Hindawi Publishing Corporation

Case Reports in Critical Care

Volume 2012, Article ID 189705, 4 pages
doi:10.1155/2012/189705

Case Report
Spontaneous Ovarian Hyperstimulation Syndrome in
a Triplet Pregnancy

Nisha Rani Agrawal,1 Garima Gupta,1 Kusum Verma,1 and Neeraj Varyani2
1 Department of Obstetrics and Gynecology, Institute of Medical Sciences, Banaras Hindu University,
Uttar Pradesh, Varanasi 221005, India
2 Department of General Medicine, Institute of Medical Sciences, Banaras Hindu University,

Uttar Pradesh, Varanasi 221005, India

Correspondence should be addressed to Nisha Rani Agrawal, nisharaniagrawal@gmail.com

Received 18 August 2012; Accepted 1 October 2012

Academic Editors: C. Diez, M. Doganay, and C. Lazzeri

Copyright © 2012 Nisha Rani Agrawal et al. This is an open access article distributed under the Creative Commons Attribution
License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly
cited.

Ovarian hyperstimulation syndrome (OHSS) is a potentially life-threatening complication, usually iatrogenic after ovulation
induction. OHSS is a very rare event in spontaneously conceived pregnancies. Only few cases have been reported in literature
to the best of our knowledge. We report a very rare case of spontaneous critical OHSS (according to classification of severity of
OHSS as mentioned in Greentop guidelines no. 5, 2006) associated with triplet pregnancy in a 26-year-old woman suffering from
severe abdominal pain, distension, and dyspnea. Our case highlights the importance of a strong suspicion for OHSS when a clinical
presentation could not be explained by common medical conditions.

1. Introduction our case. Misdiagnosis as neoplasm may result in inadvertent

interventions. Nevertheless, alternative diagnoses should
Ovarian hyperstimulation syndrome is a rare and potentially always be considered, such as complication of an ovarian cyst
life-threatening complication of ovarian stimulation. OHSS (torsion, haemorrhage), pelvic infection, intraabdominal
is in almost all cases an iatrogenic complication, the inci- haemorrhage, malignancy, ectopic pregnancy, and appen-
dence of which is increasing world-wide through an increase dicitis.
in controlled ovarian hyperstimulation cycles. Thus knowledge and prompt recognition of ovarian
OHSS has a broad pathophysiologic spectrum ranging hyperstimulation are essential for its prevention and man-
from mild illness to severe disease. As many as 33% of in vitro agement.
fertilization (IVF) cycles have been reported to be associated
with mild forms of OHSS [1]. More severe OHSS has been 2. Case Presentation
reported in 3.1–8.0% of IVF cycles [1]. Women without
any pharmacological intervention are rarely diagnosed with A 26-year primigravida at 6 weeks of gestation presented with
spontaneous OHSS [2] which needs prompt evaluation and severe abdominal pain, distension, and dyspnea for the past
management. Risk factors for OHSS include young age, low 1 month, which was slow in onset and gradually progressing.
body weight, polycystic ovarian syndrome, higher doses of The patient conceived spontaneously and denied having ever
gonadotropins, and previous episodes of hyperstimulation. taken any ovulation inducing agent. Menarche commenced
Even after an extensive search of literature, we were at age of 13 years and subsequently she had oligomenorrhea.
unable to find the incidence of critical spontaneous OHSS She had a history of primary infertility of 8 years for which
which makes our case even more significant. Symptoms of she was neither investigated nor was on any treatment.
OHSS do not cease immediately after pregnancy, but may There was no history of fever, cough, chest pain, palpitation,
persist or even worsen during days after treatment [3] as in jaundice, melena, hematemesis, decreased urine output,
2 Case Reports in Critical Care

termination of pregnancy was done on patient’s request at

10 weeks of gestation. Subsequently she developed pyoperi-
toneum which did not resolve with conservative manage-
ment. Therefore she underwent exploratory laparotomy for
drainage of pyoperitoneum and pus was sent for culture
and sensitivity testing. Dense adhesions with septations were
present. There was no evidence of uterine or intestinal
perforation. Polycystic right ovary of approximately 8 ×
6 cm adherent to parietal abdominal wall was present, which
reconfirmed our diagnosis of critical OHSS. E. coli was grown
Figure 1: Ultra Sonography showing three gestational sacs with
in pus culture which was sensitive to ceftriaxone. Antibiotics
gross ascites. were prescribed accordingly. Following this patient’s general
condition improved and is in our followup.

