[Case of infantile arrhenoblastoma and possible misdiagnosis of pseudohermaphrodism for arrhenoblastoma].
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Diagnosis, Differential↗
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[A case of arrhenoblastoma of the ovary (arrhenoblastoma trabeculare ovarii)].
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Adult↗
Arrhenoblastoma during pregnancy.
Arrhenoblastoma is a rare complication of pregnancy. A case of fatal nonvirilizing arrhenoblastoma in a pregnant adolescent, the third such case to appear in the literature, is discussed. Fifteen cases of arrhenoblastoma in pregnancy that have appeared in the recent literature are reviewed; 87% of these tumors were virilizing. When virilization is absent the diagnosis is difficult. Among the more common presenting symptoms are weight loss, nausea and vomiting, abdominal pain, and a palpable abdominal mass. Arrhenoblastoma occurring during pregnancy has a 31% maternal mortality, 44% rate of malignancy, and a 50% perinatal mortality.
Adolescent↗
A case of malignant arrhenoblastoma.
The patient was a 72-year-old married woman from whom a recurrent arrhenoblastoma had been removed three time in the course of six years. She was clearly virilized and her voice had become deep. Endocrine studies before removal of the recurrences showed pronounced elevation of the serum testosterone values. The 17-KS values did not as clearly parallel growth of the tumour. Histochemical examination showed moderate 3beta-steroid dehydrogenase activity in limited areas. The histological features were those of so-called intermediate type of arrhenoblastoma with a very scanty tubular component. Histologically the tumour was malignant, but with a relatively low degree of malignancy. Regardless of the treatment there was twice a recurrence of the tumour. The increase of the serum testosterone values during the follow-up period reflected recurrence on of the tumour.
Abdominal Neoplasms↗
[Arrhenoblastoma with following pregnancy].
The emergence, the morphology, the characteristic symptoms, the treatment and prognosis of a rare tumour of the ovary, arrhenoblastoma, is dealt with in this article. This is followed by the writer's own experience of the case of a 24 year old patient who had a normal pregnancy and birth 2 years after the existirpation of a tumour on the left side, which, having been detected by change, proved to be a non-malignant arrhenoblastoma. The symptoms of virilism so typical of the tumour had disappeared. The normal female characteristics had returned. The patient has now been free of symptoms for 10 years. In cases of increasing virilism differential diagnostics should consider the possibility of this rare tumour.
Adolescent↗
The hormone pattern in a case of arrhenoblastoma.
A case of ovarian arrhenoblastoma in a 14-year-old girl is reported. The patient presented with primary amenorrhea, severe diffuse hirsutism, moderate clitorial enlargement and slight decrease in breast size. Hormonal examinations revealed high plasma testosterone and androstenedione levels, normal plasma prolactin, drhydroepiandrosterone-sulphate, 17-alpha-hydroxyprogesterone, urinary 17 beta oestradiol, oestrone, FSH and LH. Androgen concentrations decreased under dexamethasone suppression test. Following tumor ablation menses occurred spontaneously and normal hormone patterns were observed.
17-Ketosteroids↗
Hirsutism caused by an androgen-producing ovarian tumor. A case of arrhenoblastoma.
A 34-year-old woman with excessive facial hirsutism and a 18-month history of amenorrhea was found to have an ovarian arrhenoblastoma of the intermediate type. The endocrine profile was determined before, during and after surgery. Determination of hormone levels indicated that, although both the delta 4 and delta 5 pathways were involved, the delta 4 pathway was probably predominant in androgen biosynthesis. Four weeks after removal of the tumor, the menstrual cycle was normalized and has subsequently remained regular. Six yr after operation, the patient had lost her hirsutism and neither radiological, palpatory nor endocrine signs of either recurrence or metastasis have been observed.
Adult↗
Clinical features and hormonal characteristics in a case of ovarian arrhenoblastoma.
We report a case of a 34-year-old woman affected with ovarian arrhenoblastoma characterized by very high testosterone (T) levels (34.0-60.0 ng/ml; n.v.0.2-0.9) and suppressed gonadotropin levels. The physical examination revealed: severe hirsutism, acne, amenorrhea and other virilization signs. Basal hormonal evaluation also showed a markedly elevated 17-hydroxyprogesterone (17-OHP) and a mild delta 4 Androstenedione (A) and dehydroepiandrosterone sulfate (DHEAs) increase. ACTH test induced only slight changes in androgen secretion. By contrast, dexamethasone test greatly decreased A and DHEAs whereas T levels were only partially suppressed. Moreover, hCG test was clearly stimulatory for T and A. Suppressed gonadotropin levels did not respond to LHRH stimulation. The removal of the neoplasia was followed by normalization of T levels and increase of serum gonadotropins with subsequent restoration of a normal responsiveness to LHRH and resumption of an ovulatory menstrual cycle. This observation suggests that the high T levels played a primary role in the pathogenesis of the gonadotropin suppression and anovulation. Recovery of acne was complete whereas hirsutism score was reduced but still elevated after one year. This may be due to postoperative A and DHEAs levels slightly above the normal range, indicating the presence of adrenal hyperandrogenism.
17-alpha-Hydroxyprogesterone↗
Familial thyroid nodulation and arrhenoblastoma.
A patient who had thyroid nodules and a functioning ovarian arrhenoblastoma had a family history of thyroid nodulation affecting both males and females in four consecutive generations. One relative had multiple follicular adenomas of the thyroid. The findings support the proposal that this is another genetically determined endocrine organ tumor complex.
Adenoma↗
Testosterone secretion by cultured arrhenoblastoma cells: suppression by a luteinizing hormone-releasing hormone agonist.
