Haemorrhagic ascites: An atypical presentation of endometriosis

In: Journal of Clinical and Scientific Research · 2024 · vol. 13(2) , pp. 144–146 · doi:10.4103/jcsr.jcsr_216_22 · W4399700513
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This case report describes a rare instance of haemorrhagic ascites in a 26-year-old woman, which was ultimately diagnosed as ovarian endometriosis and treated successfully with oral dienogest.

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This case report describes a 26-year-old multiparous woman who presented with acute abdominal pain and severe anemia due to massive haemorrhagic ascites. Diagnostic imaging identified bilateral ovarian endometriotic cysts, and the patient was successfully treated with medical management using oral dienogest, leading to the complete resolution of the ascites and endometrial masses without surgical intervention. The authors note that while this presentation is rare and typically associated with nulliparous women, it highlights the importance of considering endometriosis in differential diagnoses for unexplained ascites to avoid unnecessary invasive procedures. This paper is centrally about endometriosis — specifically documenting a rare case of haemorrhagic ascites caused by ruptured ovarian endometriotic cysts.

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Abstract

Abstract Haemorrhagic ascites is primarily associated with perforated abdominal viscera, malignancies and tuberculosis or hepatic pathologies. However, its presentation due to ovarian endometriosis is a rare finding. We report the rare occurrence of haemorrhagic ascites due to ovarian endometriosis in a 26-year-old multiparous female presentingwith severe abdominal pain. The other probable causes of ascites were excluded and computed tomography of abdomen and pelvis revealed moderate ascites with bulky left ovarian cyst. Abdominal paracentesis revealed haemorrhagic ascitic fluid which on cytopathology did not reveal evidence of malignancy. Diagnostic work-up including magnetic resonance imaging and diagnostic laparoscopy confirmed the diagnosis. She was treated with oral dienogest which caused regression of the mass and pain. This case highlights that endometriosis may be included in the differential diagnosis while treating haemorrhagic ascites in women in reproductive age.
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Introduction

