Bilateral testicular masses and adrenal insufficiency: is congenital adrenal hyperplasia the only possible diagnosis? First two cases of TARTS described in Addison-only X-linked adrenoleukodystrophy and a brief review of literature.

BACKGROUND: Testicular adrenal rest tumors (TARTs) are benign masses deemed to originate from pluripotent testicular steroidogenic cells that grow under chronic ACTH stimulation. These lesions, occasionally misdiagnosed as Leydig cell tumors (LCTs), are typically described in patients with congenital adrenal hyperplasia (CAH). X-linked adrenoleukodystrophy (X-ALD) is an inherited disorder of beta-oxidation with accumulation of very long chain fatty acids (VLCFAs) in various tissues, and a rare cause of primary adrenal insufficiency (PAI). TARTs have never been associated with X-ALD. CASE 1 DESCRIPTION: A 19-year old male, who had previously undergone bilateral enucleation of presumed LCTs, was referred to our unit. Follow-up scans showed persistent bilateral lesions compatible with TARTs. Biochemical exams revealed PAI but excluded CAH. A serum VLCFAs panel was consistent with X-ALD, with gene testing confirming the diagnosis. Histological revision of the previously resected testicular lesions was compatible with TARTs. Start of glucocorticoid replacement therapy was associated with a reduction of testicular masses. CASE 2 DESCRIPTION: A 26-year old X-ALD male was diagnosed with bilateral testicular lesions compatible with TARTs. These lesions increased after ACTH elevation following switch to modified-release hydrocortisone. Clinical and sonographic findings allowed for a "watchful-waiting" approach, avoiding unnecessary surgery. CONCLUSION: These are the first cases reported of TARTs in patients with X-ALD-associated PAI. Testicular lesions in patients with an early onset of ACTH elevation, regardless of the cause, should always be thoughtfully investigated, as they may reveal themselves as TARTs. We suggest that all patients affected from chronic ACTH elevation of a young age of onset should undergo testicular ultrasound in order to evaluate the presence of these lesions. GRT in these patients might also help preserving fertility.

Schiefer counter: An alternative method for clonogenic assay evaluation.

INTRODUCTION: Clonogenic assay evaluates the potential of cells to undergo division or generate clones following treatment with a chemical or other agent, thereby allowing the evaluation of cytotoxic and/or antiproliferative effects. Clonogenic assay analysis using traditional methods tends to be time-consuming and yield inconsistent results, whereas results from analyses conducted using automated image processing methods may be misleading or subject to misinterpretation. Thus, the aim of this work was to validate and demonstrate the applicability of a recently developed software. METHODS: Repeatability of measurements was evaluated by comparing results from 10 replicate images from a single well. To evaluate the viability of the software, results were compared with those obtained from manual counting, crystal violet optical density, and up-to-date automated methods. A clonogenic index was experimentally developed using the individual area occupied by colonies, while clone stratification was used to differentiate between antiproliferative and cytotoxic effects. RESULTS: The developed software showed to be a reliable and consistent tool for clonogenic assay evaluation, presenting a repeatability mean error of 0.79% for the number of colonies and 0.89% for the total area of colonies, as well as exhibiting a significant correlation (p < 0.05) with results obtained from widely adopted gold standard methods. The software was also able to detect an appropriate dose-dependent effect as well as a predominant cytotoxic effect of vincristine on MCF-7 cells and calculate the clonogenic index. DISCUSSION: Therefore, this software is adequate for the analysis of clonogenic assay images, differentiating between cytotoxic and antiproliferative trends.

Towards quality assessed characterization of nanomaterial: Transfer of validated protocols for size measurement by dynamic light scattering and evaluation of zeta potential by electrophoretic light scattering.

