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Case Report ISSN: 2090-5351
Journal of Case Reports in Medicine
J Case Rep Med, 2019 doi: 10.15761/JCRM.1000127 Volume 8(1): 1-5
Large Solitary Luteinized Follicular Cyst of Pregnancy and Puerperium in Ruptured Ectopic
Pregnancy Managed Laparoscopically
Vimee Bindra*, Frsha Nozer Taraporewalla, Aditya D Kulkarni, Neha Agrawal
Department of Obstetrics and Gynecology (Drs. Bindra and Taraporewalla) and Department of Pathology (Drs. Agrawal and Kulkarni),
Apollo Hospitals, Hyderabad, India
Abstract
Background: Ovarian cysts in pregnancy are encountered in approximately 1 in 100 cases. Of these, most are benign. One such
non-neoplastic lesion is Large Solitary Luteinized Follicular Cyst of Pregnancy and Puerperium (LSLFCPP). ey are a rare le-
sion with unknown pathogenesis. ey grow rapidly to very large sizes, causing symptoms. ey are hormonally inactive. Maternal
and fetal outcome is usually unaected.
Objective: We present a case of a Large Solitary Luteinized Follicular Cyst of Pregnancy and Puerperium encountered inciden-
tally during the management of ruptured ectopic pregnancy. A 25 year old G2A1 with 7 weeks 3 days gestational age by dates
presented to the emergency department with the chief complaints of sudden onset of pain in the lower abdomen accompanied by
nausea and giddiness since 12 hours. What sets our case apart from rest in literature is it’s diagnosis very early in pregnancy. Also,
it is, to our knowledge, the only case reported in literature, in an ectopic pregnancy.
Conclusion: LSLFCPP is a rare, yet important entity to be kept in mind when evaluating patients with large ovarian cysts in preg-
nancy. Comprehensive management is the need of the hour, in order to ensure ideal maternal and fetal wellbeing.
no other relevant surgical or medical history. Family history was un-
remarkable.
On examination, she was conscious, coherent and oriented. Patient
was pale, with tachycardia (PR-114/min) and was maintaining nor-
mal blood pressure and saturations. Respiratory and cardiovascular
systems were normal. On abdominal examination, she had diuse
tenderness, guarding and rigidity. On bimanual examination, there
was cervical motion tenderness, forniceal fullness with a closed exter-
nal cervical os and no bleeding per vaginum.
An emergency ultrasound done revealed a bulky uterus with a het-
erogenous collection in the pelvis posterior to the uterus, suggestive
of hemorrhage due to a suspected ruptured ectopic. Bilateral ovaries
were not visualized due to hemoperitoneum. Multiple cystic lesions
were noted in right adnexa - suggestive of ?hydrosalpinx. Preoperative
Hemoglobin was 5.5 gm/dl. Coagulation prole, serum electrolytes,
Correspondence to: Vimee Bindra, Department of Obstetrics and Gy-
necology, Apollo Hospitals, Hyderabad, India, E-mail: vimee.bin-
dra@gmail.com
Keywords: Ectopic pregnancy; Ovarian lesion; Non-neoplastic; Rapid
growth; Cystectomy; Large leutinised follicle, Ruptured ectopic, Laparos-
copy, Ovarian cyst
Received: January 26, 2019; Accepted: February 11, 2019; Pub-
lished: February 13, 2019
Introduction
Ovarian masses during pregnancy are not uncommon, with incidenc-
es ranging from 0.1% [1] to 4.5% [2], with an average incidence of
1% [3,4]. Majority of such cases are detected incidentally due to the
routine use of ultrasound in the early rst trimester. Majority of them
are follicular cysts, benign, small (<5 cm), asymptomatic and resolve
spontaneously by mid-second trimester [2]. Any rapid growth or per-
sistence beyond the second trimester is considered to be indicative of
malignancy. e overall rate of malignancy in ovarian masses detect-
ed in pregnancy is 4 % [3]. However, there are a few benign lesions
that mimic malignancy due to their rapid growth and large size. Here,
we present a rare case of Large Solitary Luteinized Follicular Cyst of
Pregnancy and Puerperium (LSLFCPP) that was discovered inciden-
tally during management of an ectopic pregnancy. ere are only a
handful of such cases published in literature, and pose a challenge due
to their similar clinical picture as with malignant ovarian neoplasms.
