1 Unità Operativa di Medicina della Riproduzione and 2 Unità Operativa di Anatomia Patologica, Istituto Clinico Humanitas, Rozzano (Milan), Italy
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Abstract |
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Key words: aspermia/cryopreservation/imperforate anus/TESE/testicular pain
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Introduction |
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This case report describes the management of a rare combination of male infertility due to aspermia and chronic unilateral orchialgia in a patient who had undergone surgical correction of anal atresia during infancy.
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Case report |
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Genital examination revealed testes of normal size (18 ml by Praders orchidometer) and consistency. The right epididymis and vas deferens were normal, but the left epididymis and vas deferens were enlarged and hardened. No pain was evoked by palpation, and there were no varicocele or hydrocele. The penis and urethral meatus were normal. A rectal examination was not practicable because of an anal sub-stenosis.
Laboratory analysis
Analysis of a small drop of semen retrieved on a slide after masturbation showed azoospermia, low pH (6.8, suggesting a prostatic origin), and leukospermia (>20 neutrophilic leukocytes per microscopic field 400x). Post-ejaculatory urinalysis was negative for germ cells, and a post-ejaculatory urine culture was also negative. FSH, testosterone and karyotype were normal.
Ultrasound (US) examination
Scrotal US revealed tubular ectasia of the left rete testis (Figure 1) and an enlarged left epididymis. The testicular parenchyma was normal, without any nodules or increased blood flow at duplex scanning. Right testis, epididymis and vas deferens were normal. Transabdominal ultrasound scan (a transrectal scan was impracticable) showed a small prostate with a small (5 mm diameter) paramedian left cyst. The seminal vesicles were normal (maximum anteriorposterior diameter 9 mm), as were the bladder and kidneys.
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Ten months later, the patient was urgently admitted to hospital because of a dramatic worsening in left testicular pain, which had become continuous and unbearable for 5 months. It was exacerbated by sexual activity to the point that he was obliged to refrain from intercourse, and was so intense that it significantly interfered with his work and everyday activities. He had previously taken antibiotics (cyprofloxacine, tetracycline, aztreonam) and non-steroidal anti-inflammatory drugs without any benefit. A physical examination revealed a painful, enlarged and irregular left epididymis, and the left vas felt like a pencil. There was no change in the transabdominal ultrasound scan findings, but scrotal ultrasound scan showed a worsening of the rete testis ectasia. Because of the pain, the patient consented to an exploratory intervention, with left epididymo-orchiectomy if necessary. He also authorised the extraction and cryopreservation of testicular sperm for subsequent ICSI.
Surgical procedure
The patient underwent exploratory left inguinotomy, with the spermatic cord being bluntly dissected with a finger down to the scrotum and the testis delivered through the wound. The inguinal portion of the vas deferens was also enlarged. The caput epididymis was found to be enlarged, with brown tubules 23 mm in diameter. The anatomical patency of the seminal ducts downstream from the epididymis was therefore investigated by cannulating the proximal vas deferens with a 25 G short butterfly needle and injecting 30 ml of saline solution mixed with 1 ml of 10% methylene blue. The saline solution easily passed into the bladder, thus excluding a distal seminal duct obstruction and the need for vasography (Goldstein, 1995). The injected solution was recovered through an 18 F Foley catheter and centrifuged, but no sperm were found in the Makler chamber, thus suggesting a proximal (epididymal) obstruction (Colpi et al., 1994
). The epididymal fluid was then aspirated and analysed but no sperm were found. Finally, testicular touch preparation cytology revealed the presence of many sperm thus suggesting normal spermatogenesis. The surgical procedure was completed with a radical orchiectomy.
Testicular sperm extraction and cryopreservation
The removed testis was opened in the operating theatre by means of a longitudinal incision. Most of the testicular lobules were removed and collected in four tubes filled with Quinns hTF medium and HEPES (Biocare Europe). Later, in the IVF Laboratory, the lobules were transferred into Petri dishes with Quinns Sperm Washing Medium (BioCare Europe) and dissected into small pieces under a stereomicroscope using two coverslides (24 mmx50 mm). The bioptic sample was collected in a sterile conical tube (Falcon) and centrifuged at 600/800 g for 10 min. The sperm concentration determined using a Makler chamber was 2.5x106/ml, for a total number of 25x106 sperm. Eosin-tested sperm viability was 65%. After dilution with freezing medium (Irvine), 35 0.50 ml sterile straws were loaded and frozen following a rapid two-phase protocol (Wolf and Patton, 1989).
Histological examination
The rete testis and proximal epididymal tubules showed cystic ectasia, with no evidence of sperm inside; the rete testis was also affected by focal dystrophic calcification. Testicular histology revealed chronic aspecific orchitis associated with thickened or oedematous basal membranes. The seminiferous tubules had diameters ranging from normal to hyalinized, and contained a mean of 10 mature spermatids per tubule.
Follow-up
The patient was discharged from the clinic after 24 h. There were no post-operative complications. After 7 days, the patient had sexual intercourse with his partner without feeling any pain. Eight months later the patient is completely free of pain and his plasma testosterone concentration is normal. During the course of the next few months, the couple will be included in an IVF programme.
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Discussion |
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In the case of the impelling problem of chronic left scrotal pain, and as antibiotic and non-steroidal anti-inflammatory drugs had failed to improve the symptoms, we might have performed an epididymectomy. However, this procedure is not indicated in chronic pain due to epididymitis or in seminal duct obstruction (Padmore et al., 1996) and, in a study of 10 patients undergoing epididymectomy, nine required subsequent orchiectomy for the definitive treatment of pain (Davis et al., 1990
). The co-existence of tubular ectasia of the left rete testis must also be stressed because, if the pain had been secondary to distension of the seminal ducts, an epididymectomy would not have resolved the problem inside the testis.
Testicular denervation (Choa and Swami, 1992) has been excluded as an alternative to epididymectomy because it does not work when there is an underlying organic disease (Heidenreich et al., 1997
).
Inguinal orchiectomy was adopted because it guarantees a 73% success rate (Davis et al., 1990) and allows the extraction and cryopreservation of a considerable number of sperm (TESE).
More specific or conservative procedures for the treatment of infertility (but not pain) were rejected before or during the operation. Transurethral resection of the ejaculatory ducts (Pryor and Hendry, 1991), would presumably not have worked because of (i) the anatomical patency of the left distal seminal duct, and (ii) the absence of sperm in the seminal ducts. It is also worth pointing out that the latter ruled out surgical sperm retrieval from the epididymis (MESA) (Silber et al., 1988
), proximal vas deferens (Hovatta and von Smitten, 1993
) and distal seminal duct (Colpi et al., 1992
).
The site of the obstruction therefore remains unclear. Biochemical analysis of specific substances for each level of the seminal ducts (i.e. L-carnitine, -glucosidase, etc.) (Behre et al., 2000
) was not considered because of the absence of spermatic fluid. In theory, the site of the obstruction had to be proximal to the rete testis, and multiple aetiological factors contributed to the clinical picture (i.e. neurological anejaculation plus chronic seminal duct infection, causing the anatomical alterations of the left seminal duct).
To the best of our knowledge, this is the first description of a case of sperm extraction from a whole testicle removed for chronic testicular pain. The relief from pain and the availability of sperm for a large number of ICSI cycles suggest that such an approach should be considered when untreatable orchialgia and aspermia coexist in infertile men who have undergone the surgical correction of imperforate anus.
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Notes |
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References |
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Submitted on June 17, 2002; accepted on August 8, 2002.