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Testicular Tumor in a Cryptorchid Hermaphrodite; a Case Report

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Testicular Tumor in a Cryptorchid Hermaphrodite; a Case Report CASE REPORT PROF-2366 TESTICULAR TUMOR IN A CRYPTORCHID HERMAPHRODITE; A CASE REPORT Prof. Faisal G. Bhopal Dr. Faryal Azhar, Sadaf Faisal Bhopal, Kamran Faisal Bhopal Article Citation Bhopal FG, Azhar F, Bhopal SF, Bhopal KF. Testicular tumor in a cryptorchid hermaphrodite; a case report. Professional Med J 2013; 20(6): 1058-1064. INTRODUCTION Abdomen CASE HISTORY Umbilicus was central & inverted. There was visible A 45 years old Labourer father of 5 children from bulge in Right Iliac Fossa. A palpable mass was Mandi Bahauddin was admitted through out-patient present in hypogastrium and RIF measuring 10 x 9 cm department in February, 2010. Presented with painful on ultrasound abdomen and pelvis. Large fibrous swelling in lower abdomen for last 15 days. Swelling mass in right side of pelvis 10x9 cm pushing urinary rapidly increased in size. It started in the RIF then bladder up. It was tender, hard, partly mobile, upper involved the hypogastrium and became painful. Pain limit was reachable but lower limit was not. Liver and was constant dull ache, relieved somewhat after spleen were not palpable. On digital rectal micturition. There was H/O Anorexia & Significant examination, a hard extra-rectal mass was palpable weight loss over 6 months. There was no H/O Bowel anteriorly, about 7 cm from anal verge. Scrotum was complaints or urinary complaints. not developed. Testes were not palpable in scrotum or groin bilaterally. His phallus was normally developed There was no past history of prolonged illness with external urinary meatus at the tip. There was requiring hospitalization or investigations. normal distribution of the pubic hair and other secondary sexual characters were present. In Personal history. He was Smoker for last 20 years, Respiratory and Cardiovascular system were smoked 1 pack per day. He was married with 5 unremarkable. children (4 sons 1 daughter). Youngest is 3 years old. He belonged to Lower Middle class. Family history PROVISIONAL DIAGNOSIS of Bilateral undescended was Insignificant. testes with Lower Abdominal Mass (? Testicular Tumor) was made. On examination, he was Middle aged man sitting comfortably, well oriented with Pulse of 82/min, BP: INVESTIGATIONS 110/70 mm of Hg, Temp: 98fh & R/R: 18/min. FBC: Hb 13.4 g/dl, TLC 9100 /cumm, There was no pallor, Jaundice, Clubbing, Cyanosis or Platelets 230000 /cumm Pedal oedema. JVP was normal. No Lymph nodes or ESR: 29, Thyroid swelling were palpable. Neutrophils 77%, Lymphocytes 12%, Systemic examination was Unremarkable Monocytes 6%, LFTs : Bilirubin : 0.8 mg/dl, Oro-dental hygiene was good. ALT: 19, Professional Med J 2013;20(6): 1058-1064. www.theprofesional.com 1058 TESTICULAR TUMOR IN A CRYPTORCHID HERMAPHRODITE 2 PT: 15/13, MRI pelvis APTT: 31/30, 20 cm sized mass in the pelvis extending into the lower BSR: 135 mg/dl, abdomen with well defined borders. Uterine cavity RFTs : Urea: 33 mg/dl. rudimentary vagina can be appriciated. Alpha Feto Protein: 4.19 ng/ml (Normal value upto 8.4) Conclusion B-HCG: 15.6 mIU/ml (Normal value <5), Tumour arising from right undescended testis. Lymph S. LDH: 925 U/L (Normal value < 480) nodes behind the uterus forming a mass, pushing the urinary bladder forward. Semen Analysis Azoospermia Diagnosis of testicular tumour with bilateral Ultrasound abdomen and pelvis: Large fibrous mass cryptorchidism was made. in right side of pelvis 10x9 cm pushing urinary bladder up. Professional Med J 2013;20(6): 1058-1064. www.theprofesional.com 1059 TESTICULAR TUMOR IN A CRYPTORCHID HERMAPHRODITE 3 Left Testis at the end of fallopian tube & uterus with LN mass, after removal of Right testicular tumour. Resected specimen Showing, Uterus with both fallopian tubes, LN mass behind uterus, left testis at the end of left fallopian tube & right testicular tumour. OPERATIVE FINDINGS A Large right testicular intra abdominal mass. Uterus with fallopian tubes and blind ended vagina was present. Left intra abdominal testis was normal looking. There was a Large lymph node mass in the hypogastrium behind the uterus, extending to blood vessels and right ureter. No ascites or liver mets were present. OPERATIVE PROCEDURE Right testicular tumour mass was resected. Uterus with fallopian tubes, left testis and blind ended vagina Left Normal Testis were resected en-block with lymph nodes except a rim Professional Med J 2013;20(6): 1058-1064. www.theprofesional.com 1060 TESTICULAR TUMOR IN A CRYPTORCHID HERMAPHRODITE 4 Resected specimen Showing, Uterus & both fallopian tubes opened, LN mass behind uterus, left testis at the Right testicular tumour Cut section. end of left fallopian tube. right testicular tumour. of tissue which was engulfing iliac blood