The development of ascites is primarily due to hepatic pathologies, congestive cardiac failure or renal diseases such as nephrotic syndrome. Pelvic diseases including ovarian cancers, pelvic tuberculosis and Meig’s syndrome are also known to cause ascites. However, endometriosis in association with ascites is a rare finding. Endometriosis is an oestrogen-dependent disorder and generally characterised by pelvic pain, dysmenorrhoea, dyspareunia and tenderness in the pelvis or infertility. Rarely, it is known to be the cause for ascites with haemorrhagic type. It was first reported in 1954, and since then, around 60 cases have been reported.[,] It was primarily seen in young nulliparous African females. However, few studies have shown no significant association between parity and incidence of endometriosis.[,] For such cases, evaluation to rule out the other probable diagnosis helped reach the final cause of endometriosis. We report the rare occurrence of haemorrhagic ascites due to ovarian endometriosis in a 26-year-old multiparous female. CASE REPORT A 26-year-old multiparous woman (para 2, live birth 2, both delivered by caesarean section) was referred from a private hospital to the emergency department with complaints of acute abdomen pain which was under evaluation for 3 days. She had nausea and constipation with no complaints of vomiting. The patient had developed sudden onset of pain in the abdomen with distension 3 days back. At the private hospital, she was evaluated for ascites. The β-human chorionic gonadotropin (β-hCG) was <2 mUI/mL and urine pregnancy test was negative, so ectopic pregnancy was ruled out. She was also operated on for appendicectomy 8 years ago and had undergone bilateral tubal ligation 2 years ago. Seven years ago, inguinal lymph node biopsy had revealed features suggestive of tuberculosis (TB) for which the patient had received anti-TB treatment for 6 months. Abdominal computed tomography with contrast (CECT) done at the private hospital had revealed moderate ascites, few air-fluid levels and bulky left ovary with the cyst. The patient also underwent abdominal paracentesis and 60 mL of haemorrhagic fluid was drained. As the haemoglobin level (5.8 g/dL) was low two units of packed red blood cells (PRBCs) were transfused. Paracentesis was performed to drain the haemorrhagic fluid. An exploratory laparotomy was advised for which she was referred to our tertiary healthcare centre. On admission, severe pallor was noted. The abdomen was soft with no guarding or rigidity but with tenderness in the left iliac fossa and with hypoactive bowel sounds. Per vaginal examination showed a bulky uterus with posterior forniceal tenderness (+++) and brownish discharge. Laboratory testing had revealed low haemoglobin (7.4 g/dL); aspartate aminotransferase 68 IU/L, alanine aminotransaminase 106 IU/L, international normalised ratio 1.3, and D-dimer 2.3 ng/mL. Ascitic fluid analysis showed proteins 3.96 g/dL (albumin 2.34 g/dL); glucose 35.3 mg/dL, serum total protein 5.3 g/dL, albumin 2.8 g/dL, serum albumin ascitic fluid albumin gradient (SAAG) 1.3, total bilirubin 0.7 mg/dL, serum total protein 5.3 g/dL, albumin 2.8 g/dL, alkaline phosphatase 108 U/L, red blood cells >100,000/mm3, total count 2200/mm3 (with polymorphs 65% and lymphocytes 35%) packed cell volume (23.5%); ascitic fluid adenosine deaminase was 16 IU/L. Cancer antigen (CA125) was borderline elevated (29IU/mL). Patient and family were counselled for laparoscopy, as required. Magnetic resonance imaging (MRI) which showed cystic collection at inferomedial to the left ovary measuring 2.7 cm × 2.4 cm × 1.9 cm in size and the right ovarian endometriotic cyst measuring 8.1 cm × 6.9 cm × 6.2 cm. These observations of a tubo-ovarian mass (T-O mass) were suggestive of endometriosis. The sagittal section images also suggested massive haemorrhagic ascites with the presence of T-O mass (Figure 1) and the constellation of findings raised the possibility of the ruptured left endometriotic cyst with pelvic haematoma. After the initial diagnostic abdominal paracentesis, therapeutic paracentesis was done. This had revealed haemogrrhagic ascitic fluid. After the therapeutic abdominal paracentesis, abdominal pain had reduced, and clinically, she had become better. Repeat MRI showed no or minimal ascites after the procedure. Oral dienogest 2 mg/day was initiated and administered for one year. MRI was repeated at 3, 6 and 12 months, which revealed no endometriotic masses and showed complete resolution of disease. Ascites was slowly resolved and eventually disappeared documented in follow-up imaging.

