Human Reproduction vol.15 no.5 pp.1058–1060, 2000
CASE REPORT
A tale of two syndromes: ovarian hyperstimulation and
abdominal compartment
Tulin Cil, Ian S.Tummon1, Andrew A.House,
Brian Taylor, Glen Hooker, Jason Franklin,
Richard Rankin and Mark Carey
Department of Obstetrics and Gynaecology, The University of
Western Ontario, 339 Windermere Road, London, Ontario N65
5A5, Canada
1To
whom correspondence should be addressed
Abdominal compartment syndrome complicated severe
ovarian hyperstimulation in a 35 year old woman with
multiple bowel resections due to Crohn’s disease. Pain
from ovarian enlargement necessitated hospital admission.
Despite intravenous fluid administration and heparin prophylaxis, ilio-femoral deep vein thrombosis developed.
Treatment by intravenous heparin was complicated by
repeated intra-ovarian bleeding, anaemia and acute renal
failure requiring haemodialysis. Intra-abdominal pressures
were elevated. After placement of an inferior vena caval
filter and discontinuation of heparin, there was slow spontaneous recovery without surgery.
Key words: abdominal compartment syndrome/ovarian
hyperstimulation
Introduction
Full understanding of the complications of ovarian hyperstimulation syndrome (OHSS) is essential. Severe OHSS is almost
always iatrogenic in origin and accurate informed consent
about OHSS is the physician’s responsibility (Mathur and
Jenkins, 1999). Ovarian hyperstimulation syndrome cannot be
reliably predicted before treatment (Delvigne et al., 1993)
and ovarian stimulation is needed for effective treatment of
infertility (Daya et al., 1995; Zahed et al., 1997).
The present case is a description of OHSS uniquely complicated by abdominal compartment syndrome (ACS) resulting
in critical, life-threatening illness.
Case report
A 35 year old woman with now quiescent Crohn’s disease had
3 years of primary infertility due to bilateral tubal obstruction.
She had initially been diagnosed with ulcerative colitis, but
after failure of an ileo-anal pouch procedure, Crohn’s disease
was diagnosed, the pouch removed and a permanent ileostomy
created. In-vitro fertilization (IVF) was advised as the most
appropriate fertility-promoting treatment. A long luteal proto1058
col with nafarelin 200 µg intranasally twice daily (Synarel®;
Ferring Inc., North York, Ontario, Canada) followed by 150
IU follicle stimulating hormone (FSH) (Fertinorm®; Serono,
Oakville, Ontario, Canada) was used. Ultrasound showed a
6 cm left hydrosalpinx. After 12 days of FSH stimulation,
oestradiol was 11 700 pmol/l and 10 follicles measured
艌1.8 cm diameter. Ovulation was triggered with 10 000 IU
human chorionic gonadotrophin (HCG) (Pregnyl®; Organon,
Scarborough, Ontario, Canada) and 14 oocytes were recovered.
In order to aspirate follicles from the left ovary, the hydrosalpingeal fluid was aspirated transvaginally.
The day after oocyte retrieval the patient felt unwell and
complained of pelvic pain and pressure. She was admitted to
hospital and treated with i.v. crystalloid, analgesics and heparin
5000 IU s.c. every 12 h to prevent deep vein thrombosis.
Two days after oocyte retrieval, abdominal pain became
generalized, and urine output and ileostomy output decreased.
She was afebrile with signs of left lower quadrant
peritonitis. Plain abdominal radiographs showed no free air.
Ultrasonography 2 days after oocyte retrieval showed the
ovaries measured 15.5 cm (right) and 11.1 cm (left) in
maximal diameter, with a combined volume of 1341 ml
(calculated by length⫻antero-posterior diameter⫻transverse
diameter⫻0.526 – for a non-spherical volume). The patient
was started on i.v. clindamycin and gentamicin on the
presumption of pelvic sepsis. Seven embryos were cryopreserved and no fresh embryo transfer was performed.
Three days after oocyte retrieval her left calf and thigh were
swollen and tender. Duplex Doppler scans confirmed extensive
left ilio-femoral deep vein thrombosis and she was placed on i.v.
heparin to maintain a therapeutic partial thromboplastin time.
During days 4–7 after oocyte retrieval abdominal girth
increased and leukocytes rose from 23.1⫻109/l to 32.2⫻109/l.
Repeated intra-ovarian bleeding complicated treatment by
intravenous heparin and haemoglobin fell to 66 g/l from a
pretreatment baseline of 132 g/l. Renal failure developed with
a progressive increase in serum creatinine from 52–539 µmol/l
and urine output of ~10ml/h despite i.v. fluid replacement.
