Carcinosarcoma arising from atypical endometriosis 435

A case of Sertoli–Leydig cell tumor in

a postmenopausal woman
A. CARINGELLA*, V. LOIZZI*, L. RESTAy, R. FERRERI* & G. LOVERRO*
Departments of *Obstetrics and Gynecology and yPathological Anatomy and Genetics, University of Bari,
Piazza Giulio Cesare, Bari, Italy

Abstract. Caringella A, Loizzi V, Resta L, Ferreri R, Loverro G. A case of Sertoli–Leydig cell tumor in
a postmenopausal woman. Int J Gynecol Cancer 2006;16:435–438.

Sertoli–Leydig cell tumor belongs to the group of sex cord–stromal tumors of the ovary. These neoplasms
account for less than 0.5% of all ovarian tumors and are more often encountered in young women between
the ages of 20 and 30 years who usually become virilized. We described an unusual case of Sertoli–Leydig

Address correspondence and reprint requests to: Vera Loizzi, MD,

Viale J.F. Kennedy 80, 70124 Bari, Italy. Email: vloizzi@tiscali.it

# 2006 IGCS, International Journal of Gynecological Cancer 16, 423–447

436 A. Caringella et al.

cell tumor in a postmenopausal women who presented with a solid right pelvic mass, a large amount of
ascites, and laboratory tests revealing an elevated CA125, all suggesting a pelvic malignancy. Although five
similar cases of postmenopausal women with Sertoli–Leydig cell tumor of ovary have been reported in the
literature, we believe that this is an useful addition to the literature.

KEYWORDS: postmenopause, Sertoli–Leydig cell tumor.

Sertoli–Leydig cell tumor belongs to the group of sex

cord–stromal tumors of the ovary. The terms of arrhe-
noblastoma, androblastoma, or gonadal stromal tumor
of android type have all been used as synonyms for
this cancer. They account for less than 0.5% of all ovar-
ian tumors and are usually unilateral(1). The clinical
characteristics are related to the degree of histologic
differentiation(2). Age at diagnosis averages 25 years,
with 70–75% of these tumors occurring during the sec-
ond and third decade of life and less than 10% occur-
ring either prior to menarche or after menopause(3).
When the tumor occurs in young women, it usually,
but not necessarily, becomes virilized(4). The occur-
rence in women older than 65 years is very rare, and it
has been reported in only five cases of the current liter-
ature(5). However, some of these cases have reported
postmenopausal women who presented signs of virili-
zation such as increasing facial hair growth, deepening Figure 1. Axial computed tomography examination of the pelvic mass.
of the voice, a dull pain in the lower part of the abdo-
men, and enlargement of the clitoris. We presented an a Sertoli–Leydig cell tumor of the right ovary, with low
additional case of a Sertoli–Leydig cell tumor in a post- degree of differentiation confirmed on permanent sec-
menopausal woman with no signs of virilization. tion (Figs. 2, 3). No areas of malignant transformation
were identified on multiple sections of the surgical
specimen. Because of the absence of any sign of viriliza-
Case report tion, the patient did not undergo any hormonal analy-
sis. One week after surgery, the serum levels of CA125
A 65-year-old multiparous woman presented to our
attention with complaints of diffuse abdominal pain
and abdominal distension. On physical examination,
the abdomen was distended with a fluid wave and
dullness to percussion consistent with significant accu-
mulation of ascites.
A computed tomography scan of the abdomen and
the pelvis revealed a 19- 3 15- 3 7-cm complex cystic
and solid right pelvic mass with a large amount of
ascites (Fig. 1). Laboratory tests revealed an elevated
CA125 of 119 U/mL of serum. A chest X-ray showed
no evidence of pleural effusion or lung metastasis. A
laparotomy was performed with findings of 9 L of
straw-colored, slightly turbid ascitic fluid; a mobile
complex right ovarian mass; and no evidence of intra-
peritoneal or retroperitoneal disease. A total abdominal
hysterectomy with bilateral oophorectomy, peritoneal Figure 2. Low-power view of Sertoli–Leydig tumor. The neoplasm is
cytology, omentectomy, appendicectomy, and perito- composed of undifferentiated long cells mixed to areas containing
neal biopsies was performed. Frozen section revealed cords (hematoxylin and eosin stain, original magnification 320).