vessels. Sexual differentiation is on the basis of Psychological Sex and Organic sex Post-Operative Course Post-Operative Course was uneventful. Patient was Organic sex depends on Chromosomal Sex, Gonadal referred to NORI Hospital for further treatment (Chemo sex & Phenotypic sex / Radiotherapy). The Blood Group of the patient and all his children was Male done which was “O” positive. XY, Gonad-Testes, Mullerian regression factor. DNA testing and karyotyping was not done because of Testosterone is responsible for development of psychosocial issues. Wollfian duct, while DHT for development of External genetalia HISTOPATHOLOGY 2 specimens were sent for histopathology. Female Specimen1was tumour & specimen 2 was of left XX, female because of presence of Ovary, Absence of testis and uterus. Specimen 1: was reported as Testes, No testosterone to develop Wollfian duct. classical seminoma. Specimen 2: Left testis showing Absent Mullerian regression factor results in atrophy. Nodes showing metastatic seminoma with a persistence of Mullerian duct structure. triangular cavity in front of this mass. DISCUSSION Intersex KEY ISSUES: were Disparity between chromosomal, gonadal and 1) Presence of Uterus with fallopian tubes and phenotypic sex. blind ended vagina. 2) Seminoma Testis in Right Undescended Summary in our case, Phenotypic male with well Testis. developed phallus and other secondary sexual 3) Fertility issues in Patients with Bilateral character. But gonads were bilaterally impalpable. Cryptorchidism. Intra- abdominal testes. Lt testis normal & Classical seminoma in right testis (Gonadal sex-male). Professional Med J 2013;20(6): 1058-1064. www.theprofesional.com 1061 TESTICULAR TUMOR IN A CRYPTORCHID HERMAPHRODITE 5 Presence of female internal organs. What is the families1. possibility? Epidemiology and other outcome studies Presented at In Male Pseudohermaphrodite, Gonad could both be the Society for Fetal Urology Biannual Meeting, San testes/ovary. Chromosomal and gonadal sex-male but Francisco, California, May 7, 2004. external genitalia-female. Impaired synthesis, secretion, conversion or action of Paternity rates among formerly cryptorchid and testosterone. Impaired synthesis, secretion, or failure control men are shown in Table2 to respond to mullerian regression factor. Female genital duct in otherwise normal male. This is one Giltay et al. describe an unusual case of true possibility hermaphroditism—that is, the presence of both ovaries and testes in a single individual. The boy's Female Pseudohermaphrodite somatic tissues are likewise a mixture of Chromosomal and gonadal sex-female but external karyotypically normal male and normal female cells3. genitalia-male. Classical example congenital adrenal hyperplasia. Our case does not fit into this category. Zayed F et al reported A male phenotype (XY) hermaphrodite treated for seminoma, fathered a Defect in chromosomal / gonadal sex healthy child by IVF–ICSI technique4. It is generally Mixed gonadal dysgenesis is common cause of known that almost all hermaphrodites are infertile, intersex. Phenotype female or male. however, German et al. observed spermatogenesis in an hermaphrodite5. Furthermore, in abstract, both Testes are located intra-abdominally. Uterus,vagina Inatomi et al. and Manba et al. have reported the and at least one fallopian tube invariably present. High delivery of infants fathered by a true hermaphrodite incidence of tumour development in gonad. Karyotype under natural conditions in Japan6. 45x/46xy but always chromatin negative. This is another possibility. A 38years old paient was seen in 2001 by urologist for infertility and impalpable in the right side of the Cryptorchidism pelvis3456789. He had laparotomy, and right sided Cryptorchidism is the most common genital problem orchidectomy of a right testicular seminoma which encountered in pediatrics. Cryptorchidism literally was excised in addition to what appeared as a uterus means hidden or obscure testis and generally refers to and tubes in 2001. This patient getting treated and an undescended or maldescended testis. Overall, 3% cured from seminoma, the couple had in vitro of full-term male newborns have cryptorchidism, fertilization (IVF) and intracytoplasmic sperm injection decreasing to 1% in male infants aged 6 months to 1 (ICSI) using frozen testicular sperm and had a healthy year. The prevalence of cryptorchidism is 30% in baby7. premature male neonates. Factors that Predispose to cryptorchidism include prematurity, low birth weight, A successful pregnancy outcome using frozen small size for gestational age, twinning, and maternal testicular sperm from a chimeric infertile male with a exposure to estrogen during the first trimester. A recent 46, XX/46, XY karyotype was reported in 2005 by study found that almost 23% of index patients with Sugawara et al8. A successful second
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