Tumor de Células de Sertoli del ovario: Reporte de un caso / Sertoli cell tumor of the ovary. A case report

RESUMEN Introducción: Los tumores de Células de Sertoli son una entidad rara, con una frecuencia de 0,2-0,5 % del cáncer de ovario. El diagnóstico inicial de este tipo de tumores es difícil, debido a las variedades histológicas que presenta y a su diferenciación. Objetivo: Demostrar el valor del completamiento del proceso diagnóstico con técnicas de inmunohistoquímica en tumores ováricos para identificar una variante rara como el tumor de Células de Sertoli. Presentación del caso: Se presenta un caso de un tumor de ovario primitivo de Células de Sertoli en una paciente de 43 años de edad, con antecedentes personales de hipotiroidismo, intervenida quirúrgicamente por un tumor sólido del ovario derecho en el Hospital General Docente "Dr. Agostino Neto". En el intraoperatorio, se encontró un tumor gigante de aspecto maligno. El resultado anatomopatológico informó un tumor carcinoide típico. La paciente fue remitida al Instituto Nacional de Oncología y Radiobiología, donde la inmunohistoquímica de la biopsia informó un tumor de Células de Sertoli. Actualmente la paciente se encuentra asintomática. Conclusiones: El diagnóstico anatomopatológico preciso y la inmunohistoquímica correcta son muy importantes para el manejo y tratamiento de este tipo de tumor.

ABSTRACT Introduction: Sertoli cell tumors are a rare entity with an incidence of ovarian cancer of 0,2-0,5 %. The initial diagnosis of this type of tumors is difficult to make due to the histological varieties and its differentiation. Objective: To demonstrate the value of immunochemical techniques in the completion of the diagnostic process of ovarian tumors aimed at identifying a rare variant such as Sertoli cell tumor. Case presentation: A 43-year old woman with a primitive Sertoli cell tumor of the ovary is presented. The patient had personal history of hypothyroidism. She underwent surgery at "Dr. Agostinho Neto" General Teaching Hospital because she had been diagnosed with a solid tumor of the right ovary. During the intraoperative period, a giant tumor with a malignant appearance was found. The pathological result reported a typical carcinoid tumor. The patient was referred to the National Institute of Oncology and Radiobiology where the immunohistochemistry of the biopsy reported a Sertoli cell tumor. Currently, the patient is asymptomatic. Conclusions: The accurate pathological diagnosis and the correct immunohistochemistry are very important for the management and treatment of this type of tumor.

SUMO specific peptidase 3 regulates autophagy in mouse testicular Sertoli cells / 上海交通大学学报（医学版）

Objective: To investigate the regulation of autophagy by SUMO specific peptidase 3 (SENP3, normally called SUMO specific protease 3) in mouse testis. Methods: Immunofluorescence was used to detect the localization of SENP3 in spermatogenic cells and Sertoli cells of testis. Senp3 wild type (Senp3+/+) mice and Senp3 gene knockout heterozygous (Senp3+/-) mice were subjected to starvation treatment to induce autophagy. Testicular tissue proteins were extracted, and the extent of autophagy was detected by Western blotting. The extent of autophagy of Sertoli cells was detected and compared with that of spermatogenic cells in testis by transmission electron microscopy and immunofluorescence. Results: SENP3 mainly localized in the nucleus of Sertoli cells. Compared to Senp3+/+ mice, the extent of starvation-induced autophagy in Sertoli cells of Senp3+/- mice increased. Conclusion: SENP3 can inhibit autophagy in Sertoli cells during nutrient deficiency, which may play a role in controlling the extent of autophagy.

Malignant Sertoli cell tumor of the testis masquerading as seminoma with bone metastasis

Sex cord–stromal tumors of the testes are rare malignancies as compared to germ cell tumors. Pure Sertoli cell tumors are still rare representing <1% of testicular cancers and the malignant forms are too rare. Furthermore, the occurrence of metastasis in such cases is extremely rare with <30 cases reported in literature so far to the best of our knowledge. We present herein a case of malignant Sertoli cell tumor in a 48-year-old male who was initially misdiagnosed as seminoma based on histology and clinical presentation. Four months later, he presented with symptoms due to bony metastasis and found to have widespread metastatic disease which is a very rare presentation of Sertoli cell tumors. Diagnosis of sex cord–stromal tumor requires high index of suspicion as these tumors are most of the times misdiagnosed as germ cell tumors due to their rare incidence and atypical presentation leading to mismanagement. Timely diagnosis at an early stage can provide therapeutic benefit due to lack of well-defined treatment options at advanced stages.