# 2006 IGCS, International Journal of Gynecological Cancer 16, 423–447

 A Sertoli–Leydig cell tumor 437

Clinically, patients with Sertoli–Leydig cell tumors

may suffer loss of female secondary sex characteristics,
with atrophy of the breast and disappearance of
female body contours. Later on, progressive masculin-
ization supervenes, including acne, temporal balding,
deepening of the voice, and enlargement of the clitoris.
These symptoms are as a result of androgen excess.
Clinically obvious virilization usually goes along with
elevation of serum testosterone levels. However, less
than 50% of the patients with Sertoli–Leydig cell
tumors have no endocrine manifestations but only
present with abdominal swelling or pain(2,5).
Because these tumors occur predominantly in
young women and are bilateral in less than 5% of
cases, conservative removal of the tumor and adjacent
Figure 3. High-power view of Sertoli–Leydig tumor revealing long fallopian tube is justifiable, if preservation of fertility is
cells with marked dark nuclei and cords that resemble that of sex an important consideration and if there is no evidence
cords from the primitive gonad (hematoxylin and eosin stain, origi- of extension beyond the involved ovary. In our case,
nal magnification 3100).
the patient was a 65-year-old woman who presented
with a solid right pelvic mass, a large amount of asci-
had fallen to normal, and there was no reaccumulation
tes, and laboratory tests revealing an elevated CA125,
of ascites. The patient was started on follow-up visits
all suggesting a pelvic malignancy. We performed
every 3 months for the first 2 years and was found to
a total abdominal hysterectomy with bilateral oopho-
be with no evidence of disease. A review of the litera-
rectomy, peritoneal cytology, omentectomy, appendicec-
ture on Sertoli–Leydig cell tumor in postmenopausal
tomy, and peritoneal biopsies. Histologic examination
women is provided in Table 1.
revealed a Sertoli–Leydig cell tumor of ovary, with
low degree of differentiation and no areas of malig-
Discussion
nant transformation on the other sections. In view of
Sex cord–stromal tumors of the ovary are uncommon the low-grade risk of the tumor, we decided to start
ovarian malignancies, which account for less than 8% the patient on follow-up visits every 3 months for the
of all ovarian tumors(6,7). Accounting for less than 1% first 2 years, and she has been free of disease until the
of ovarian neoplasms(1), Sertoli–Leydig cell tumors are current report.
rare sex cord–stromal neoplasms that contain Sertoli No sign of virilization was observed in our patient; this
and/or Leydig cells in varying proportions and vary- should be interesting since some reports on postmeno-
ing degrees of differentiation. They occur in young pausal women with this tumor have showed clinical pro-
women between the ages of 20 and 30 years and usu- gressive masculinization with the presence of increasing
ally but not necessarily become virilized. Bilaterality is facial hair growth, deepening of the voice, a dull pain in
exceptional, with only 3 out of 207 cases reviewed by the lower part of the abdomen, and enlargement of the
Young and Scally in 1985(8). Ninety-seven percent of clitoris(9,10). We believe that these findings in post-
the tumors were found to be stage I, whereas only 3% menopausal woman could be the result of peripheral
of them had spread beyond the ovary, usually within androgen aromatization(10). Our case was an unusual
the pelvis, and rarely into the upper abdomen(2). presentation of Sertoli–Leydig cell tumor associated
with a pelvic mass, a large amount of ascites, and labo-
ratory tests revealing an elevated CA125 that is not typ-
Table 1. Review of the literature on Sertoli–Leydig cell tumor ically seen in this tumor. In fact, the areas with tumor
in postmenopausal women Sertoli cells are usually negative for epithelial mem-
Age Sign of brane antigen, carcinoembryonic antigen, CA19.9,
Authors Year (years) virilization CA125, or S-100 protein(10). We believe that the eleva-
Gheorghisan-Galateanu et al.(10) 2003 69 Yes tion of CA125 could have been secondary to the pres-
Hansen and Sorensen(9) 1993 65 Yes ence of ascites; however, its level was much lower than
Tampakoudis et al.(11) 2004 71 No that typically seen with ascites of malignant origin.
Dhont et al.(5) 1986 73 No There are no solid data to suggest that adjuvant
Clinton et al.(12) 1981 65 No
therapy has any value in preventing a recurrence in
# 2006 IGCS, International Journal of Gynecological Cancer 16, 423–447
438 A. Caringella et al.
patients with stage I lesions. Although five similar
cases of postmenopausal women with Sertoli–Leydig
cell tumor of the ovary have been reported in the
current literature(5), we believe that this is an useful
addition to the literature.
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Accepted for publication August 3, 2004