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Formation of an Artificial Vagina in a Case of Testicular Feminization A

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Formation of an Artificial Vagina in a Case of Testicular Feminization A Postgrad Med J: first published as 10.1136/pgmj.43.496.117 on 1 February 1967. Downloaded from Case reports 117 Formation of an artificial vagina in a case of testicular feminization A. E. R. BUCKLE J. WATSON M.B.(Lond.), F.R.C.S., M.R.C.O.G. M.A., M.B.(Cantab.), F.R.C.S. Consultant, Department of Obstetrics and Gynaecology Consultant Plastic Surgeon Lewisham General Hospital, London, S.E.13 THE syndrome of testicular feminization is found oxosteroids 18-2 mg in 24 hr; urinary 17-hydroxy- in persons of female phenotype, which includes corticoids 23-9 mg in 24 hr; pituitary follicle- female external genitalia, who have primary stimulating hormone negative (less than 6 mouse amenorrhoea; the labia tend to be under- units); buccal smears showed no sex chromatin developed, the vagina ends blindly and the cervix present. Later analysis showed a chromosome is absent. Usually pubic and axillary hair is scanty number of 46 with normal XY complement. or absent after puberty and breast development is With an etablished diagnosis of testicular good. The internal genitals are absent or rudi- feminization syndrome, it was decided to attempt mentary and the gonads, which may be intra- plastic vaginal enlargement together with removal abdominal or in the inguinal canal, are testes of the gonads, because of the risk of malignant (Morris, 1953). The chromosomes appear normal with XY sex chomosomes. The condition is in- change (Morris, 1953). At the time that this patient herited, either in a sex-linked recessive or sex- was originally seen, four other cases of the testicu- limited dominant manner (Taillard & Prader, lar feminization syndrome (one with a teratoma of 1957). the intra-abdominal testis) were under intensive by copyright. Although the vagina may be of sufficient length biochemical investigation in the hospital and the to allow of coitus, this is not always the case. patient was therefore further investigated prior to Reports on the surgical correction of the in- surgery. adequate vagina are rare and we would like to report on the management of a patient with Further investigations testicular feminization who requested plastic en- Plasma 17-hydroxycorticoids: at the lower limit largement prior to marriage. of the female range. Plasma 1 7-oxosteroids: upper limit of the Case report female range and more typical of levels found in The patient, aged 35 years, attended as an out- men. patient in May 1964. She gave a history of pri- Plasma testosterone levels: in the male range http://pmj.bmj.com/ mary amenorrhoea on account of which she had and definitely exceeding the range found in nor- sought advice at another hospital 12 years earlier. mal females. At that time she had been told that the uterus was Urinary oestrogen levels: at the upper limit of absent and that the vagina was very short; no the normal male range, below the average daily further investigation was carried out and, although amount excreted by a normal female during a surgical correction was mentioned, no arrange- complete cycle, but higher than in women with ments were made for this to be done. She was now primary amenorrhoea. intending marriage and sought advice regarding The effect of ACTH and HCG administration on October 2, 2021 by guest. Protected the possibility of operation to allow of normal on the daily excretion of oestradiol 17-,8, oestrone marital relations. The family history was negative. and oestriol and on plasma testosterone was esti- On examination the patient was tall (height mated before operation. 1-9 m), attractive, blonde-haired and of normal At the time of operation, blood was obtained female build (see Fig. 1). Pubic and axillary hair from the gonadal venous plexus for the estimation was absent, breast development normal and the of plasma testosterone levels. The latter level was external genitalia of female type, although the found to be some ten times higher than levels labia majora were underdeveloped. The vagina found in the peripheral plasma. The half-life of was short (2 cm) and blind ending. testosterone in the plasma was determined and found to be normal. The secretion rates of Investigations dehydroepiandrosterone and its sulphate were Haemoglobin 12 g / 100 ml; chest X-ray normal; found to be of male and not female pattern. Full serum cholesterol 235 mg/ 100 ml; urinary 17- details of these investigations, together with the Postgrad Med J: first published as 10.1136/pgmj.43.496.117 on 1 February 1967. Downloaded from 118 Case reports ...... by copyright. FIG. 1 results from the four other cases of the syndrome apex of the space. Haemostasis was obtained and mentioned above, have been published elsewhere the cavity sprayed with 'Polybactrin' antibiotic (Deshpande et al., 1965). spray. The graft-covered