Doppler ultrasound demonstrated patent renal veins with normal renal arterial blood flow bilaterally. There was no evidence
of hydronephrosis and the renal parenchyma appeared normal.
Urine electrolytes and clinical assessment did not support a
pre-renal component to renal insufficiency.
Six days after oocyte retrieval, ultrasonographic measurements of the ovary showed that the ovary had increased to
16.6 cm (right) and 17.1 cm (left) with a combined volume
© European Society of Human Reproduction and Embryology
OHSS and abdominal compartment syndrome
Figure 1. Computerized tomography (CT) of pelvis: CT slice
through the lower abdomen. Enlarged cystic ovaries are seen
between two diagonal arrows. A fluid–fluid level is seen in the left
ovarian cyst (vertical arrow), between haemorrhage and cyst fluid.
The density of the haemorrhagic portion was 56.8 CT units.
Figure 2. Pelvic and upper abdominal pressures.
of 2193 ml. Seven days after oocyte retrieval computerized
tomography (CT) of the abdomen and pelvis (Figure 1)
revealed massively enlarged ovaries lying side by side with
combined dimensions of 20⫻14⫻20 cm. Many cysts showed
evidence of haemorrhage. Neither ascites nor pleural effusions
were radiologically evident.
On days 8 and 9 after oocyte retrieval the patient became
anuric despite vigorous volume expansion with crystalloid
and colloid. The abdomen was tense and distended with no
ileostomy output. The working diagnosis was early onset
severe OHSS (Lyons et al., 1994) complicated by ACS.
Elevated intra-abdominal pressure (IAP) may have reduced
venous return along ilio-femoral vessels and compromised
renal and splanchnic perfusion. On day 8 IAP was 54 cm H2O
in the bladder and 21 cm H2O in the stomach (Figure
2), determined by manometry of the bladder catheter and
nasogastric tube respectively. The discrepancy between upper
and lower abdominal pressures could be attributed to ovarian
sequestration within the lower abdomen and pelvis and com-
partmentalization secondary to numerous intra-abdominal
adhesions.
Decompressive laparotomy was contemplated to relieve
intra-abdominal tension. The complexity of adhesions, tissue
fragility, anaemia, renal failure and anti-coagulated status all
contributed to elevate surgical risk. An interdisciplinary team
made the decision to manage the patient conservatively with
continued intravascular fluid support and careful monitoring.
On day 9, after transfusion, the haemoglobin was 96 g/l,
haematocrit 0.28, leukocytes 28⫻109/l. and creatinine
597 µmol/l. Arterial blood gases showed metabolic acidosis
and compensatory respiratory alkalosis with pH 7.25, HCO3
10 mmol/l, and pCO2 (partial pressure) 24 mm Hg. On day
10 after oocyte retrieval i.v. heparin was discontinued, a jugular
haemodialysis catheter and an infra-renal inferior vena caval
filter were placed. Haemodialysis was initiated for control of
metabolic derangement. Repeat CT revealed no change in
ovarian size and showed numerous haemorrhagic areas with
the ovaries.
On day 11 urine output rose marginally and stabilized at
5–15 ml/h, minimal ileostomy output resumed and her abdomen
progressively became less tense.
Over the course of the next month, the patient displayed
steady clinical improvement and significant recovery of renal
function with a creatinine of 92 µmol/l. She was discharged
33 days after oocyte retrieval and placed on oral anticoagulation therapy, with a view to return for return of
cryopreserved embryos after her convalescence.
Discussion
The present case illustrates a unique overlap of two syndromes,
OHSS and ACS. Severe OHSS may cause critical illness with
ovarian enlargement, ascites, hypercoagulability and renal
failure (Aune et al., 1991; Navot et al., 1992; Simon et al.,
1998). ACS is a result of pathologically increased IAP resulting
in organ dysfunction (Sugerman et al., 1999). The scenario
may consist of a tense abdomen, progressive oliguria despite
volume expansion, hypercarbia and respiratory distress (Burch
et al., 1996). Common causes of ACS include abdominal
trauma or repair of ruptured abdominal aortic aneurysm
(Nathens et al., 1997). ACS is well known in the realms of
trauma surgery and intensive care but not in association with
assisted reproduction. However, ACS may occur in association
with an ovarian mass (Celoria et al., 1987) and may have an
indolent onset (Reeves et al., 1997). The possibility of an
endocrine induction of ACS has also been postulated (Carlo
et al., 1998). ‘Pelvic compartment syndrome’ is a term that
has been used to describe renal failure due to bilateral ureteric
compression from a retroperitoneal haematoma (Hessman and
Rommens, 1998).
