urethroplasty for severe hypospadias.” Int J Urol 14(2): 161-163. 
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  4. Giordano, D. R., M. Carnevale, et al. (1988). “Ambulatory treatment by extracorporeal shock wave lithotripsy for an urethral stone in a hypospadic boy.” Child Nephrol Urol 9(4): 239-240. 
    PubMed
  5. Kaneko, T., H. Nishimatsu, et al. (2008). “[Laser hair removal for urethral hair after hypospadias repair].” Nihon Hinyokika Gakkai Zasshi 99(1): 35-38. 
    PubMed
  6. Singh, I. and A. K. Hemal (2001). “Recurrent urethral hairball and stone in a hypospadiac: management and prevention.” J Endourol 15(6): 645-647. 
    PubMed ; CrossRef
  7. Singh, V., K. M. Singh, et al. (2010). “Tricholithobezoar: an unusual long-term complication of hypospadias surgery.” Urol J 7(1): 15. 
    PubMed
  8. Shaeer, O. K. and A. El-Sadat (2006). “Urethral substitution using vein graft for hypospadias repair.” J Pediatr Urol 2(5): 518-521. 
    PubMed ; CrossRef
  9. Hosseini, J., H. Mokhtarpour, A.R. Bagher Tabrizi, et al. (2011). “Urethroscopic holmium: yag laser ablation of large urethral stone after two-stage urethroplasty.” J Lasers Med Sci 2(2):87-88.
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