Case Summary
A 25 year old G2A1 with 7 weeks 3 days gestational age by dates pre-
sented to the emergency department with the chief complaints of
sudden onset of pain in the lower abdomen accompanied by nausea
and giddiness since 12 hours. She conceived spontaneously, her at-
home urine pregnancy test was positive aer missed period. Patient
did not have any prenatal or early antenatal ultrasound done. She had
J Case Rep Med, 2019 doi: 10.15761/JCRM.1000127 Volume 8(1): 2-5
Bindra V (2019) Large Solitary Luteinized Follicular Cyst of Pregnancy and Puerperium in Ruptured Ectopic Pregnancy Managed Laparoscopically
renal function tests and liver function tests were normal. Serum βhCG
was 4,999 mIU/ml, corresponding to 4-5 weeks gestational age.
Based on her history, clinical picture and ultrasound report, a diag-
nosis of ruptured ectopic pregnancy was made. Patient underwent
emergency laparoscopy.
On laparoscopy, she was found to have 1½ - 2 litres of haemoperito-
neum with clots (Figure 1).
Figure 1: Initial view of abdomen and pelvis showing hemoperito-
neum and clots
A le tubal ectopic in the process of tubal abortion was noted at the
ampullary end of the le fallopian tube, which was the source of
bleeding (Figure 2).
Figure 2: Le ampullary tubal pregnancy in the process of abortion
(source of bleeding)
Le ovary appeared healthy. A large, multilocular right ovarian cyst
was noted measuring 15x15x12 cm(approximately) containing clear
serous uid (Figure 3).
Figure 3: Large multiloculated right ovarian cyst
ere was no torsion (Figure 4) or areas of hemorrhage or necrosis.
Figure 4: Right ovarian pedicle with fallopian tube with no evidence
of torsion
Peritoneal uid was collected for cytology. orough abdominal la-
vage was done and clots were cleared. Le salpingectomy was done.
Right ovarian cystectomy was done, cyst was aspirated within endo-
bag without spillage into abdominal cavity. Cyst wall was retrieved
within intact endobag without any cavitary spill. Hemostasis was con-
rmed. Abdominal drain was kept and ports removed under vision.
To compensate for blood loss, 2 units of packed red cells were trans-
fused intraoperatively. Postoperatively, patient was managed in inten-
sive care and a further 1 unit of packed red cells and 2 units of fresh
frozen plasma were transfused.
Retrospectively, serum CA-125 was done, which was reported as 76
U/ml. Patient recovered well from surgery, and was discharged in a
stable condition 72 hours aer surgery.
e histopathology report of the ovarian cyst revealed brin deposi-
tion with multiple layers of leutinized theca cells - consistent with a
corpus luteal cyst (Figures 5 and 6).
J Case Rep Med, 2019 doi: 10.15761/JCRM.1000127 Volume 8(1): 3-5
Bindra V (2019) Large Solitary Luteinized Follicular Cyst of Pregnancy and Puerperium in Ruptured Ectopic Pregnancy Managed Laparoscopically
Figure 5: Microscopic examination of cyst wall showing luteinized
theca cells (H & E stain 400x magnication)
Figure 6: Microscopic examination showing multiple layers of lutein-
ized theca cells (H & E stain 400x magnication)
e ovarian cyst uid and the peritoneal uid also had no atypical
cells. Patient was asymptomatic on her follow up visits at 1 week and
at 3 months post-procedure.
Discussion and Conclusion
Ovarian masses are commonly seen in pregnancy. Functional cysts
are the most common pathology, followed by benign cystic terato-
mas, serous cystadenomas, paraovarian cysts, mucinous cystadeno-
mas and rarely malignant tumours [2]. e majority of adnexal mass-
es in pregnancy are small, <5 cm and resolve spontaneously [2]. Cysts
>5 cm also resolve in most cases but they are at high risk of torsion
and hemorrhage; and hence greater chances of surgical intervention
during pregnancy.