mould was inserted into the space, the labia sutured over the mould and http://pmj.bmj.com/ Operation an in-dwelling catheter inserted. After induction of anaesthesia, a split-skin graft The patient was now positioned for laparotomy. was taken from the patient's right thigh. This graft The abdomen was opened through a Pfannenstiel was subsequently sutured to itself, raw surface incision and, on entering the peritoneal cavity, the outwards, over an acrylic resin mould, 15 cm in site of the vaginal mould was seen producing a length. A solid mould was chosen in this particular surface elevation of the peritoneum between instance rather than one constructed of rolled bladder and rectum. The absence of the uterus plastic sponge (the technique usually employed) and Fallopian tubes was confirmed, the gonad on on October 2, 2021 by guest. Protected because this was thought safer, for the vault of the the left side lying just caudal to the normal ovarian new vagina would be in apposition to the pelvic position and the gonad on the right lying at the peritoneum; there was thus a possible danger of level of the internal inguinal ring. Blood was escape of fragments of sponge into the peritoneal obtained from the venous plexus for testosterone cavity if the graft failed and the peritoneal layer estimation, bilateral gonadectomy performed and broke down. After the donor site had been the abdomen closed in layers. dressed, the patient was placed in the lithotomy Post-operatively, the patient was given prophy- position. lactic antibiotic cover for 14 days, bladder drain- The apex of the blind-ending vagina was incised age being continued for a similar period of time. transversely and the space between bladder and There was a rise in temperature on the 3rd post- rectum separated widely in both longitudinal and operative day, associated with slight purulent loss transverse directions. At the completion of the from the centre of the vaginal mould. The tem- dissection, the peritoneum could be seen at the perature soon subsided and, apart from bowel Postgrad Med J: first published as 10.1136/pgmj.43.496.117 on 1 February 1967. Downloaded from -Case reports 119 distension, the patient remained well after opera- the evidence McMillan concludes that they are tion. On the 14th day, she was taken to the operat- Fallopian in origin. ing theatre where the donor site on the right thigh Discussion was inspected and re-dressed. Examination of the The clinical features of testicular feminization vagina showed that almost 90% of the donor skin have been described in detail by Morris (1953) and had taken, there being a small area of granulation by Hauser et al. (1957). The mechanism of the tissue at the vault on the right side. The cavity was defect which leads to the condition is uncertain, irrigated with saline and the mould re-inserted although the inherited pattern suggests a single without labial suture. Catheter drainage was dis- gene defect and consequently an enzyme defect continued at this time. (Polani, 1962). It had earlier been suggested by The patient was discharged from hospital 4 Morris that the oestrogen might be different from weeks after operation with the mould still in posi- normal in view of the unusual tissue response; tion. She was seen weekly in the out-patient de- alternatively there might be target organ resistance partment, the mould being removed at each visit to normal circulating oestrogen. and the vagina inspected. The vault granulations Current work (Deshpande et al., 1965), how- were cauterized and, after 3 months, the use of ever, shows that hormone production follows the the acrylic mould was discontinued, the patient normal male pattern. Despite this, the testicular being given a size 6 glass dilator to use daily. sex cells fail to develop normally and the Wolffian Stilboestrol 0 5 mg was given daily for 6 months. system is amost completely suppressed. Miillerian development is inhibited to some extent. Whether Follow-up these effects are due to an anti-androgen or to The patient subsequently married and was seen some unusual oestrogen produced under genetic for follow-up 6 months after marriage and 12 direction remains to be discovered. months after discharge from hospital. Vaginal Vaginal adequacy varies from case to case and examination showed the artificial vagina to be of there have been few reports of surgical correction. by copyright. normal length and healthy. Intercourse was satis- Bulska, Teter & Ruszkowski (1960) reported on factory and orgasm was achieved. the successful management of one such case. The technique of operation in our case has followed Histological report (Dr M. 0. Skelton) the standard Mclndoe-Read procedure, the latter 'The right testis measures 3 X 2 cm. At its medial having been recently described and reviewed by pole is a firm mass of pinkish-yellow tissue having Jackson (1965). It appears clear from the experi- the macroscopic appearance of muscle.
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