A tale of two syndromes: ovarian hyperstimulation and abdominal compartment: Case report

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


    Abstract
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
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
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
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, 1999Go). Ovarian hyperstimulation syndrome cannot be reliably predicted before treatment (Delvigne et al., 1993Go) and ovarian stimulation is needed for effective treatment of infertility (Daya et al., 1995Go; 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
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
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 protocol 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 >=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 lengthxantero-posterior diameterxtransverse diameterx0.526 – 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 23.1x109/l to 32.2x109/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 of 2193 ml. Seven days after oocyte retrieval computerized tomography (CT) of the abdomen and pelvis (Figure 1Go) revealed massively enlarged ovaries lying side by side with combined dimensions of 20x14x20 cm. Many cysts showed evidence of haemorrhage. Neither ascites nor pleural effusions were radiologically evident.



View larger version (166K):
[in this window]
[in a new window]
 
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.

 
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., 1994Go) 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 2Go), 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 compartmentalization secondary to numerous intra-abdominal adhesions.



View larger version (14K):
[in this window]
[in a new window]
 
Figure 2 . Pelvic and upper abdominal pressures.

 
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 28x109/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
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
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., 1991Go; Navot et al., 1992Go; Simon et al., 1998Go). ACS is a result of pathologically increased IAP resulting in organ dysfunction (Sugerman et al., 1999Go). The scenario may consist of a tense abdomen, progressive oliguria despite volume expansion, hypercarbia and respiratory distress (Burch et al., 1996Go). Common causes of ACS include abdominal trauma or repair of ruptured abdominal aortic aneurysm (Nathens et al., 1997Go). 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., 1987Go) and may have an indolent onset (Reeves et al., 1997Go). The possibility of an endocrine induction of ACS has also been postulated (Carlo et al., 1998Go). `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, 1998Go).

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., 1984Go). 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 grading classification and defined treatment options (Burch et al., 1996Go). 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, 1999Go). Abdominal compartment syndrome can be a life-threatening complication of critical OHSS.


    Acknowledgments
 
The authors thank Mason Ross BSc for editorial advice.


    Notes
 
1 To whom correspondence should be addressed Back


    References
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
Aune, B., Hoie, K.E., Qian, P. et al. (1991) Does ovarian stimulation for in-vitro fertilization induce a hypercoagulable state? Hum. Reprod., 6, 925–927.[Abstract]

Burch, J.M., Moore, E.E., Moore, F.A. et al. (1996) The abdominal compartment syndrome. Surg. Clin. North. Am., 76, 833–842.[ISI][Medline]

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.[Medline]

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.[Abstract]

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.[Abstract]

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.[ISI][Medline]

Kron, I.L., Harman, P.K. and Nolan, S.P. (1984) The measurement of intra-abdominal pressure as a criterion for abdominal re-exploration. Ann. Surg., 199, 28–30.[ISI][Medline]

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.[Abstract]

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.[Free Full Text]

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.[ISI][Medline]

Reeves, S.T., Pinosky, M.L., Byrne, T.K. et al. (1997) Abdominal compartment syndrome. Can. J. Anaesth., 44, 308–312.[Abstract]

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.[ISI][Medline]

Sugerman, H.J., Bloomfield, G.L. and Saggi BW. (1999) Multisystem organ failure secondary to increased intraabdominal pressure. Infection, 27, 61–66.[Medline]

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.[Abstract]

Submitted on September 28, 1999; accepted on January 4, 2000.