Expression of CLAUDIN-11 in the testicular tissue of the patient with non-obstructive azoospermia and its clinical significance / 中华男科学杂志

<p><b>Objective</b>To study the expression of CLAUDIN-11 in the testis tissue of non-obstructive azoospermia (NOA) patients with different severities and investigate its clinical significance.</p><p><b>METHODS</b>Sixty-two NOA patients were divided into a hypospermatogenesis (HS) group (n = 30) and a Sertoli cell only syndrome (SCO) group (n =32). The expression of CLAUDIN-11 in the testicular tissue of the patients was detected by immunohistochemistry, that of CLAUDIN-11 mRNA determined by real-time fluorescence quantitative polymerase chain reaction (RT-qPCR), and the levels of serum reproductive hormones measured by chemiluminescent immunoassay.</p><p><b>RESULTS</b>Immunohistochemistry showed that the expression of CLAUDIN-11 was mainly in the cytoplasm of the Sertoli cells around the seminiferous tubule wall in the HS group, but diffusely distributed in the membrane of the Sertoli cells in the SCO group. RT-qPCR revealed a significantly lower expression of CLAUDIN-11 mRNA in the HS than in the SCO group (0.008 ± 0.001 vs 0.013 ± 0.002, t = 10.616, P<0.01). The level of serum luteotropic hormone (LH) was also markedly lower in the HS than in the SCO group (［3.62 ± 1.34］ vs ［4.96 ± 3.10］ IU/L, P<0.05) and so was that of follicle-stimulating hormone (FSH) (［5.36 ± 2.80］ vs ［10.65 ± 9.18］ IU/L, P<0.05).</p><p><b>CONCLUSIONS</b>The up-regulated expression of CLAUDIN-11 in Sertoli cells may play an important role in the development and progression of spermatogenic dysfunction in NOA patients.</p>

Large-cell calcifying Sertoli cell tumour with macrocalcification in partially resected testis of young adult patient

@#Introduction: There are less than 100 cases of Large-cell calcifying Sertoli cell tumour (LCCSCT) reported in English literature. Most of them are benign, bilateral and affect paediatric population. Malignant cases occur in older patients. LCCSCT is often associated with Carney complex or PeutzJaghers syndrome. We present the clinicopathological features of a young adult, with unilateral “stonelike” LCCSCT, without changes in hormonal status and no clinical characteristics of noted genetic disorders. Case Report: A 24-year-old male presented with painless hardening of the right testis. There was no gynaecomastia, and serum levels of human chorionic gonadotropin and α-fetoprotein were normal. Ultrasound depicted hyperechogenic, clearly demarcated intratesticular lesion. Partial orchiectomy was performed. Macroscopically, tumour appeared as almost entirely calcified round mass, measuring 10 mm. Histopathological evaluation showed well-circumscribed, unencapsulated tumour composed of massive calcified geographic formations, surrounded with tumour cells. Neoplastic cells were large, polygonal, with abundant eosinophilic cytoplasm, and formed irregular cords, pseudo tubular structures, and nests in a fibrous and myxoid stroma, surrounded with lymphocytes. Other forms of calcification were also present: Needle-like deposits and lamellar, mulberry-like structures. There was no necrosis, mitotic activity and nuclear pleomorphism. Immunohistochemical study was positive for inhibin α and negative for Melan A, EMA, synaptophysin, chromogranin and AFP. Discussion: LCCSCT needs to be differentiated from other, more frequent, sex cord stromal tumours. Clinical and genetical evaluation of these patients had to be performed, due to connection of LCCSCT with genetic abnormalities. In evidently benign cases, organ-sparing surgery should be considered for younger patients, followed by long term follow-up.