Large Solitary Luteinized Follicular Cysts of Pregnancy and Puerpe-
rium (LSLFCPP) are a rare form of solitary follicular cysts [5]. On
gross examination, they are large (range - 8 to 55 cm, average - 25 cm),
unilateral, unilocular, rapidly growing cysts with a smooth, external
surface. ey have thin cyst walls (up to 5 mm thick), and contain
clear, serosanguinous or mucinous uid. e inner cyst lining is also
smooth [7].
Histologically, the cyst is lined by one to several layers (usually 10)
of luteinized granulosa and theca cells, with no clear dierentiation
between the two. Nests of luteinized cells can also be seen embedded
in the brous cyst wall. Cytoplasm is vacuolated, eosinophilic to clear,
with characteristic focal marked nuclear pleomorphism and hyper-
chromasia (bizarre nuclei), and absent mitotic activity [5,7].
ey do not cause any maternal or fetal endocrinological changes,
and usually have no untoward eects on pregnancy [5], although
there have been reports of preterm delivery [2,8]. However, due to
their large size, they can become symptomatic, and necessitate surgi-
cal removal either antenatally, during cesarean or in the puerperium.
Although the pathogenesis of LSLFCPP is unclear, based on its as-
sociation with pregnancy, hCG is believed to play a role in its aetio-
pathogenesis - either due to increased tissue sensitivity to hCG or due
to high levels of hCG in the maternal circulation. However, in our case,
the serum hCG level was corresponding to the expected gestational
age, and was not overtly elevated. Additionally, the patient had no risk
factors for having higher tissue sensitivity to hCG (polycystic ovarian
syndrome, diabetes mellitus, ovulation induction) [6]. Case reports
on LSLFCPP diagnosed in the puerperium, when the hCG levels are
low, may point to an additional unknown etiology [9]. Clement and
Scully, who rst described LSLFCPP, postulated that the initial devel-
opment of these cysts may be stimulated by hCG during the antenatal
period, and rising pituitary gonadotropins (follicle stimulating hor-
mone / luteinizing hormone) in non-lactating women in the postpar-
tum period, may contribute to their further enlargement [2].
On ultrasound evaluation, they appear to be single, solitary cysts with
smooth, thin walls; containing homogeneous anechoic (serous) ma-
terial [2]. In our case however, the cyst was multilocular, in contrast
to the other cases published, where unilocular cysts were reported.
ere is no role of tumour markers (CA125, beta hCG, LDH, alpha
feto protein) in diagnosis or prognosis of LSLFCPP. But, they may
help in dierentiating it from malignant ovarian neoplasms like gran-
ulosa cell tumours [5,7].
Long term follow up of cases reviewed in literature show no recur-
rence or malignant potential [2,5,7,9].
We searched published literature for reported cases of such type of
cysts in early pregnancy. Of the published case reports, none were di-
agnosed so early in the rst trimester (as there are relatively lower lev-
els of hCG in early pregnancy) and none had as large a cyst as was in
our case. e largest reported cysts in rst and early second trimester
are 9.5 cm and 9.0 cm at 14 weeks in two case reports by Hadad, et al.
J Case Rep Med, 2019 doi: 10.15761/JCRM.1000127 Volume 8(1): 4-5
Bindra V (2019) Large Solitary Luteinized Follicular Cyst of Pregnancy and Puerperium in Ruptured Ectopic Pregnancy Managed Laparoscopically
and Fang, et al. [2]. Also, to our knowledge, ours was the only case in
literature which was diagnosed in a patient with an ectopic pregnancy.