Quality control analysis of nanomaterials has been identified as a major issue to pursue their development in different industrial fields including nanomedicine. One difficulty is the lack of standardized and validated protocols suitable to achieve their characterization. In a previous work, we have developed standardized protocols for the evaluation of the size and zeta potential of nanomaterials based on methods described in the ISO standard and have performed validation of each one. The present work was aimed to transfer these protocols in three independent receiving laboratories. No official guideline was described in the literature to achieve such a transfer. A comparative study for receiving laboratories equipped with the same instrument as the sending laboratory was designed based on the Code of Federal Regulation edited by the Food and Drug Administration. For the receiving laboratory equipped with an instrument working at a different wavelength, a new validation was designed and applied. Corresponding statistical methods were used for the analysis of the results. A successful transfer of the protocols in all receiving laboratories was achieved. All laboratories recorded consistent results applying in blind the protocol of size measurements on two samples of nanomaterials from which included one reference.

The prokineticin Bv8 sensitizes cutaneous terminals of female mice to heat.

BACKGROUND: Injection of the noxious peptide Bv8 has previously been shown to induce a biphasic thermal hyperalgesia in rodents, the first peak presumably due to peripheral sensitization. This hypothesis has never been directly confirmed. We have assessed whether Bv8 can indeed sensitize peripheral nerve fibres in the mouse to heat. METHODS: We used recordings from single cutaneous fibres, cutaneous calcitonin gene-related peptide (CGRP) release and immunostaining in nerves and plantar skin to evaluate the Bv8 effects on cutaneous nerves. RESULTS: Application of Bv8 at nanomolar concentrations (30-310 nmol/L) to skin preparations significantly increased the heat-induced discharge, the heat-induced afterdischarge and reduced threshold temperature of single unmyelinated polymodal fibres. Furthermore, application of Bv8 to hind-paw skin flaps or trigeminal ganglia significantly elevated their heat-induced CGRP release. Capsaicin-induced and to a lesser extent also KCl-induced CGRP releases were also augmented after Bv8 treatment. Immunohistochemistry revealed co-localization of prokineticin 2 (Bv8 ortholog in rodents) and CGRP in both plantar skin and nerve tissues. These results confirm that Bv8 sensitizes cutaneous nerve endings to heat, partly, although not exclusively through TRPV1 activation. CONCLUSION: Our results thus support the hypothesis that the first hyperalgesic phase to follow Bv8 injection to hind paws of intact animals is due to peripheral sensitization of nociceptors. WHAT DOES THIS STUDY ADD?: Our data provide mechanistic insights into the effect Bv8 application exerts on afferent nerve endings and into the concomitant development of thermal hyperalgesia.

PC1, a non-peptide PKR1-preferring antagonist, reduces pain behavior and spinal neuronal sensitization in neuropathic mice.

Peripheral neuropathy is characterized by abnormal pain responses triggered by the release of several mediators and neuronal hyperexcitability at the spinal cord level. Emerging evidence indicates that the enhanced activity of dorsal horn neurons requires communication with glia and microglia, cells that are physiologically involved in neuronal wellbeing. Prokineticins (PKs), which include PK1 and PK2, represent a novel family of chemokines characterized by a unique structural motif comprising five disulfide bonds. They are expressed in the peripheral and central nervous system. PKs bind two G protein coupled receptors, PKR1 and PKR2, and participate in the regulation of several biological processes, including pain sensation. This study aimed to investigate the anti-nociceptive effect of PC1, a non-peptide PKR1-preferring antagonist, in a mouse model of neuropathic pain. To do this, we assessed the activity of spinal cord nociceptive neurons as well as astrocyte and microglia phenotypes after repeated administration of PC1 in vivo. PC1 treatment strongly delayed the development of thermal hyperalgesia and tactile and mechanical allodynia. It also reduced spinal microglial and glial activation 8 days post injury in spared nerve injury (SNI) mice. Neuropathic mice showed an increased level of PK2 protein in the spinal cord, mostly in astrocytes. PC1 treatment completely reversed the increased responsiveness to mechanical stimuli, the decreased threshold of neuronal activation, and the increased spontaneous activity that were observed in nociceptive specific (NS) neurons of SNI mice.

