Retzius space haematoma as a rare cause of concealed retroperitoneal postpartum haemorrhage following spontaneous vaginal delivery

A 34-year-old woman presented to hospital in shock 8 hours after a spontaneous home vaginal delivery. She is a known carrier of haemophilia A. Examination revealed a tender but firm, central and contracted uterus. There was a small second-degree perineal tear which was not actively bleeding and no other trauma or bleeding visualised in the vault or cervix. Bedside ultrasound showed a large 1 L blood clot anterior to the empty uterus. Exploratory laparotomy was performed which revealed a haematoma in the Retzius space with extension beneath the anterior serosa of the bladder, through the left broad ligament and into the retroperitoneal space. Bleeding from the Santorini plexus was controlled with haemostatic sutures, reinforced with FLOSEAL and intra-abdominal packing. A CT angiogram and relook laparotomy the following day confirmed haemostasis and facilitated removal of intra-abdominal packs. This case demonstrates a very rare cause of postpartum retroperitoneal haemorrhage.

obstetrics and gynaecology, pregnancy

Concealed postpartum haemorrhage requires a high index of suspicion and urgent management to reduce morbidity and mortality. Women typically present with shock out of proportion to their estimated external blood loss. Concealed postpartum haemorrhage may be intrauterine, intra-abdominal or within a haematoma.1 Puerperal haematomas are mainly located in the vulva, vagina, broad ligament and rarely may be retroperitoneal.2 Retroperitoneal haematomas are a rare cause of concealed postpartum haemorrhage and may be difficult to diagnose.2 They are most commonly caused by splenic artery rupture, renal artery aneurysms, ruptured renal angiomyolipoma and ovarian artery rupture.2 Retzius space haematoma after spontaneous vaginal delivery, as described in this case, is an exceptionally rare cause of retroperitoneal haemorrhage and poses difficulty in both diagnosis and management. Case presentation A 34-year-old woman presented to our emergency department in shock 8 hours after a spontaneous vaginal delivery at home. Her labour was reportedly uncomplicated, lasting 10 hours with a short second stage resulting in delivery of a live female infant weighing 2500 g. The total blood loss was weighed by the doula at 400 mL and had settled after delivery. The placenta was brought in and on examination appeared complete. The patient had developed worsening abdominal pain after delivery prompting referral to the hospital. There was limited antenatal care during this pregnancy. She had one previous uncomplicated spontaneous vaginal birth at term and no history of other pregnancies. Apart from mild haemophilia A, there was no significant surgical or medical history. On arrival the patient was haemodynamically unstable and drowsy. Heart rate was 100 and blood pressure was 70/30. Two large bore cannulas were inserted and intravenous fluid resuscitation was initiated immediately. The abdomen was soft with the fundus firm, central and contracted below the umbilicus. The uterus was explicitly tender to touch in the suprapubic region. Speculum examination showed a second-degree perineal tear that was not actively bleeding. There was no active bleeding in the vault and no other signs of vaginal or cervical trauma. A digital vaginal examination was performed which was unremarkable and did not expel any blood clots or placental tissue. An in-dwelling catheter was inserted at this time which drained clear urine. Investigations The haemoglobin level was 7.7 g/dL and a unit of packed red blood cells was administered. Platelets were 301×109/L and coagulation studies were normal. Due to acuity of the situation factor VIII levels were not performed. Urgent formal ultrasound was performed at the bedside which revealed a well contracted, empty uterus with a large area of mixed echogenicity measuring 14.3×11.3×12.0 cm anterior to the uterus, indicating a haematoma >1 L in volume (figure 1). There was significant free fluid in the abdomen. Differential diagnosis The differential diagnosis includes broad ligament haematoma, supralevator haematoma, intraperitoneal haematomas, uterine rupture and undetected cervical or vaginal tears. When presented with concealed postpartum haemorrhage, differential diagnosis should also include retroperitoneal haematomas including uterine/splenic/renal/adrenal/ovarian artery rupture, renal angiomyolipomas, malaria in pregnancy, bleeding disorders, hepatic rupture, spontaneous rupture of vessels due to AV-malformation, endometriosis causing spontaneous rupture of uterine and ovarian vessels and Retzius space haematomas.1 Treatment Exploratory laparotomy was performed by two obstetric consultants who revealed a haemorrhagic infiltration in the Retzius space. Several large blood clots were removed allowing visualisation of a large haematoma which infiltrated beneath the anterior serosa of the bladder, through the left broad ligament and into the retroperitoneal space (figure 2). The bladder was obscured by the large adherent subperitoneal haematoma which by this stage had clotted, causing thickening of the anterior bladder wall. This was unable to be evacuated due to risk of damage to the bladder. Instead, the bladder was distended with 400 mL normal saline to confirm its position and integrity. The urine remained clear throughout the procedure. Due to the inherent difficulty of this case as a result of local anatomy distortion and extensive haematoma infiltration, the general surgical consultant was called to provide assistance in theatre. The uterus, ovaries, tubes, broad ligaments and uterine arteries were identified and intact. Injury to the liver and spleen was excluded. Active bleeding was seen anterior to the bladder in the Retzius space, causing ongoing difficulty with visualisation of the surgical field. Consideration was given to performing a uterine artery ligation or hysterectomy, however, due to the location of the active bleeding, this was thought to be futile as uterine vessels were unlikely to be involved. The source of the bleeding was initially thought to be a deep musculoserosal tear of the anterior vaginal wall, however, vaginal examination confirmed an intact vaginal mucosa above the second-degree perineal tear. The source was later identified as a ruptured vessel in the plexus of Santorini, which was seen to be actively bleeding and oversewn with haemostatic sutures. Due to continuous ooze, likely related to mild haemophilia, FLOSEAL was applied for haemostasis and the abdomen was packed with three saline soaked abdominal packs. The perineal tear was repaired and the patient was then transferred to a tertiary hospital with angiogram facilities. Total estimated blood loss at this time was 3.5 L. A total of 2 units packed red blood cells and 3 units fresh frozen plasma were administered. Outcome and follow-up The patient remained stable overnight. A CT angiogram the following day revealed no active bleeding vessels. A relook laparotomy was performed which also confirmed haemostasis and identified no further injury. A large contained haematoma was again identified over the anterior bladder wall, tracking into the left broad ligament, left side wall and left anterior abdominal wall. Due to significant clotting and haemostasis, this was not evacuated. The abdominal packs were removed and the wound was closed. The patient made a full recovery and was discharged a few days later. On review 4 weeks postoperatively, she was fit and healthy with no abdominal discomfort or tenderness. Review of her medical history revealed no evidence of clinical haemophilia.

