European Journal of Obstetrics & Gynecology

and Reproductive Biology 54 (1994) 137-139

Case reports

Cystadenofibroma of the ovary in young women

Asnat Groutz* a, Igal Wolmana, Yoram Wolf”, Dov Luxmana, Joseph Sagi”, Ariel J. Jaffab,
Menachem Peter David a
‘Department of Obstetrics and Gynecology ‘B’, hDepartment of Obstetrics and Gynecology ‘A’, Serlin Maternity Hospital, Tel-Aviv Medical
Center, Sackler Faculty of Medicine. Tel-Aviv University, Tel-Aviv, Israel

(Accepted 9 December 1993)

Abstract

Cystadenofibroma of the ovary, a relatively rare benign tumor, usually appears during the fourth and fifth decades. This tumor
has malignant ultrasonographic features and may also macroscopically appear as malignant during surgery. Since this tumor has
a rare malignant potential, it is well-advised to be aware of the possibility of a cystadenofibroma before selecting an aggressive
surgical approach in young patients. We present five cases of young patients with cystadenofibromas of the ovary.

Key words: Cystadenofibromas; Young patients

1. Introduction cycle) were regular. No oral contraceptives were used.

Ultrasonographic evaluation revealed a non-septate
Adenofibroma of the ovary is a relatively rare benign left ovarian cyst (12 cm in diameter), filled with clear
tumor which originates from the germinal lining and the content with papillary formation within the cyst. Chest
stroma of the ovary. This tumor may be solid, semisolid and abdominal X-rays were unremarkable, as well as in-
or cystic, depending upon the relative amount of the travenous pyelogram. Blood chemistry parameters and
epithelial and stromal constituent and upon the secre- markers (CA-125 CEA, @-hCG, a-fetoprotein) were all
tory activity of the epithelial component [I]. Since within the normal range.
adenofibroma may present as a malignant tumor on a At explorative laparotomy, a left ovarian cyst con-
preoperative ultrasonographic examination, as well as taining clear fluid and some papillary structures on its
macroscopically during surgery, it causes a diagnostic base was found. The uterus, right ovary and both fallo-
and operative problem when treating young patients. pian tubes were all normal. A left oophorectomy and a
We present five cases of young patients with cyst right ovarian wedge resection were performed.
adenofibroma of the ovary. Pathological examination revealed an ovarian serous
papillary cystadenofibroma of the left ovary. Cytologic
2. The five cases evaluation of the cystic fluid was normal. The wedge
resection showed normal ovarian tissue.
2.1. Case 1
2.2. Case 2
A lPyear-old single white nulligravida was referred
to the Serlin Maternity Hospital because of a left ovari- A 21-year-old single white nulligravida was referred
an cyst which was found on a routine gynecological to the our center because of a finding of a right ovarian
examination. Her past medical history was unremark- cyst. Her past medical history revealed 2 years’ ingestion
able. The patient’s menses (Cday duration, 30-day of oral contraceptives. The patient’s menses (Cday

* Corresponding author, Serlin Maternity Hospital, P.O. Box 7079, 61070 Tel-Aviv, Israel

0028-2243/94/$07.00 0 1994 Elsevier Science Ireland Ltd. All rights reserved.

SSDI 0028-2243(93)01792-R
138
duration, 28-day cycle) were regular. Ultrasonographic
examination revealed a right ovarian cyst (7 cm in di-
ameter), containing clear fluid, non-septate, with one
papillary formation.
Chest and abdominal X-rays were unremarkable.
Preoperative intravenous pyelogram (IVP) was negative.
CA-125 CEA, a-fetoprotein and &hCG were negative
as well.
Exploratory laparotomy revealed a right ovarian cyst
(7 cm in diameter) tilled with clear fluid with a small
papillary formation in its base. The uterus, left ovary
and both fallopian tubes were normal.
The cystic formation was conservatively removed and
a wedge resection of the contralateral ovary was per-
formed. Pathological examination of the cystic forma-
tion revealed ovarian cystadenolibroma. The wedge
resection showed normal ovarian tissue. Cytologic
examination of the fluid within the cyst revealed no ma-
lignant cells.
2.3. Case 3
A 20-year-old single white nulligravida was referred
to our center because of abdominal pain and the
discovery of a left ovarian cyst.
Her past medical history was unremarkable.
Gynecological history revealed regular menses (Cday
duration, 28-day cycle), and a l-year use of oral con-
traceptives.
Ultrasonographic examination revealed a left ovarian
cyst (8 cm in diameter) with clear content, non-septate,
with two papillary formations within.
Chest and abdominal X-rays and a preoperative intra-
venous pyelogram were all unremarkable. Blood chem-
istry parameters and blood markers such as CA-125,
CEA, ar-fetoprotein and &hCG were all within the nor-
mal range.
At laparotomy a left ovarian cyst 8 cm in diameter
containing multiple papillary formations inside and out-
side the capsule was found. The uterus, the right ovary
and both tubes were normal. A left conservative cystec-
tomy and a right wedge resection were performed.
Pathological examination of the cystic formation
revealed a serous papillary cystadenofibroma. The
wedge resection showed normal ovarian tissue. Cyto-
logic examination of the cyst was normal.
2.4. Case 4
A 20-year-old single white nulligravida was referred
to our center because of a finding of an ovarian cyst dur-
ing routine gynecological examination.
Her past medical history was unremarkable. Gyne-
cological history revealed regular menses (Cday dura-
tion, 28-day cycle), and 3-years’ use of oral con-
traceptives.
A. Grout- et al. /Eur. J. Obstet. Gynecol. Reprod. Biol. 54 (1994) 137-139
Ultrasonographic examination revealed a left ovarian
cyst, (6 cm in diameter). The cyst was not septated, it
contained clear fluid, with several papillary formations.
Chest and abdominal X-rays and a preoperative intra-
venous pyelogram were all unremarkable. Blood chem-
istry parameters and blood markers such as CA-125
CEA, a-fetoprotein and &hCG were all within the nor-
mal range.
At explorative laparotomy, a left ovarian cyst (6 cm
in diameter) containing clear fluid and some papillary
formations on its base was found. The uterus, right
ovary and both fallopian tubes were all normal. A left
conservative cystectomy and a right ovarian wedge
resection were performed.
Pathological examination of the cystic formation
revealed a serous papillary cystadenofibroma. The
wedge resection showed normal ovarian tissue. Cyto-
logic examination of the cyst was normal.
2.5. Case 5
A 19-year-old single white nulligravida was referred
to our center because of irregular bleeding and a finding
of a right ovarian cyst.
Her past medical history was unremarkable. Gyne-
cological history revealed regular menses (Cday dura-
tion, 28-day cycle) until 3 months prior to the hospital-
ization.
Ultrasonographic examination revealed a right non-
septate ovarian cyst (9 cm in diameter) with clear con-
tent and one papillary formations within.
Chest and abdominal X-rays were unremarkable.
Preoperative intravenous pyelogram (IVP) was negative.
CA125 CEA, a-fetoprotein and @hCG were negative
as well.
Exploratory laparotomy revealed a right ovarian cyst
(9 cm in diameter) tilled with clear fluid with one
papillary formation in its base. The uterus, left ovary
and both fallopian tubes were normal.
The cystic formation was conservatively removed and
a wedge resection of the contralateral ovary was per-
formed. Pathological examination of the cystic forma-
tion revealed ovarian cystadenolibroma. The wedge
resection showed normal ovarian tissue. Cytologic
examination of the fluid within the cyst revealed no ma-
lignant cells.
3. Discussion
The term adenolibroma of the ovary was first
employed to describe a papillary cystadenoma contain-
ing short, wide papilla mainly composed of dense con-
nective tissue covered by a layer of epithelial cells. Later
it was suggested that adenotibromas were benign com-
pound tumors composed of intermingling neoplastic
epithelial cells and fibroblasts of varying proportions
A. Croutz et al. /Eur. J. Obstet. Gynecol. Reprod. Biol. 54 (1994) 137-139 139

