Ovarian Hyperstimulation Syndrome with pleural effusion: a case report.

Abstract

To report a case of severe ovarian hyperstimulation syndrome (OHSS) with right pleural effusion following controlled ovarian hyperstimulation.
A 24-year-old woman had severe OHSS as a complication of gonadotropin stimulation. The clinical picture showed enlarged ovaries, massive ascites, pleural effusion, abdominal pain, and dyspnea. Beside the medical treatment, abdominal paracentesis for the drainage of the massive ascites and tube thoracostomy were performed, resulting in expansion of the lung.
Physicians can reduce the risk of OHSS by monitoring gonadotropin therapy and by withholding human chorionic gonadotropin medication. In in vitro fertilization protocols it can be advantageous to postpone the embryo transfer by freezing the embryos. Placement of a chest tube is a safe and efficient method for the treatment of pleural effusion.

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BioMed Central
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Cases Journal
Open Access
Case Report
Ovarian Hyperstimulation Syndrome with pleural effusion: a case
report
Recep Yildizhan*1, Ertan Adali1, Ali Kolusari1, Mertihan Kurdoglu1,
Cagdas Ozgokce1 and Fulya Adali2
Address: 1Department of Obstetrics and Gynecology, Yuzuncu Yil University, Van, Turkey and 2Department of Radiology, Woman and Child
Disease Hospital, Van, Turkey
Email: Recep Yildizhan* - recepyildizhan@yahoo.com; Ertan Adali - ertanadali@yahoo.com; Ali Kolusari - dralikolusari@yahoo.com;
Mertihan Kurdoglu - mkurdoglu@dotor.com; Cagdas Ozgokce - cagdasozgokce@gmail.com; Fulya Adali - fulyaadali@yahoo.com
* Corresponding author
Abstract
Background: To report a case of severe ovarian hyperstimulation syndrome (OHSS) with right
pleural effusion following controlled ovarian hyperstimulation.
Case presentation: A 24-year-old woman had severe OHSS as a complication of gonadotropin
stimulation. The clinical picture showed enlarged ovaries, massive ascites, pleural effusion,
abdominal pain, and dyspnea. Beside the medical treatment, abdominal paracentesis for the
drainage of the massive ascites and tube thoracostomy were performed, resulting in expansion of
the lung.
Conclusion: Physicians can reduce the risk of OHSS by monitoring gonadotropin therapy and by
withholding human chorionic gonadotropin medication. In in vitro fertilization protocols it can be
advantageous to postpone the embryo transfer by freezing the embryos. Placement of a chest tube
is a safe and efficient method for the treatment of pleural effusion.
Background
Ovarian hyperstimulation syndrome (OHSS) is the most
serious complication of controlled ovarian hyperstimula-
tion. OHSS is cystic enlargement of the ovaries and a fluid
shift from the intravascular space to the third space due to
increased capillary permeability. Its occurrence is depend-
ent on the administration of human chorionic gonadotro-
pin (hCG) after an exaggerated ovarian response to
gonadotropin stimulation. The syndrome is relatively
common, occurring in up to 5% of women undergoing in
vitro fertilization (IVF) or intrauterine insemination (IUI)
procedures [1].
Despite close monitoring during ovarian stimulation and
rigid guidelines and criteria for canceling cycles, OHSS
still occurs. Clinical manifestations of OHSS can be classi-
fied into three forms. In mild forms of OHSS the ovaries
are enlarged, while in moderate forms there is additional
accumulation of ascites with mild abdominal distension.
Its severe form in ~0.5% of stimulated cycles [2] is charac-
terized by hemoconcentration, thrombosis, oliguria,
pleural effusion, rarely pericardial effusion, and respira-
tory distress [3]. It can lead to life-threatening complica-
tions including thromboembolic events and even death.
Published: 18 November 2008
Cases Journal 2008, 1:323 doi:10.1186/1757-1626-1-323
Received: 27 September 2008
Accepted: 18 November 2008
This article is available from: http://www.casesjournal.com/content/1/1/323
© 2008 Yildizhan et al; licensee BioMed Central Ltd.
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0),
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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Cases Journal 2008, 1:323http://www.casesjournal.com/content/1/1/323
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Case presentation
A 24-year-old woman was hospitalized in a state hospital
because of confirmed bilateral ovarian cysts and ascites.
Then she was referred to our hospital because of increas-
ing abdominal girth and dyspnea of 1 week's duration.
The patient's weight was 80 kg and height was 162 cm.
The stimulation was performed by an external unit using
a long protocol with follitropin alfa. The stimulation cycle
had to be discontinued because of OHSS. The patient
showed a severe form of OHSS with massive ascites,
abdominal pain, dyspnea, blood pressure 80/40 mmHg,
pulse rate 100 beats/min, plasma estradiol 4000 pg/mL,
β-hCG 86 IU/L, hemoconcentration of 52%, hemoglobin
of 18.0 g/dL, leucocytosis of 29 × 103/μL, PaO2 of 70
mmHg, PaCO2 of 40 mmHg, pH of 7.44, and oxygen sat-
uration of 91.2%. The ultrasonographic examination
revealed bilaterally enlarged multicystic ovaries and a
large amount of ascites (Figure 1).
Immediately after admission, infusion therapy was
started, consisting of normal saline-infusion 0.9% 1000
ml (Pharmacia), glucose 5% 1000 ml (Pharmacia), four
times 50 ml of human serum albumin 20%, and low-
molecular weight heparin 5000 IU 2 × 1 per day. Body
weight, abdominal circumference, intake and outputs,
ultrasonography, and laboratory studies were monitored
strictly daily. Renal function was supported using diuret-
ics (Furosemide 20 mg ampoule and Furosemide tablet).
Renal function was not disturbed and there were no seri-
ous changes in serum electrolytes. The patients' girth
increased between days 2 and 7, and then abdominal
paracentesis for the drainage of the massive ascites
became necessary. The patient reported increasing dysp-
nea. The anteroposterior chest X-ray (Figure 2) revealed
right pleural effusion, whereupon a chest-tube was placed
for treatment of pleural effusion.
Discussion
Ovarian hyperstimulation syndrome (OHSS) is an iatro-
genic, serious complication of controlled ovarian hyper-
stimulation, usually self-limited, but occasionally life
threatening and typically occurs with gonadotropin,
rarely with clomiphene citrate [4]. Without hCG, OHSS is
extremely rare. The symptoms are more severe and persist
longer if pregnancy is successful. Patients who are preg-
nant sustain the ovarian luteinization process by the pro-
duction of hCG. Although the pathophysiology of this
syndrome has not been completely elucidated, the under-
lying mechanism responsible for the clinical manifesta-
tions of OHSS appears to be an increase in capillary
permeability of mesothelial surfaces [5]. There is increas-
ing evidence that certain vasoactive substances such as
vascular endothelial growth factor (VEGF), cytokines (IL-
2, IL-6, and IL-8), tumor necrosis factor-alpha (TNF-
alpha), and the ovarian renin-angiotensin system, which
are activated by gonadotropin, can lead to increased vas-
cular permeability and extravascular fluid accumulation
in OHSS [6,7].
VEGF has a major role in the pathogenesis of OHSS. VEGF
is a heparin-binding glycoprotein with vascular permea-
bility-enhancing, angiogenic, and endothelial cell-specific
mitogenic activities [8]. VEGF increases vascular permea-
bility, which may explain fluid leakage in the third space.
This leakage is responsible for the development of ascites,
pleural effusions, edema, and hemoconcentration [9].
Ultrasonographic examination revealed bilaterally enlarged multicystic ovaries
Figure 1
Ultrasonographic examination revealed bilaterally
enlarged multicystic ovaries.
The anteroposterior chest X-ray revealed right pleural effu-sion
Figure 2
The anteroposterior chest X-ray revealed right pleu-
ral effusion.