With regards to the dierential diagnosis of this type of cyst, Hyper-
reactio leutealis (HL) is a rare condition seen with elevated levels of
hCG like in multiple pregnancies, gestational trophoblastic disease
and hydrops fetalis. It is seen in 25% of molar pregnancy and 10% of
choriocarcinoma[6]. ey are usually bilateral, solid ovarian masses
with sheets of luteinized granulosa and theca interna cells [7]. HL
was ruled out in our case as it was unilateral and hCG levels were not
that high as to explain its occurrence in early pregnancy.
Follicular and corpus luteum cysts of pregnancy are common, but
they rarely exceed 4-6 cm in size [8]. Histologically, there are focal
areas of stratied polygonal cells, and they stain positive for inhibin
and calretinin [7].
Based on ultrasound ndings, serous cystadenomas, and less com-
monly, mucinous cystadenomas are other likely dierential diagnoses
(unilocular / bilocular anechoic cysts with smooth walls and thin sep-
tae) [2]. However, they do not exhibit rapid growth like in LSLFCPP.
Histologic ndings also dier (distinctive architecture, cilia, uniform
single layer of cells lining the cysts) [7].
e most common malignant neoplasm that may be similar to LSLF-
CPP, and may lead to a diagnostic dilemma, is the rare cystic variant
of granulosa cell tumour [9]. On gross morphology, both appear sim-
ilar. Microscopically, lack of luteinization, presence of granulosa cells
(smaller, more uniform, no pleomorphism), grooved nuclei, Call-Ex-
ner bodies and mitotic activity dierentiate granulosa cell tumours
from LSLFCPP [7,9].
LSLFCPP is a rare, yet important entity to be kept in mind when eval-
uating patients with large ovarian cysts in pregnancy. ere can be
so many dilemmas when such a cyst is encountered - to determine
whether the cyst is benign or malignant, whether to manage conser-
vatively with close follow up or with surgery, and the timing of sur-
gery (antenatal, intracaesarian or postnatal), the extent of surgery
(cystectomy / oophorectomy / radical surgery), the eect of the cyst
/ surgery on the maternal and fetal well-being. e rst, and perhaps
most important step, when such a diagnosis is made should ideally be
thorough and empathetic counseling. It would be prudent to remem-
ber that not all large multiloculated cysts are malignant, and not all
cysts require urgent surgery. Although ultrasound can help us posi-
tively identify the nature of the cyst in most cases [4], patients should
be closely monitored and periodically followed up. e patient must
be counseled about potential risks (obstetric complications like
preterm labour; ovarian torsion, hemorrhage or rupture; need for
emergency surgery) and encouraged to visit the hospital in case she
has any symptoms. Although the risk of malignancy is <1% [2], a
rapidly growing cyst must not be ignored. Surgery can be done via
laparotomy or laparoscopy. At surgery, comply with all the standards
of care so as to retrieve the cyst intact or without spillage. In our case,
laparoscopy was done and the cyst wall was retrieved aer aspiration
of contents within an endobag without any peritoneal spillage.
As there was an ectopic pregnancy, and normal ovarian tissue could
be delineated, in our case, cystectomy was done and the normal ovar-
ian tissue was preserved. e pathology report supported the diagno-
sis of LSLFCPP.
Expectant management of ovarian cysts in pregnancy is successful in
most cases, and should be routinely oered [4]. In case of large cysts
as well, conservative treatment can be opted for, unless there is tor-
sion or clinical discomfort for the patient, in which case the patient
may need surgery during pregnancy.
Comprehensive management is the need of the hour, in order to en-
sure ideal maternal and fetal wellbeing.
Acknowledgements
We thank and acknowledge the support of our institution, emergency
sta, operating theatre sta for successfully managing this case as a
team.
Conict of Interest Statement
All the authors involved in this case report declare that there is no
conict of interest for this particular study.
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J Case Rep Med, 2019 doi: 10.15761/JCRM.1000127 Volume 8(1): 5-5
Bindra V (2019) Large Solitary Luteinized Follicular Cyst of Pregnancy and Puerperium in Ruptured Ectopic Pregnancy Managed Laparoscopically
Copyright: © 2019 Bindra V. is is an open-access article distributed under the terms of the Creative Commons Aribution License, which
permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
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