Controlling the activation of the Bv8/prokineticin system reduces neuroinflammation and abolishes thermal and tactile hyperalgesia in neuropathic animals.

BACKGROUND AND PURPOSE: Chemokines are involved in neuroinflammation and contribute to chronic pain processing. The new chemokine prokineticin 2 (PROK2) and its receptors (PKR1 and PKR2 ) have a role in inflammatory pain and immunomodulation. In the present study, we investigated the involvement of PROK2 and its receptors in neuropathic pain. EXPERIMENTAL APPROACH: Effects of single, intrathecal, perineural and s.c. injections of the PKR antagonist PC1, or of 1 week s.c. treatment, on thermal hyperalgesia and tactile allodynia was evaluated in mice with chronic constriction of the sciatic nerve (CCI). Expression and localization of PROK2 and of its receptors at peripheral and central level was evaluated 10 days after CCI, following treatment for 1 week with saline or PC1. IL-1ß and IL-10 levels, along with glia activation, were evaluated. KEY RESULTS: Subcutaneous, intrathecal and perineural PC1 acutely abolished the CCI-induced hyperalgesia and allodynia. At 10 days after CCI, PROK2 and its receptor PKR2 were up-regulated in nociceptors, in Schwann cells and in activated astrocytes of the spinal cord. Therapeutic treatment with PC1 (s.c., 1 week) alleviated established thermal hyperalgesia and allodynia, reduced the injury-induced overexpression of PROK2, significantly blunted nerve injury-induced microgliosis and astrocyte activation in the spinal cord and restored the physiological levels of proinflammatory and anti-inflammatory cytokines in periphery and in spinal cord. CONCLUSION AND IMPLICATIONS: The prokineticin system contributes to pain modulation via neuron-glia interaction. Sustained inhibition of the prokineticin system, at peripheral or central levels, blocked both pain symptoms and some events underlying disease progression.

Peripheral administration of prokineticin 2 potently reduces food intake and body weight in mice via the brainstem.

BACKGROUND AND PURPOSE: Prokineticin 2 (PK2) has recently been shown to acutely reduce food intake in rodents. We aimed to determine the CNS sites and receptors that mediate the anorectic effects of peripherally administered PK2 and its chronic effects on glucose and energy homeostasis. EXPERIMENTAL APPROACH: We investigated neuronal activation following i.p. administration of PK2 using c-Fos-like immunoreactivity (CFL-IR). The anorectic effect of PK2 was examined in mice with targeted deletion of either prokineticin receptor 1 (PKR1) or prokineticin receptor 2 (PKR2), and in wild-type mice following administration of the PKR1 antagonist, PC1. The effect of IP PK2 administration on glucose homeostasis was investigated. Finally, the effect of long-term administration of PK2 on glucose and energy homeostasis in diet-induced obese (DIO) mice was determined. KEY RESULTS: I.p. PK2 administration significantly increased CFL-IR in the dorsal motor vagal nucleus of the brainstem. The anorectic effect of PK2 was maintained in mice lacking the PKR2 but abolished in mice lacking PKR1 and in wild-type mice pre-treated with PC1. DIO mice treated chronically with PK2 had no changes in glucose levels but significantly reduced food intake and body weight compared to controls. CONCLUSIONS AND IMPLICATIONS: Together, our data suggest that the anorectic effects of peripherally administered PK2 are mediated via the brainstem and this effect requires PKR1 but not PKR2 signalling. Chronic administration of PK2 reduces food intake and body weight in a mouse model of human obesity, suggesting that PKR1-selective agonists have potential to be novel therapeutics for the treatment of obesity.

Pharmacological activity of a Bv8 analogue modified in position 24.