Discussion

Endometriosis is commonly detected in 10%–15% of females in the age group of 25–44 years.[] However, very few cases of endometriosis are reported to be the cause of haemorrhagic ascites. In endometriosis, the endometrial glands with stroma are detected outside the uterus such as in the peritoneum or as cysts on the ovary. Haemorrhagic ascites is mainly found with malignancies such as hepatoma, ovarian cancer and injury to abdominal viscera such as ruptured spleen or in tuberculosis or ectopic pregnancy. In our patient β-hCG was <2 mIU/mL and urine pregnancy test was negative; hence, ectopic pregnancy was ruled out. The exact cause for the development of ascites in endometriosis is unclear, but it has been proposed that the rupture of chocolate cysts causes the blood and endometrial tissue to enter the peritoneal cavity. Another possibility is the obstruction of subdiaphragmatic lymphatics or retrograde menstruation is also proposed for the development of haemorrhagic ascites with endometriosis. Endometriosis associated with ascites was first described in 1954, and since then, more than 60 cases have been reported in the literature. The majority of cases are seen in nulliparous (82%) females with an average age of 27 years.[] Clinical presentation includes abdomen or flank pain, distension, anorexia, weight loss, menometrorrhagia or irregular menses. On physical examination, the pelvic mass may be palpable along with the signs of shock in case of severe haemorrhagic ascites. In few cases, pleural effusion (38%) was also reported along with endometriosis-associated haemorrhagic ascites indicating extensive spread and severity of disease.[] Few cases have reported haemorrhagic shock, which can be life-threatening.[] Such acute presentation is indicative of ovarian cancer, which can be differentiated by performing tumour markers (CA-125) which will be negative, but it has low specificity for detecting endometriosis. Laparoscopy can be the final technique to evaluate the cause in case of an unknown aetiology. The other differential diagnosis of haemorrhagic ascites is rupture of ovarian cyst, spleen or other abdominal viscera, ovarian cancer, ectopic pregnancy, tuberculosis or Meig’s syndrome, which can be differentiated by imaging techniques. Considering that the patient may land in haemorrhagic shock, it is essential to resuscitate with blood products such as PRBC after draining the fluid through the paracentesis. Definitive treatment is the surgical excision of the endometrial tissue or hysterectomy with the removal of both ovaries. However, considering fertility, medical management with hormonal therapy, including progestin, progestin and oestrogen combination, depomedroxyprogesterone acetate, danazol or leuprolide, which reduces the secretion of follicle-stimulating hormone and luteinising hormone, which suppresses the growth of ectopic endometrial tissue can be advised. Long-term follow-up is recommended as the chances of recurrence are high. Several cases of recurrence of haemorrhagic ascites associated with endometriosis have been reported requiring frequent intervention and long-term medical management. After undergoing unilateral oophorectomy or cystectomy for recurrent ascites-associated endometriosis more than 50% of cases experienced a reported recurrence; however, in patients with bilateral salpingo-oophorectomy, no recurrence was reported.[] In patients with recurrence, malignancy should be suspected, and therefore, regular monitoring is crucial to identify such changes at the earliest. The present case was of a young but multiparous female with no menstrual disturbance or past relevant medical history, which might precipitate in causing endometriosis-associated haemorrhagic ascites. After ruling out the other probable diagnosis and imaging results, the decision to start medical management was made that not only avoided laparoscopy but also helped in the complete regression of mass and pain. Haemorrhagic ascites can be a life-threatening complication of endometriosis; hence, prompt diagnosis and early management can minimise the severity of the disease. Clinicians should be aware that endometriosis can be a cause of haemorrhagic ascites in women of child-bearing age. Imaging and laboratory workup help to rule out malignancies, TB and perforation or rupture of viscera is crucial. The involvement of the pleural cavity should also be evaluated, as certain cases have been reported in the literature. Medical management can yield good results, but in cases of recurrence, surgical intervention with regular follow-up is recommended. Declaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.

References

1. Gungor T, Kanat-Pektas M, Ozat M, Zayifoglu Karaca M. A systematic review:Endometriosis presenting with ascites. Arch Gynecol Obstet 2011;283:513–8.2. Wang X, Li Y, Tong J, Chang B, Zhang Y, Liu Y, et al. Endometriosis presenting with recurrent massive hemorrhagic ascites and diagnosed by core needle biopsy:A case report. Medicine (Baltimore) 2019;98:e15477.3. Liu X, Long Q, Guo SW. Surgical history and the risk of endometriosis:A hospital-based case-control study. Reprod Sci 2016;23:1217–24.4. Saha R, Kuja-Halkola R, Tornvall P, Marions L. Reproductive and lifestyle factors associated with endometriosis in a large cross-sectional population sample. J Womens Health (Larchmt) 2017;26:152–8.5. Parasar P, Ozcan P, Terry KL. Endometriosis:Epidemiology, diagnosis and clinical management. Curr Obstet Gynecol Rep 2017;6:34–41.6. Gonzalez A, Artazcoz S, Elorriaga F, Timmons D, Carugno J. Endometriosis presenting as recurrent haemorrhagic ascites:A case report and literature review. Int J Fertil Steril 2020;14:72–5.

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