Women presenting in shock post partum should prompt a thorough examination for concealed and revealed haemorrhage. In this case, there was no evidence of external trauma, no evidence of retained products or atony and no external bleeding which posed an initial diagnostic dilemma. Severe suprapubic pain and clinical shock out of proportion to estimated blood loss prompted an urgent ultrasound which revealed a large clot anterior to the uterus. This leads to urgent surgical intervention which ultimately identified a retroperitoneal haematoma secondary to bleeding in Retzius space. Retzius space contains loose connective tissue and fascia posterior to the pubic symphysis and anterior to the bladder. Retzius space haematomas are a rare occurrence, primarily associated with urogynaecological surgeries such as tension-free vaginal tape.3 They are even more uncommon in obstetrics, with only two other cases following spontaneous vaginal delivery reported in the literature.4 5 Management of this condition is extremely difficult due to distortion of the local anatomy by extensive haematoma infiltration causing limited visibility and difficulty identifying key anatomy. Having a low threshold for involving the general surgeons is recommended. In this case, haemostasis was achieved with haemostatic sutures to the plexus of Santorini, FLOSEAL and packing. Other cases report success with bilateral uterine artery and internal iliac artery ligation,4 and drainage of the haematoma.5 Haematoma in the Retzius space is thought to occur due to rupture of the plexus of Santorini, the vessels of which may be weakened during pregnancy and damaged during descent and delivery of the fetal head.2 Various coagulopathies are also implicated as predisposing factors, and indeed this patient had mild haemophilia A which likely contributed to the extent of haematoma and the failure to control haemorrhage with haemostatic sutures alone.6 Due to limited antenatal care and the acuity of presentation, factor VIII levels were unable to be measured, and therefore, it is unknown how much this affected the presentation. Learning points. Concealed postpartum haemorrhage should be considered in the differential diagnosis of postpartum shock. Retroperitoneal haematomas including Retzius haematomas are a rare but life-threatening cause of concealed postpartum haemorrhage. Retzius space haematomas may be very difficult to treat and it is important to have a low threshold for surgical team involvement. Women who are carriers of haemophilia are at increased risk of intra-abdominal postpartum haemorrhage. Footnotes Contributors: MLA authored the initial draft of the case report. MS was extensively involved in the editing process. Both authors contributed to the final editing, approval and submission of the case report. Both authors agreed to be accountable for the article and to ensure that all questions regarding the accuracy or integrity of the article are investigated and resolved. Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors. Competing interests: None declared. Patient consent: Obtained. Provenance and peer review: Not commissioned; externally peer reviewed.

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