BACKGROUND AND PURPOSE: The amphibian peptide Bv8 induces potent nociceptive sensitization in rodents. Its mammalian homologue, prokineticin 2 (PROK2), is strongly up-regulated in inflamed tissues and is a major determinant in triggering inflammatory pain. Bv8 and PROK2 activate two closely related GPCRs, PK(1) and PK(2) , in a relatively non-selective fashion. To characterize better the roles of the two receptors in hyperalgesia and to obtain ligands whose binding affinity and efficacy differed for the two receptors, we modified the Bv8 molecule in regions essential for receptor recognition and activation. EXPERIMENTAL APPROACH: We modified the Bv8 molecule by substituting Trp in position 24 with Ala (A-24) and compared it with Bv8 for binding and activating PK(1) and PK(2) receptors in cell preparations and in affecting nociceptive thresholds in rodents. KEY RESULTS: A-24 preferentially bound to PK(2) receptors and activated them with a lower potency (5-fold) than Bv8. When systemically injected, A-24 induced Bv8-like hyperalgesia in rats and in mice, at doses 100 times higher than Bv8. Locally and systemically injected at inactive doses, A-24 antagonized Bv8-induced hyperalgesia. In rat and mouse models of inflammatory and post-surgical pain, A-24 showed potent and long-lasting anti-hyperalgesic activity. Unlike Bv8, A-24 increased ß-endorphin levels in mouse brain. CONCLUSIONS AND IMPLICATIONS: A-24 induced its anti-hyperalgesic effect in rodents by directly blocking nociceptor PK(1) receptors and by activating the central opioid system and the descending pain control pathway through brain PK(2) receptors.

The use of a preformed spacer in two-stage revision of infected hip arthroplasties.

Two-stage revision with the use of an antibiotic-loaded cement spacer has spread widely as a successful treatment for THA infection. Between 1999 and 2008, 28 patients with infected THA were treated with two-stage implant revision using a preformed spacer. The spacer was left in situ for 5.5 months (range 1-13 months), and the patients were allowed to walk with partial weight bearing. At a mean follow-up of 53 months (range 18-106 months), recurrence of infection was observed in only one patient. Complications were observed in five patients: three spacer dislocations, one distal femoral fracture occurred during stem removal, and one femoral artery pseudo-aneurysm. The mean HHS increased from 43 points (range 13-77) to 82 points (range 35-96). Though small prospective studies are reported in literature, good eradication rate and good functional outcomes encourage for the use of an antibiotic-loaded cement spacer. The industrial production ensures procedure standardization, well-defined physical and chemical properties to the device and eliminates time necessary to intraoperatory manufacturing.

Mesenteric cystic lymphangioma treated with laparoscopic excision: case report and review of the literature.

Mesenteric cystic lymphangioma is a rare lesion. We described a case of 20 years old patient with an asymptomatic cystic lymphangioma that was detected after ultrasound scan performed for a minor abdominal trauma. The patient was treated with laparoscopic exploration and an intestinal resection by mini-laparotomy. Although very rare, cystic mesenteric lymphangiomas may cause complications; therefore, they should be always treated with surgical excision and should be included in the differential diagnosis of acute abdomen.

Cancer risk in male factor-infertility.