A patient with virilization was studied. The basal urinary excretion of 17-ketosteroids was at the upper limit of normal, but the plasma testosterone concentration was greatly elevated. Testosterone secretion could be stimulated by hCG, suppressed by dexamethasone, and was not affected by ACTH. At operation, an arrhenoblastoma of the left ovary was found. Isolated tumor cells in culture secreted testosterone. The addition of a LRH agonist (10 ng/ml) suppressed the secretion of testosterone by 50% (P less than 0.01). The inhibiting effect of a LRH agonist on steroidogenesis suggests that LRH receptors were present on this tumor and that treatment with LRH agonists might be beneficial in patients with metastatic steroid hormone-secreting ovarian and testicular tumors.
17-Ketosteroids↗
[Heterosexual precocious puberty due to arrhenoblastoma. Postsurgical spontaneous pregnancy].
It is presented the case of a female with heterosexual precocious puberty associated to hyperandrogenism and virilization due to arrhenoblastoma, who became pregnant after surgery. Clinical study: a 14-year-old female initiated at age 6 years with premature pubarche and telarche. By age 11, the patient only had one menstrual period along with virilization. Physical exam disclosed: facial acne, cricoid enlargement, breast Tanner II, pubic hair Tanner III, clitoromegaly of 4.5 cm and hypotrophy of labia majora. Ferriman and Gallwey: 12. Basal quantitation of circulating testosterone: 1.25 ng/mL (normal: 0.2 to 0.8 ng/mL), androstenedione 13.9 ng/mL (normal: 0.5 to 2.4 ng/mL). A pelvic ultrasonographic study showed: uterus of 66 x 25 x 30 mm, right ovary of 50 x 50 mm, hyperechoic with echolucid and nodular areas (vol. 65.3 cc). Left ovary was of 30 x 30 mm with echolucid areas of 2.0 mm (vol. 5.1 cc). Cariotype: 46XX. Surgical right oophorectomy was performed containing an ovoid tumor of 4 x 3 x 3 cm. Histologic analysis revealed a Sertoli Leydig (Type II, Mayoer) cell tumor. The patient resumed menses and became pregnant at age 20 (22 week obitus). In the last assessment, performed 6 years after surgery, the patient resumed normal menses, Ferriman and Gallwey 5, Tanner V and regression of clitoromegaly to 3.0 cm. In this case, surgery provided remission of hyperandrogenism, normal menstrual cycles and fertility restoration.
Adult↗
Arrhenoblastoma in a mare.
An ovarian neoplasm measuring 10 by 8 by 6 cm was surgically removed from a 14-year-old Appaloosa mare. For 2 years prior to surgery, the mare had manifested marked behavioral changes, becoming aggressive toward other broodmares. Histologically, the tumor was found to be an arrhenoblastoma. Preoperative endocrinologic findings (high serum testosterone and low serum estradiol concentrations) supported the diagnosis.
Aggression↗
[Another case of Sertoli-Leydig cell ovarian tumor (arrhenoblastoma). Review of the literature].
In reporting another case of Sertoli-Leydig cell tumor (SLCT) treated by conservative management, the principal characteristics of these tumors are reviewed and a general review is made: SLCT (arrhenoblastomas) are rare endocrine tumors (1 p. cent of tumors of the ovary). They most frequently occur in young women, are unilateral in 98 p cent of cases, and are manifest after a long period by endocrine signs (75 p. cent of cases) whose most frequent combination is: amenorrhea-hirsutism-hoarse voice. They are classified in four histological types which have different prognoses. Their potential development varies and is variously assessed: these are tumors with a low incidence of malignancy (mean 18 p. cent). Under certain conditions, this permits the institution of conservative therapy conserving future fertility. Malignant types of tumors are treated in the same way as other malignant ovarian tumors: surgery is the main tool; radiotherapy is often insufficient and chemotherapy often provides remission more frequently than cure (80 p. cent 5 year survival).
Adult↗
[Morphological and endocrinological study of ovarian arrhenoblastoma (author's transl)].
Morphological and endocrinological studies were performed on a 19-year-old case of an arrhenoblastoma with marked virilization. The tumor was an intermediate type of Meyer's classification. Histochemically, 3 beta-HSD and G-6-PDH activities were demonstrated in Leydig cells. These cells also had ultrastructures typical of steroid-producing cells. Basal blood cells of pregnenolone, dehydroepiandrosterone, testosterone (T), androstenedione (A), progesterone, estradiol, estrone, LH, and FSH were determined pre- and postoperatively. T and A showed a very high level preoperatively, and were markedly decreased immediately after removal of the tumor. Stimulation of tumor cells by HMG-HCG did not show any significant changes in their main products of T and A. These findings suggest that Leydig cells of the present tumor produced mainly A and T independently on gonadotropins, and these hormones had virilized the patient.
Adult↗
Mixed tumor: mucinous papillary cystadenocarcinoma associated with arrhenoblastoma.
A case of a 41-year old woman with an enlargement of the inferior part of the abdomen is presented. An echographic examination established the diagnosis of a right ovarian cyst. During the operation, a 14-cm (5 1/2") tumor, presenting a large ruptured cyst and solid mass, was found. From the cystic activity, a viscous fluid was oozing. On the left ovary, a 4 cm (1 1/2") cyst was also found. Within this case report, we underline the unusual association of a mucinous papillary cystadenocarcinoma with an arrhenoblastoma in the right ovary together with the presence of an cystadenoma in the left ovary.
Adult↗
Arrhenoblastoma; the incidence of malignancy and the relationship to pregnancy, to sterility, and to treatment.
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Female↗