In vitro chemokine (C-C motif) receptor 6-dependent non-inflammatory chemotaxis during spermatogenesis

BACKGROUND: Chemokine (C-C motif) receptor 6 (CCR6) is present in sperm and plays a significant role in sperm motility and chemotaxis acting in the reproductive tracts. However, the expression and functional significance of CCR6 in testis are still poorly understood, especially in the process of spermatogenesis. METHODS AND RESULTS: CCR6 was expressed in spermatogenic cell lines and its expression was shown in an age-dependent upregulation manner from puberty to adulthood in mouse testis. Immunostaining results confirmed the localization of CCR 6 in testis. Further chemotaxis assays demonstrated that spermatogenic cells GC-1 and -2 exhibited a directional movement toward CCR6-specific ligand such as CCL20 or Sertoli cells in vitro. CONCLUSIONS: The present findings indicate that CCR6 is involved in the chemotaxis of spermatogenic cells in vitro and promotes chemotaxis under non-inflammatory conditions during normal spermatogenesis.

Tumor de células de sertoli esclerosante. Reporte de un caso / Sclerosing sertoli cell tumor. Case report

RESUMEN El tumor esclerosante de células de Sertoli del testículo es una entidad patológica infrecuente, con todo, que solo se han descrito 22 casos en la literatura científica. Son descritos como tumores unilaterales que en su mayoría se presentan entre la tercera y sexta década de vida, siendo carentes de malignidad, sin embargo, en ninguno de los casos reportados se han realizado seguimientos por periodos prolongados. Se presenta un caso clínico cuyo objetivo es socializar el primer caso de tumor esclerosante de células de Sertoli (SSCT), diagnosticado por el departamento de patología de la Universidad industrial de Santander, que se registra en Colombia. Después de tres años de seguimiento clínico el paciente no presenta recidiva, así como tampoco otras lesiones tumorales. Se considera la orquiectomia el tratamiento curativo.(AU)

SUMMARY Sclerosing tumor of Sertoli cells of the testis is an infrequent pathological entity, only 22 cases have been described in the scientific literature. They are described as unilateral tumors that mostly occur between the third and sixth decade of life, being devoid of malignancy, however, in none of the cases have been followed for prolonged periods. We present a clinical case whose objective is to socialize the first case of Sertoli cell sclerosing tumor (SSCT), diagnosed by the pathology department of the industrial university of Santander, which is registered in Colombia. After three years of clinical follow-up, the patient does not present recurrence, as well as other tumor lesions. Orchiectomy is considered the curative treatment.(AU)

H₂O₂ alters metabolism in TM4 Sertoli cells in the mouse / 中华男科学杂志

Objective@#To explore the mechanisms of oxidative stress-induced damage to TM4 Sertoli cells in the mouse using metabolomics techniques based on gas chromatography-mass spectrometry (GC-MS).@*METHODS@#We established the model of oxidative stress-induced damage to mouse TM4 Sertoli cells by treatment with H₂O₂. Then, we detected the survival rate and apoptosis rate of the TM4 cells by MTT and flow cytometry respectively, measured the concentration of ROS in the TM4 cells with the DCFH-DA fluorescent probe, and determined the levels of endogenous metabolites in the TM4 cells by GC-MS after H₂O₂ intervention.@*RESULTS@#After 2 hours of treatment with H₂O₂ at 600 μmol/L, the survival rate of the TM4 cells was reduced to about 50%, and the total apoptosis rates in the low- (100 μmol/L), medium- (300 μmol/L), and high-dose (600 μmol/L) groups were (19.45 ± 0.53), (20.12 ± 0.58), and (37.13 ± 0.35)%, respectively, increased in a dose-dependent manner as compared with (10.28 ± 0.35)% in the blank control (P 1, P <0.05).@*CONCLUSIONS@#Oxidative stress-induced damage and apoptosis of TM4 Sertoli cells are closely associated with the metabolism of amino acid, glucose, and energy in the cells.