Severe forms of male-factor infertility are associated with an increased risk of testicular cancer and scrotal ultrasonography is widely used for diagnosis. In this study, 2172 male members of infertile couples referred to our Reproductive Medicine Unit were submitted to scrotal ultrasonography and 835 selected patients had been followed during a 2-year period. Eight out of nine neoplastic nodules found at the initial examination were unpalpable and discovered by ultrasonography. Ten tumoral lesions were found in 370 testicular biopsies performed for diagnostic purposes or to extract spermatozoa; and eight additional neoplastic lesions were discovered during the 2-year follow-up of 835 patients. The cumulative rate of neoplastic disease was 3.2%. Thirteen cases (1.5%) were malignant (12 germ cell tumours and one non-Hodgkin lymphoma of testicular origin); the remaining 14 were benign forms (Leydig cell tumours and hyperplasias, Sertoli cell nodules, adenomatoid tumours). Testicular volume (cut-off: 12ml) resulted weakly correlated with germ cell cancer (p=n.s., odds ratio 2.01) while low total sperm count (<40x10(6)) (p=0.002, odds ratio 8.4), previous cryptorchidism (p=0.04, odds ratio 7.5) and hypergonadotrophic hypogonadism (p=0.04, odds ratio 7.9) were associated with an increased risk. But a stronger correlation with germ cell cancer was found in the patients with some utrasonographic anomalies, i.e. testicular microlithiasis (p=0.0015, odds ratio 37.1) or larger calcifications not fitting the description of testicular microlithiasis (p<0.0001, odds ratio 69.5). Our findings indicate that scrotum ultrasonography should always be advised in subfertile men with <40x10(6) spermatozoa/ejaculate or hypergonadotrophic hypogonadism or previous cryptorchidism, and that particular care should be taken in the presence of testicular microlithiasis or testicular calcifications. These men should be aware of the existence of higher risk of testicular cancer and trained in testicular self-examination.

Unicentric Castleman's disease in peripancreatic tissue: case report and review of the literature.

Castleman's disease is a rare disorder of the lymphoid tissue that usually occurs in the mediastinum even if extrathoracic involvement, including neck, axilla, mesentery and retroperitoneum, has also been described. We report a case of a 69 years old men with peripancreatic localisation, mimicking a pancreatic neoplasm treated with local excision. Only seven cases of pancreatic and peripancreatic localisation are described in the world-wide literature. This particular site of disease may give troublesome differential diagnosis due to non specific clinical signs and radiological features. Often surgical excision is both diagnostic and therapeutic.

Combined resection and multi-agent adjuvant chemotherapy for intra-abdominal desmoplastic small round cell tumour: case report and review of the literature.

Desmoplastic small round cell tumor (DSRCT) is a rare, highly aggressive malignancy with distinctive histological and immunohistochemical features occurring in young population with male predominance. We report a case of DRSCT occurred in a 17 years old patient which presented with a large upper left quadrant abdominal mass that was treated with a very aggressive surgical approach and multi-agent chemotherapy. At a 12 months follow-up he is free of recurrence. This kind of tumour has a very poor prognosis. No standard treatment protocol has been established. Aggressive surgery combined with postoperative multi-agent adjuvant chemotherapy is justified not only to relieve symptoms but also to try to improve the outcome.

Clinical and molecular cytogenetic studies in three infertile patients with mosaic rearranged Y chromosomes.

Isodicentrics (idic) are structural anomalies of the Y chromosome associated with a 45,X cell line and a broad spectrum of phenotypes. We characterized the rearranged Y chromosomes from three azoospermic males by fluorescence in-situ hybridization (FISH) and PCR. Chromosome study was performed on lymphocytes and testicular biopsy. FISH analysis and PCR established the degree of mosaicism and analysed specific Y regions. Two patients showed a 45,X/46,X,?idic(Y) karyotype with varying degrees of mosaicism. FISH demonstrated the presence of two centromeres and two SRY regions. In the lymphocytes of the third patient, the presence of a small Y-derived marker was also observed. An additional cell line with two idic(Y) was present in the testicular biopsy of the same patient. PCR showed the breakpoint between SY182 (KALY) and SY121 in Yq11.221-q11.222 region in all the cases. For the evaluation of the mosaicism, different tissues must be investigated. The phenotypical sex depends more on the number of copies of the SRY gene rather than on the percentage of 45,X cells, at least in the gonads. The combined use of classical and molecular cytogenetics is necessary for delineating the chromosome regions involved allowing a better genotype-phenotype correlation.

Two-stage revision surgery to treat an infected hip implant. A comparison between a custom-made spacer and a pre-formed one.