[l]. Although the majority of the reported adeno- come in all patients, unrelated to whether they under-
fibromas are of the serous type they may also be of the went conservative cystectomy, oophorectomy or total
endometrioid, clear cell or mutinous types [ 1,2]. abdominal hysterectomy and bilateral salpingoophorec-
Serous cystadenofibromas generally appear during tomy. In view of the fact that this tumor is reported to
the fourth and fifth decades. It seems that cystadeno- appear bilaterally in 6-30% of the cases [3,5,9], a con-
fibromas which contain endometrioid, clear cell or tralateral wedge resection is recommended.
mutinous epithelia tend to appear l-2 decades later Since cystadenofibroma of the ovary is a benign
[2-61. Cystadenofibromas were reported to appear in an tumor with a rare potential for malignant transforma-
earlier age in a particular subgroup of women with ante- tion, we believe that when treating young patients, con-
natal exposure to diethylsibestrol (DES) [7]. servative cystectomy and a contralateral ovarian wedge
Our live patients were not antenatally exposed to resection is a safe approach. A frozen section pathology
DES and were all very young (19-2 1 years old). at surgery may be very helpful in many of these cases.
This tumor may present with symptoms such as pain, In view of the fact that this tumor has malignant
vaginal bleeding, increased girth, dysuria, feminization ultrasonographic features and since it may appear as
and rectal urgency [4-81. In three of our patients the malignant during surgery (due to its numerous papillary
tumor was found on a routine examination, in one protrusions), it is well advised to be aware of the
patient it was found due to irregular bleeding and in one possibility of a benign tumor before an aggressive surgi-
it was found due to abdominal pain. Several authors cal approach is selected.
[9,10] suggest that vaginal bleeding may occur due to
tumoral excessive estrogen excretion which causes ab- 4. References
normal endometrial growth and subsequent bleeding. It
is thought that this excessive estrogen excretion may I Wolfe SA, Seckinger DL. Varied anatomical types of ovarian
also cause feminization. Others [5,8,9] could not demon- adenofibroma. Am J Obstet Gynecol 1967; 99: 121-125.
2 Bell DA. Mutinous adenofibromas of the ovary. A report of IO
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4 Compton HL, Fich FM. Serous adenolibroma and
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Macroscopically this tumor appears encapsulated,
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women with antenatal exposure to diethylsilbestrol. Gynecol
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of age or postmenopausal [4,6,8,9,12]. The malignant adenolibromatous tumors. Cancer 1984; 53: ll56- 1163.
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and are believed to develop in the preexisting benign 10 McNulty JR. The ovarian serous cystadenolibroma, a report of
tumors [ 111. No malignant changes were found in our 25 cases. Am J Obstet Gynecol 1959; 77: 1338-1347.
patients. II Saffos RO, Benrubi Gl, Rathigan RM et al. Coexisting adeno-
There is no doubt that when confronted with a patient carcinoma and cystadenolibroma (malignant cystadenolibroma)
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12 Moore DH, Fowler WC, Santrach PJ. Malignant endometrioid
is no need for a conservative approach, even if the tumor cystadenof ibroma of the ovary. Arch Gynecol Obstet 1991; 249:
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and the need for conservative treatment arises. Czer- dometrioid adenofibroma with omental implants and collision
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440-444.
cystadenofibroma and found the same favorable out-