Role of TGF-β1 in Sertoli cells and tight junction / 中华男科学杂志

Objective@#To explore the role of TGF-β1 in the proliferation and apoptosis of Sertoli cells and its effect on the expressions of tight junction-related proteins and genes in rats.@*METHODS@#Rat Sertoli cells were isolated in vitro, primarily cultured, and divided into groups A (blank control), B (TGF-β1 receptor blocker), C (TGF-β1), and D (TGF-β1 + receptor blocker). The proliferation and apoptosis of the cells were detected by CCK-8 and flow cytometry, respectively. After establishment of the dual-chamber model for the primary culture of Sertoli cells, the trans-epithelia electrical resistance (TER) value was measured and the relative expressions of Occludin, ZO-1 and Claudin Ⅱ determined by RT-PCR and Western blot.@*RESULTS@#The OD value of the proliferation of the Sertoli cells was markedly higher in group C than in groups A and D (0.79 ± 0.04 vs 0.66 ± 0.05 and 0.68 ± 0.02, P0.05). The TER value was dramatically decreased in group C as compared with groups A and D (［176.37 ± 16.61］ vs ［281.42 ± 9.83］ and ［254.37 ± 13.55］ /cm2, P0.05) or their protein expressions (F = 0.28 and 1.31, P>0.05). Both the mRNA and protein expressions of Occludin were markedly lower in group C than in A and D (P<0.01 and P<0.05), with statistically significant differences among the four groups (F = 6.86 and 6.87, P<0.01).@*CONCLUSIONS@#TGF-β1 can promote the proliferation of Sertoli cells in rats and act on the tight junction of the cells by regulating the expression of Occludin.

Sertoli cell nodule of the testis: An incidental finding

The case of a 21year old man with complaints of pain in the lower abdomen reported in the surgery OPD. USG showed a case of undescended testis. A radical Orchidectomy was performed and the specimen was received in the department of pathology. Grossly the testis was normal .Microscopic examination showed the encapsulated nodule composed of tubules lined by immature Sertoli cells interspersed with tubules and Spermatogonia and an interwoven network of hyalinised basement membrane having foci of calcification. The findings were identical to those of a Sertoli cell nodule-A typically microscopic encapsulated lesion commonly detected in crypt orchid testes. Here we present a case of undescended testis where incidentally we found all the features of sertoli cell nodule

Probucol alleviating mouse Sertoli cell injury induced by oxygen-glucose deprivation/reoxygenation / 实用医学杂志

Objective To investigate the effect of probucol on Sertoli cell injury induced by oxygen-glucose deprivation/reoxygenation(OGD/R)and mechanism. Methods Primary Sertoli cells treated with oxygen glucose deprivation for 2 h and reoxygenation for 3 h were modeled. We incubated Sertoli cells with probucol in a concentration of 5,10,and 20 μmol/L from reoxygenation to the end. The cell viability was evaluated using Cell Proliferation Assay kit. ROS production was observed by flow cytometry. The levels of MDA ,iron and GSH or the activity of glutathione-dependent peroxidase were measured by Assay Kit. Results The ferroptosis inhibitors pre-vented cell death(P < 0.01). The cell death rate was decreased by probucol in time- and dose-dependent manner (P < 0.01). Meanwhile, probucol caused increase of ROS,MDA,GSH content and GPXs activity(P < 0.05). Conclusion Probucol protects Sertoli cells from OGD/R damage via inhibiting lipid peroxidation-mediated ferroptosis.

The significance of serum inhibin B levels in evaluating the Sertoli cell function of cryptorchidism / 中华泌尿外科杂志