Diagnosis of ACS is based on symptoms, signs and indirect
measurements of IAP. As in this case, IAP is assessed by
instilling sterile saline into an indwelling catheter, which is
then clamped (Kron et al., 1984). A manometer is placed and
a pressure reading is obtained utilizing the symphysis pubis
as the zero mark. Such measurements provide an objective
criterion to classify IAP. Burch and co-workers proposed a
1059
T.Cil et al.
grading classification and defined treatment options (Burch
et al., 1996). Patients with IAP ⬍ 15 cm H2O (Grade I) rarely
need treatment. Grade II includes IAP 15–25 cm H2O; such
patients require close monitoring but no intervention in the
absence of clinical findings. Patients with IAP of 25–35 cm
H2O (Grade III) frequently require surgical decompression via
laparotomy. Grade IV is defined as IAP ⬎ 35 cm H2O
and patients in this category usually require decompressive
laparotomy. Given the time-limited nature of OHSS and the
surgical risk in this unique case, laparotomy may have resulted
in either improvement or castration or catastrophe. This patient
recovered spontaneously without surgery.
In the age of assisted reproduction it is essential to understand
potential hazards of treatment (Mathur and Jenkins, 1999).
Abdominal compartment syndrome can be a life-threatening
complication of critical OHSS.
Acknowledgements
The authors thank Mason Ross BSc for editorial advice.
References
Aune, B., Hoie, K.E., Qian, P. et al. (1991) Does ovarian stimulation for invitro fertilization induce a hypercoagulable state? Hum. Reprod., 6, 925–927.
Burch, J.M., Moore, E.E., Moore, F.A. et al. (1996) The abdominal
compartment syndrome. Surg. Clin. North. Am., 76, 833–842.
Carlo, V.M., Ramirez Schon, G., Suarez Irrizary, G. et al. (1998) The
abdominal compartment syndrome: a report of 3 cases including instance
of endocrine induction. Bol. Asoc. Med. P. R., 90, 121–125.
Celoria, G., Steingrub, J., Dawson, J.A. and Teres, D. (1987) Oliguria from
high intra-abdominal pressure secondary to ovarian mass Crit. Care Med.,
15, 78–79.
Daya, S., Gunby, J., Hughes, E.G. et al. (1995) Natural cycles for in-vitro
fertilization: cost-effectiveness analysis and factors influencing outcome.
Hum. Reprod., 10, 1719–1724.
Delvigne A., Dubois M., Battheu B. et al. (1993) The ovarian hyperstimulation
syndrome in in-vitro fertilization: a Belgian multicentric study. II. Multiple
discriminant analysis for risk prediction. Hum. Reprod., 8, 1361–1366.
Hessman, M. and Rommens, P. (1998) Bilateral ureteral obstruction and renal
failure caused by massive retroperitoneal hematoma: is there a pelvic
compartment syndrome analogous to abdominal compartment syndrome?
J. Orthop. Trauma, 12, 553–557.
Kron, I.L., Harman, P.K. and Nolan, S.P. (1984) The measurement of intraabdominal pressure as a criterion for abdominal re-exploration. Ann. Surg.,
199, 28–30.
Lyons, C.A., Wheeler, C.A., Frishman, G.N. et al. (1994) Early and late
presentation of the ovarian hyperstimulation syndrome: two distinct entities
with different risk factors. Hum. Reprod., 9, 792–799.
Mathur, R.S. and Jenkins, J.M. (1999) Patients should be allowed to weigh
the morbidity of OHSS against the benefits of parenthood. Hum. Reprod.,
14, 2183–2189.
Nathens, A.B., Brenneman, F.D. and Boulanger, B.R. (1997) The abdominal
compartment syndrome. Can. J. Surg., 40, 255–258.
Navot, D., Berg, P.A. and Laufer, N. (1992) Ovarian hyperstimulation
syndrome in novel reproductive technologies: prevention and treatment.
Fertil. Steril., 58, 249–261.
Reeves, S.T., Pinosky, M.L., Byrne, T.K. et al. (1997) Abdominal compartment
syndrome. Can. J. Anaesth., 44, 308–312.
Simon, A., Revel, A., Hurwitz, A. et al. (1998) The pathogenesis of ovarian
hyperstimulation syndrome: a continuing enigma. J. Assist. Reprod. Genet.,
15, 202–208.
Sugerman, H.J., Bloomfield, G.L. and Saggi BW. (1999) Multisystem organ
failure secondary to increased intraabdominal pressure. Infection, 27, 61–66.
Zayed, F., Lenton, E.A. and Cooke, I.D. (1997) Natural cycle in-vitro
fertilization in couples with unexplained infertility: impact of various factors
on outcome. Hum. Reprod., 12, 2402–2407.
Received on September 28, 1999; accepted on January 4, 2000
1060