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Clinical characteristics of OHSS include ascites and pleu-
ral effusion induced by increased vascular permeability,
where VEGF was suspected to be the culprit. Through up-
regulation of VEGF, hCG plays a significant role in the
pathogenesis of OHSS [10]. Navot et al. [11] detected high
levels of prorenin and angiotensin 2 in patients' follicle
fluid and high renin concentrations in their plasma.
OHSS, characterized by third space fluid shift and intra-
vascular volume depletion, results in massive ascites and
hydrothorax. There are various comments about the
development of the pleural fluid. It is adduced that high
estrogen levels cause pleural effusion. Pleural effusion
usually occurs in the severe forms of OHSS [12]. In our
case pleural effusion occurred during the initial days of
treatment. Loret de Mola et al [13] observed that pleural
effusion in OHSS usually occurs on the right side of the
lung, as in our case. They offer the explanation that lym-
phatic drainage on the right is less than that on the left
and the diaphragmatic hollows are greater on the right. It
is possible that pleural effusion originates from the fluid
shift from abdominal ascites [12].
From the outset of treatment, our patient, who had severe
dyspnea and impaired blood gases, was protected from
possible pulmonary emboli. We gave a light molecular
heparin. Then we eliminated the pulmonary emboli. Tho-
racocentesis is safe and efficient for the treatment of
hydrothorax and lung collapse in cases of OHSS and may
be repeated as often as necessary [14]. Our patient also
required either paracentesis or thoracentesis four times. A
chest-tube was installed in the patient by thoracic sur-
geons.
If the OHSS develops within an IVF protocol, it may be
prudent to postpone the embryo transfer since the estab-
lishment of pregnancy can lengthen the recovery time or
contribute to a more severe course. Instead of canceling
the cycle, it is also possible to administer HCG, to retrieve
the oocytes, and to freeze all embryos.
Physicians can reduce the risk of OHSS by monitoring
FSH therapy to use this medication cautiously, and by
withholding hCG medication. The patients with OHSS
must be treated urgently and with multidisciplinary man-
agement. If left untreated, OHSS can result in serious
health complications and even death.
Consent
Written informed consent was obtained from the patient
for publication of this case report and accompanying
images. A copy of the written consent is available for
review by the Editor-in-Chief of this journal.
Competing interests
The authors declare that they have no competing interests.
Authors' contributions
All authors were involved in patient's care. RY, EA, AK pre-
pared the manuscript, MK, CO, FA edit and coordinated
the manuscript. All authors read and approved the final
manuscript.
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