Objective To investigate the value of serum inhibin B in evaluating the Sertoli cell function in cryptorchidism children by the study of the correlation between the serum inhibin B levels and cryptorchidism.Methods From August 2006 to August 2011,672 cryptorchidism aged from 6 to 72 months (mean age 27.7 months) were measured of serum inhibin B level by ELISA.Of those 672 boys,505 were unilateral,106 were bilateral and 61 were unilateral vanished testis.Three hundred and eighty-five cases aged 6 to 24 months,109 cases aged 25 to 36 months and 178 cases aged 37 to 72 months.The control group consisted of 335 boys selected from physical examination or circumcision with corresponded age,98 cases aged 6 to 24 months,101 cases aged 25 to 36 months and 136 cases aged 37 to 72 months.The blood samples for serum inhibin B were taken from everyone and,in part of cases,6 months,12 months,18 months postoperatively respectively.The comparision of serum inhibin B levels among the groups was done,and the correlation between serum inhibin B level and testicular volume was analyzed.Results Compared with the control group,the serum inhibin B levels of all groups with unilateral and bilateral cryptorchidism are lower significantly (P ＜ 0.05).No difference was found between the unilateral vanished testis and the control group (P ＞ 0.05),except those aged 6 to 24 months [38.54 pg/ml (25.98,50.24) pg/ml vs.68.04 pg/ml (44.95,115.64) pg/ml,P ＜0.05].For bilateral cases aged 6 to 24 months,the serum inhibin B level are significantly lower than that of unilateral cases [30.68 pg/ml(17.37,42.43)pg/ml vs.45.91 pg/ml (30.98,69.70) pg/ml,P ＜ 0.05].The serum inhibin B levels were significantly increased 6 months later in those who had undergone orchidopexy [37.34 pg/ml (22.79,64.25) pg/ml vs.30.48 pg/ml (16.56,50.08) pg/ ml,P ＜ 0.05].For boys with unilateral and bilateral cryptorchidism aged 6 to 24 months,inhibin B levels correlated positively with testicular volume (P ＜ 0.05).Conclusions The serum inhibin B level can be used as a reliable marker in evaluating the Sertoli cell function of cryptorchidism,and the orchidopexy had a positive impact on the serum levels of inhibin B.

Sertoli cell tumor arising in a cryptorchid testis presenting as a content of inguinal hernial sac.

Sertoli cell tumors (SCTs) are rare tumors accounting for <1% of all testicular tumors. Here, we report a rare case of SCT in a 60‑year‑old man presenting as a painless swelling in the right groin since childhood. Clinically, he presented with right‑sided inguinal hernia with absence of the right testis. He had normal left testis and had no gynecomastia or infertility. The specimen of hernial sac showed testis with a 1.6 cm × 1.5 cm nodular mass having gray tan‑cut surface. Histopathologically, the testis showed atrophy and the nodular portion showed tumor cells arranged in tubular and microcystic pattern, with no solid pattern or necrosis. The diagnosis of SCT was confirmed with immunohistochemical staining for inhibin which showed fine granular cytoplasmic positivity. Cryptorchid testis having SCT and presenting as a content of inguinal hernia is a rare occurrence.

5'-flanking regulatory sequence methylation of the Boule gene in the testis tissue of infertile men with Sertoli cell-only syndrome / 中华男科学杂志

<p><b>Objective</b>To investigate the 5'-flanking regulatory sequence methylation status of the Boule gene in the testis tissue of infertile men with Sertoli cell-only syndrome (SCOS).</p><p><b>METHODS</b>We collected biopsy samples of the testis tissue from 12 men with obstructive azoospermia (the control group) and 15 cases of SCOS, all without varicocele, cryptorchidism, or infectious disease. We extracted genomic DNA from the testis tissue of the SCOS patients, analyzed the characteristics of the 5'-flanking regulatory sequence of the Boule gene using the bioinformatics method, and detected the methylation status of the Boule gene by sodium bisulfite sequencing.</p><p><b>RESULTS</b>A CpG island was observed in the 5'-flanking regulation region of the Boule gene. The methylation level of the Boule gene was remarkably higher in the SCOS group than in the obstructive azoospermia controls (61.4% vs 21.7%, P<0.01), with significant differences in the methylation levels of 14 CpG sites, namely, －58 bp, －50 bp, －48 bp, －38 bp, －28 bp, －24 bp, －20 bp, －15 bp, －1 bp, +5 bp, +8 bp, +15 bp, +29 bp, and +58 bp.</p><p><b>CONCLUSIONS</b>The methylation level of the Boule gene is significantly higher in the SCOS patients than in the obstructive azoospermia males, which suggests that the changes in Boule methylation may be associated with spermatogenic dysfunction.</p>

Renal protection of Jiangzhi Tongluo Soft Capsule on membranous nephropathy rats and effects on expression of Bcl-2 and Bad in renal tissue / 中草药