A retrospective study was conducted to evaluate the results of two-stage surgical treatment for infected hip implant using two different spacers in antibiotic-impregnated cement, one custom-made and one pre-formed. Out of a total of 20 patients treated between 1995 and 2003, the temporary implant of one custom-made spacer was carried out in 8 cases, while in the remaining 12 cases we resorted to using a pre-formed spacer (Spacer G). In both groups we observed one recurrence of infection, for an overall 90% success rate in terms of eradication of the infection. Custom-made spacers had a greater incidence of local complications (1 breakage, 1 dislocation, 1 sinking of the revision stem) as compared to pre-formed ones (1 dislocation). The clinical results evaluated using the Harris Hip Score showed a statistically significant difference (p < 0.05) in both groups as compared to the preoperative score. The use of a pre-formed spacer, although more costly as compared to that custom-made one, was advantageous in some ways, for example in terms of standardization of the implant method, the lower incidence of mechanical complications and the better functional results.

Biliopancreatic diversion with transitory gastric restriction and duodenal bulb preservation: 88 patients since 1992.

BACKGROUND: Over 10 years, 88 patients underwent biliopancreatic diversion with transitory gastric restriction (BPD-TGR) as a first choice operation or after gastric restrictive procedures. METHODS: From 1992 to 1999, BPD-TGR was performed on 71 patients as a first choice operation (Group 1 - BMI 41.9 +/- 6.5). The TGR was achieved by a polydioxanone (PDS) band. The duodenal bulb was maintained to 5 cm distal to the pylorus, constructing an end-to-side antecolic isoperistaltic duodeno-ileal anastomosis. Since 1993, a further 17 patients underwent BPD-TGR as a correction for restrictive procedures (Group 2 - BMI 37.4, range 27.2-61.0). RESULTS: Results in weight loss in Group 1 were similar to those in our previous classical BPD. Percent excess weight loss (%EWL) was 68.0 +/- 18.4, 75.9 +/- 12.3, and 75.4 +/- 12.0 at 1,5 and 10 years respectively. No patient had severe dysproteinemia (only 3% of patients had hypoalbuminemia of 3.0-3.4 g/dl). There was no case of diarrhea or halitosis. Anastomotic ulcers occurred in 2% of the patients. In Group 2, the patients had weight loss already present from the first operation, which continued after BPD-TGR with great variability from patient to patient. %EWL was 35.1 (range 0 to 72.5) and 35.2 (range 18.4 to 43.2) at 1 and 5 years. CONCLUSIONS: BPD-TGR appears to be an effective operation with few complications and also a satisfactory correction for failed gastric restrictive procedures, or even a sequential operation in the super-obese.

Testicular sperm extraction in azoospermic men submitted to bilateral orchidopexy.

BACKGROUND: This study was carried out to evaluate whether bilateral orchidopexy represents a poor or good prognostic factor in azoospermic men undergoing testicular sperm extraction (TESE). METHODS: One hundred and seven presumed non-obstructive azoospermia (NOA) patients, according to conventional clinical parameters (volume of testis, FSH, clinical history) were submitted to testicular biopsy with TESE. Thirty men (28%) had a history of bilateral orchidopexy for cryptorchidism. RESULTS: Normal spermatogenesis or mild hypospermatogenesis was diagnosed in 12/30 ex-cryptorchid patients and in 7/77 presumed NOA patients (P = 0.0004). Conversely, pure Sertoli cell-only syndrome or complete maturation arrest was found in 10/30 ex-cryptorchid patients and in 48/77 presumed NOA patients (P = 0.0094). In 53/107 patients (49.5%), TESE allowed a positive sperm retrieval. At least one spermatozoon was observed in 22/30 ( approximately 73%) ex-cryptorchid patients and in 31/77 ( approximately 40%) presumed NOA patients (P = 0.0026). A large number of spermatozoa (equivalent to an obstructive pathology) were retrieved in 13/30 ex-cryptorchid and in 10/77 presumed NOA patients (P = 0.001). A history of bilateral orchidopexy in presumed NOA patients correlates positively for the chance of retrieving testicular spermatozoa (odds ratio 3.8; 95% confidence interval 1.41-10.21; P = 0.008). CONCLUSIONS: Although bilateral cryptorchidism is usually considered a testicular secretive dysfunction, TESE permits retrieval of a large number of spermatozoa in almost 40% of cases. Our data suggest the existence of congenital or acquired obstructive anomalies of the seminal ducts in azoospermic orchidopexed men.