altered bowel habit, antitubercular treatment, joint pain,

rashes, photosensitivity, oral ulcers, loss of appetite, vaginal 3. Discussion
bleeding, treatment for infertility, hyperemesis, headache, In this case study spontaneous OHSS in association with
visual blurring, weight loss, or tremors. Her past and triplet pregnancy of 6-weeks of gestation was observed which
family history was not significant. On examination she is a very rare occurrence and requires documentation.
was conscious, oriented, afebrile, pale and ill looking, In 1996 Olatunbosun et al. [4] first reported case of
weight: 78 kg, pulse: 110/min and regular, blood pressure: severe spontaneous OHSS associated with pregnancy and
126/80 mm Hg, and respiratory rate being 26/min. Icterus, polycystic ovarian disease to result in live births. In this case
clubbing, cyanosis, and pedal edema were absent. Lymph study patient had polycystic ovaries, thus sharing a common
nodes were not palpable. Thyroid and breast examination background of excess ovarian follicle growth as in our case
were normal. Abdomen was distended with abdominal girth study.
of 97 cm. Tense ascites and fluid thrill were present. There The etiology and pathophysiologic characteristics of
was no organomegaly. Pelvic examination revealed congested OHSS are poorly understood. OHSS is a systemic disease
cervix. Uterus and ovaries could not be palpated because of resulting from vasoactive products released from hyper-
tense ascites. Chest examination suggested signs of bilateral stimulated ovaries. It is characterized by increased capillary
pleural effusion. permeability, leading to leakage of fluid from the vascular
With this clinical presentation following conditions were compartment, with third space fluid accumulation and
considered as provisional diagnosis: intravascular dehydration [5]. Various factors including
(i) Koch’s abdomen, estrogen, histamine, prostaglandins, aldosterone, renin, and
angiotensin II have been implicated in the development of
(ii) Malignancy, this condition. Recent studies show high renin-like activity
(iii) Collagen vascular disease. and elevated angiotensin II immunoreactivity in both plasma
and ascitic fluid (angiotensin II being 6–9 fold higher
On investigating, patient had anemia (Hb-88 gm/L, Hct- than plasma) [6]. These findings are in favor of ovarian
0.27), leucocytosis (14.5 × 109 /L) with polymorphs predom- origin of the elevated renin-like activity and angiotensin
inance. Her platelet count, chest X-ray, liver, renal, thyroid II immunoreactivity in ascitic fluid of severe OHSS and
function tests, EKG, collagen profile, hormonal assays, and suggest a stimulatory role of human chorionic gonadotropin
coagulation profile were normal. Ascitic tap examination (hCG) on the ovarian renin-angiotensin system during
revealed transudative picture. Aspiration cytology was neg- severe OHSS.
ative for malignant cells and culture was negative for bacteria One third of the patients developing OHSS after IVF
and fungi. Input output charting was 2.2 L/1.2 L. Abdominal had no previous risk criteria. Exogenous and/or endogenous
sonography revealed 3 intrauterine gestation sacs of 6wk2d, hCG is suggested as an etiologic factor. Recent studies
6wk3d, and 4wk3d, respectively. Right ovary was enlarged have revealed the phenomenon of spontaneous OHSS in
8.2 × 7.4 × 8.3 cm with multiple large cysts and severe ascites pregnancy to be caused by a mutation in FSH receptor which
with bilateral pleural effusion was present, suggestive of tends to recur in subsequent pregnancies [7].
Ovarian HyperStimulation Syndrome (Figure 1). Differential diagnosis includes disorders such as hyper-
A diagnosis of critical OHSS was made. Her manage- reaction luteinalis, luteomas of pregnancy, and theca lutein
ment was carefully tailored considering her pregnancy and cyst.
severity of OHSS. Conservative management was initiated. Management of OHSS is tailored according to its severity.
Body weight, abdominal circumference, input/output, serum Each case should be classified [8] as in Table 1.
electrolytes, ultrasonography, and renal functions were Management guidelines for OHSS as per Green-top
closely monitored on daily basis. Later ultrasound-guided Guidelines no. 5 available from http://www.rcog.org.uk/
abdominal paracentesis was performed 3 times, in view of womens-health/clinical-guidance/management-ovarian-
respiratory distress and increasing abdominal girth. Medical hyperstimulation-syndrome-green-top-5 are as follows.
Case Reports in Critical Care 3