Objective: To observe the effects of Jiangzhi Tongluo Soft Capsule on blood biochemical indexes and expression of Bcl-2 and Bad in membranous nephropathy (MN) rats, explore the renal protective effects and possible mechanism of Jiangzhi Tongluo Soft Capsule on MN rats. Methods: Total 60 SD rats were randomly selected, 10 for normal group, the other 50 for MN model establishment, then randomly divided into model group, Benazepril group, and Jiangzhi Tongluo Soft Capsule group (25, 50, and 100 mg/kg), each group was treated according to the corresponding dose. The 24 h urinary protein quantitative (UTP), serum total cholesterol (TC), triglyceride (TG), total protein (TP), albumin (ALB), blood urea nitrogen (BUN), and serum creatinine (Scr) in rats in the 4th weekend were tested; The renal pathological morphology changes were observed by electron microscope and immunofluorescence technique, and detecting the expression of Bcl-2, Bad by immunohistochemical and real-time PCR methods. Results: Compared with the model group, UTP, TC, and TG in the treatment groups were lower (P 0.05), and the effect was better than Jiangzhi Tongluo Soft Capsule 25 mg/kg group (P 0.05). Compared with the model group, the expression of Bcl-2 mRNA significantly raised (P 0.05), and the effect was better than that in Jiangzhi Tongluo Soft Capsule 25mg/kg group (P < 0.05). Conclusion: The renal protection of Jiangzhi Tongluo Soft Capsule on MN rats may be related to the function of raising the expression of Bcl-2, downgrading the expression of Bad, inhibiting the sertoli cell apoptosis, and repairing the damaged cells.

Varicocele management in the era of in vitro fertilization/intracytoplasmic sperm injection / 亚洲男科学杂志(英文版)

Varicocele is the most common surgically treatable cause of male infertility, and often results in alterations in semen parameters, sperm DNA damage, and changes to the seminal milieu. Varicocele repair can result in improvement in these parameters in the majority of men with clinical varicocele; data supporting repair in men with subclinical varicocele are less definitive. In couples seeking fertility using assisted reproductive technologies (ARTs), varicocele repair may offer improvement in semen parameters and sperm health that can increase the likelihood of successful fertilization using techniques such as in vitro fertilization (IVF) or intracytoplasmic sperm injection (ICSI), or may decrease the level of ART needed to achieve successful pregnancy. Male infertility is an indicator of general male health, and evaluation of the infertile male with an eye toward future health can facilitate optimal screening and treatment of these men. Furthermore, varicocele may represent a progressive lesion, offering an argument for its repair, although this is currently unclear.

Claudin-11 and occludin are major contributors to Sertoli cell tight junction function, in vitro / 亚洲男科学杂志(英文版)

The Sertoli cell tight junction (TJ) is the key component of the blood-testis barrier, where it sequesters developing germ cells undergoing spermatogenesis within the seminiferous tubules. Hormonally regulated claudin-11 is a critical transmembrane protein involved in barrier function and its murine knockout results in infertility. We aimed to assess quantitatively the significance of the contribution of claudin-11 to TJ function, in vitro, using siRNA-mediated gene silencing. We also conducted an analysis of the contribution of occludin, another intrinsic transmembrane protein of the TJ. Silencing of claudin-11 and/or occludin was conducted using siRNA in an immature rat Sertoli cell culture model. Transepithelial electrical resistance was used to assess quantitatively TJ function throughout the culture. Two days after siRNA treatment, cells were fixed for immunocytochemical localization of junction proteins or lyzed for RT-PCR assessment of mRNA expression. Silencing of claudin-11, occludin, or both resulted in significant decreases in TJ function of 55% (P < 0.01), 51% (P < 0.01), and 62% (P < 0.01), respectively. Data were concomitant with significant decreases in mRNA expression and marked reductions in the localization of targeted proteins to the Sertoli cell TJ. We provide quantitative evidence that claudin-11 contributes significantly (P < 0.01) to Sertoli cell TJ function in vitro. Interestingly, occludin, which is hormonally regulated but not implicated in infertility until late adulthood, is also a significant (P < 0.01) contributor to barrier function. Our data are consistent with in vivo studies that clearly demonstrate a role for these proteins in maintaining normal TJ barrier structure and function.