The impact of embryo transfer on implantation--a review.

Embryo transfer has received little clinical attention and has been, until recently, the most inefficient step in in-vitro fertilization (IVF). In this article, the authors review the literature and their personal experience regarding the process of intrauterine transfer of embryos, which remains the object of much discussion. Factors which appear to influence implantation rates are: contamination of the catheter tip with cervical bacteria, stimulation of uterine contractions during the procedure, the type of catheter, ultrasound guidance during the transfer, and the position of the embryos in the uterine cavity. Easy and atraumatic transfer is essential for successful implantation and the embryos need to be placed in the middle of the cavity, away from the fundus. Knowing, beforehand, the position and length of the uterus can provide better results and may reduce the rate of ectopic pregnancies. Evidence from randomized studies has supported this claim. Despite the number of available studies controlling certain variables, most authors, even using the same catheter, ultrasound guidance and/or a trial transfer use different protocols or similar instruments in different ways. Standardization of the transcervical intrauterine transfer of embryos in a large randomized study is needed before definitive conclusions can be drawn. The goal of improved implantation and pregnancy rates deserve these efforts.

[Actinomycosis infection presenting as malignant pelvic frostbite]. / Infezione da actionomicosi presentatasi con un quadro di congelamento pelvico maligno.

Actinomycosis is a chronic inflammatory disease rarely observed in the pelvic form but rather frequently in the cervicofacial tract. The pelviabdominal form is difficult to diagnose because of its various clinical presentations that can lead to misdiagnosis by imitating a neoblastic disease, a bowel inflammatory disease, an acute diverticulitis or gynecological disorders. The case is reported of a 43 year old woman with prolonged use of an intrauterine contraceptive device and with severe phlogosis secondary to extensive pelvic actinomycosis involving both ovaries, the urinary duct and the rectosigmoid, misdiagnosed as pelvic frostbite presentation of a malignant neoplasm. A bilateral hysterosalpingo-oophorectomy surgery was performed and a urethral stent was placed before the operation. Rarely, has the pelvis been so heavily involved by a chronic actinomycosis infection.

Aspermia and chronic testicular pain after imperforate anus correction. Cryopreservation of sperm cells extracted from whole orchiectomized testis: case report.

This paper describes an unusual association of aspermia and untreatable, chronic testicular pain in a young man who underwent 14 surgical interventions for an imperforate anus. Physical examination and ultrasonography revealed left epididymal and vas enlargement, normal-sized testes, tubular ectasia of the left rete testis and a small intraprostatic paramedian left cyst. Retrograde ejaculation and urogenital infections were excluded, and the FSH and karyotype results were normal. The patient gave his consent to an exploratory intervention with possible radical left orchiectomy. The patency of the left distal seminal duct was unexpectedly normal, and no sperm were found in the epididymis or vas deferens despite their obstructive appearance. Sperm were only found in a 'testicular touch' preparation. The removed testis was immediately opened and most of the testicular lobules were removed, thus allowing the extraction of 25 x 10(6) sperm, which were cryopreserved in 35 straws. An 8-month follow-up examination documented the complete absence of pain and, during the next few months, it is planned to use the thawed sperm for ICSI. Radical orchiectomy plus the cryopreservation of sperm extracted from the whole testis must be considered in the case of the co-existence of chronic unilateral testicular pain and aspermia.