Table 1 paracetamol and if necessary oral or parenteral opiates. Non-

Grade Symptoms
steroidal anti-inflammatory agents are not recommended.
Antiemetic drugs used should be those appropriate for the
Abdominal bloating possibility of early pregnancy, such as prochlorperazine,
Mild OHSS Mild abdominal pain metoclopramide, and cyclizine. Women admitted to hospital
Ovarian size usually <8 cm with OHSS should be assessed at least daily, with more
Moderate abdominal pain frequent assessment of those with critical OHSS.
Nausea/vomiting
Moderate OHSS
Ultrasound evidence of ascites 3.1. Inpatient Monitoring of Patients with OHSS as in
Ovarian size usually 8–12 cm Table 2. Allowing women to drink according to their thirst
Clinical ascites represents the most physiological approach to replacing
Oliguria volume. Women with severe OHSS with persistent olig-
Severe OHSS Haemoconcentration >0.45
uria and haemoconcentration despite initial colloid volume
expansion may need invasive monitoring and should be
Hypoproteinemia
discussed with an anaesthetist. Diuretics should be avoided
Ovarian size usually >12 cm as they deplete intravascular volume, although they may
Tense ascites have a role with careful haemodynamic monitoring in cases
Haemoconcentration >0.55 where oliguria persists despite adequate intravascular volume
White cell count >25 × 109 /L expansion and a normal intra-abdominal pressure. Para-
Critical OHSS
Oligo/anuria centesis should be considered in women who are distressed
due to abdominal distension or in whom oliguria persists
Thromboembolism
despite adequate volume replacement. Paracentesis should be
Acute respiratory distress syndrome performed under ultrasound guidance to avoid inadvertent
puncture of vascular ovaries distended by large luteal cysts.
Table 2 Intravenous colloid replacement should be considered for
women who have large volumes of ascitic fluid drained.
Assessment Measurements Pelvic surgery should be restricted to cases with adnexal
Pain torsion or coincident problems requiring surgery and only
Breathlessness undertaken by an experienced surgeon following careful
Hydration assessment. Women should be reassured that pregnancy may
History and continue normally despite OHSS, and there is no evidence of
Weight
Examination an increased risk of congenital abnormalities.
Heart rate, blood pressure
Cardiovascular
Abdominal girth, distension, ascites 4. Conclusion
Intake and output chart
Our case highlights the importance of a strong suspicion for
Full blood count OHSS when a clinical presentation could not be explained by
Haemoglobin, haematocrit, and common medical conditions. Early identification of OHSS
white cell count in a patient can improve the quality of life. If left untreated,
Urea and electrolytes OHSS can result in serious health complications and even
Liver function tests death.
Investigations
Baseline clotting studies
Pelvic ultrasound (for ascites and Conflict of Interests
ovarian size)
Chest X-ray or ultrasonography (if The authors declare that they have no conflict of interests.
respiratory symptoms)
ECG and echocardiogram (if
suspect pericardial effusion) Acknowledgments
This case study was granted approval by Ethical Committee,
Institute of Medical Sciences, Banaras Hindu University,
Hospital admission should be recommended to women Varanasi-221005. The authors N. R. Agrawal, G. Gupta,
with severe OHSS. Women should be kept under review K. Verma, N. Varyani declare that they have obtained
until resolution of the condition. Multidisciplinary assistance written, informed consent for the publication of the details
should be sought for all women with critical or severe OHSS relating to the patient in this report, all possible steps have
who have persistent haemoconcentration and dehydration. been taken to safeguard the identity of the patient, this
Features of critical OHSS should prompt consideration of submission is compliant with the requirements of local
the need for intensive care. Pain relief is best provided with research ethics committee, all images, figures and tables are
4 Case Reports in Critical Care

free of copyright, and all authors have approved of the final

version before submission.

References
[1] A. Delvinge and S. Rozenberg, “Epidemiology and prevention
of ovarian hyperstimulation syndrome (OHSS): a review,”
Human Reproduction Update, vol. 8, no. 6, pp. 559–577, 2002.
[2] A. Ayhan, Z. S. Tuncer, and A. T. Aksu, “Ovarian hyper-
stimulation syndrome associated with spontaneous pregnancy,”
Human Reproduction, vol. 11, no. 8, pp. 1600–1601, 1996.
[3] M. Ludwig, U. Gembruch, O. Bauer, and K. Diedrich, “Ovarian
hyperstimulation syndrome (OHSS) in a spontaneous preg-
nancy with fetal and placental triploidy: information about the
general pathophysiology of OHSS,” Human Reproduction, vol.
13, no. 8, pp. 2082–2087, 1998.
[4] O. A. Olatunbosun, B. Gilliland, L. A. Brydon et al., “Spon-
taneous OHSS in four consecutive pregnancy,” Clinical &
Experimental Obstetrics & Gynecology, vol. 23, no. 3, pp. 127–
132, 1996.
[5] A. Delbaere, G. Smits, O. Olatunbosun, R. Pierson, G. Vassart,
and S. Costagliola, “New insights into the pathophysiology of
ovarian hyperstimulation syndrome. What makes the differ-
ence between spontaneous and iatrogenic syndrome?” Human
Reproduction, vol. 19, no. 3, pp. 486–489, 2004.
[6] A. Delbaere, P. J. Bergmann, C. Gervy-Decoster et al.,
“Increased angiotensin II in ascites during severe ovarian hyper-
stimulation syndrome: role of early pregnancy and ovarian
gonadotropin stimulation,” Fertility and Sterility, vol. 67, no. 6,
pp. 1038–1045, 1997.
[7] U. B. Kaiser, “The pathogenesis of the ovarian hyperstimulation
syndrome,” The New England Journal of Medicine, vol. 349, no.
8, pp. 729–732, 2003.
[8] R. Mathur, I. Evbuomwan, and J. Jenkins, “Prevention and
management of ovarian hyperstimulation syndrome,” Current
Obstetrics and Gynaecology, vol. 15, no. 2, pp. 132–138, 2005.