Complexo de Carney esporádico com tumor testicular e mixoma atrial: relato de caso / Sporadic Carney complex with testicular tumor and atrial myxoma: case report

O complexo de Carney é uma rara forma de neoplasia endócrina múltipla familial autossômica dominante. Está associado à alteração de pigmentação cutânea e mucosa, doença nodular adrenal pigmentosa primária, mixomas cardíacos e cutâneos, adenomas hipofisários funcionantes, neoplasia testicular, adenoma ou carcinoma de tireoide, além de cistos ovarianos. Aproximadamente 70% dos indivíduos diagnosticados com complexo de Carney têm pais afetados, e 30% apresentam forma esporádica. O objetivo deste estudo foi relatar um caso de complexo de Carney esporádico por mixoma cardíaco e tumor testicular. Ressalta-se a importância do caso por sua raridade e sua forma curiosa de apresentação. Homem, 33 anos, manifestou dois quadros de acidentes vasculares cerebrais em 4 meses. Na investigação apresentou pressão arterial elevada com sopro sistólico discreto e fraqueza muscular (força grau 4 em membro superior direito e grau 3 em membro inferior direito). História mórbida de tumor testicular de células de Sertoli há 7 anos com orquiectomia bilateral. História familiar sem particularidades. Na investigação, evidenciaram-se sobrecarga atrial esquerda ao eletrocardiograma e massa tumoral pedunculada compatível com mixoma atrial esquerdo ao ecocardiograma transesofágico. Foi configurada síndrome de Carney pela presença de dois critérios maiores, e o paciente foi submetido à atriotomia esquerda, com ressecção da massa tumoral e confirmação anatomopatológica. A curiosa apresentação do caso recorda que, diante de um caso de acidente vascular cerebral em paciente jovem, a suspeita clínica seja direcionada a causas mais raras. O complexo de Carney esporádico é raro, dificultando ainda mais a elucidação.

Carney complex is a rare form of autosomal dominant multiple endocrine neoplasia familial. Changing skin pigmentation and mucos, primary pigmented nodular adrenal disease, cardiac and cutaneous myxomas, functioning pituitary adenomas, testicular cancer, thyroid adenoma or carcinoma is associated, and ovarian cysts. Approximately 70% of individuals diagnosed with Carney complex have affected parents and 30% have sporadically. The aim of this study was to report a case of sporadic Carney complex due to cardiac myxoma and testicular tumor. We emphasized the importance of the case for its rarity and curious form of presentation. Man, 33, showed two episodes of strokes in 4 months. In research presented high blood pressure with mild systolic murmur and muscle weakness (grade 4 strengthin the right arm and grade 3 in the right lower limb). Morbid history of testicular Sertoli cell tumor 7 years ago with bilateral orchiectomy. No special family history. On investigation, left atrial enlargement and was evident on the electrocardiogram, and transesophageal echocardiogram revealed the presence of pedunculated tumor mass setting a left atrial myxoma. Carney's syndrome was characterized by the existence of two major criteria and patient underwent left atriotomy with resection of the tumor mass and anatomic-pathologic confirmation. The curious case presentation reminded us that before a case of stroke in a young patient should direct the clinical suspicion for rarer causes. The Carney complex sporadic is rare, yet difficult to elucidate.

Diagnostic utility of WT-1, AR and Ki-67 in Sertoli cells and germ cells for testicular biopsy / 临床与实验病理学杂志

Purpose To explore the specific diagnostic markers of Sertoli cells and germ cells for testicular biopsy. Methods Normal testis from 3 patients who suffered from carcinoma of the prostate and treated with testis castration, and 15 testicular biopsy tissues were stained by EnVision two steps with WT-1, AR and Ki-67. Results The expression of WT-1 and AR protein were found in Sertoli cells from the 3 normal testis and 15 testis biopsy, the positive rate were all 100%, and non was positive in germ cells. The germ cell was positive for Ki-67, and the positive rate was 100%. Conclusions WT-1, AR and Ki-67 may be the specific diagnostic markers of Sertoli cells and germ cells for testicular biopsy, which may contribute to the diagnosis of testicular biopsy.