Journals | Policy | Permission

Journal of Hematology

Case Report

Volume 2, Number 1, June 2013, pages 40-41

Spontaneous Hemoperitoneum in Chronic Myeloid Leukemia: An Unusual Etiology

Corpus luteal cyst hemorrhage (though common in child bearing women), is a rare cause of spontaneous hemoperitoneum in patients of chronic myeloid leukemia in reproductive age group. It also mimics ectopic pregnancy, spontaneous splenic hemorrhage in symptomatology along with other causes of spontaneous hemoperitoneum.

Multiparous woman (32 years old) with three previous normal deliveries, presented with mild vaginal bleeding and acute pain abdomen since two days. Her last menstrual period was twenty days back. There was no history of trauma. Patient was in early hypovolemic shock (pulse rate-110/minute, blood pressure-90/70 mmHg, arterial blood gases were within normal limits). Abdomen was distended and tender. Shifting dullness was elicited. Spleen was enlarged till umbilicus but non tender. Pelvic examination revealed minimal bleeding and normal sized uterus. Cervical motion tenderness and left adnexal fullness was present. However urine pregnancy test was negative. She was resuscitated with intravenous fluids and one packed cell transfusion. Her Haemoglobin was 8.3 g/dL, TLC- 1.28 × 10⁵/µL (Leucocytosis). Peripheral smear showed Blastocyte-2 Promyelocyte-3, Myelocyte14, Metamyelocyte10, Polymorphs-58, Lymphocyte3, Monocyte-3Eosinophil-5Basophils2. Nucleated RBC was 2/100 white blood cells. Beta-hCG was 0.5 mIU/mL. Based on above haematological and urinary findings, diagnosis of chronic myeloproliferative disorder with splenic haemorrhage was considered. Emergency ultrasonography and Doppler revealed enlarged spleen without any site of rupture. Hemoperitoneum was noted. An empty normal sized uterus with echogenic lesion (3.8 × 2.7 cm) in left ovary suggestive of corpus luteal cyst was noted. Hence a final diagnosis of corpus luteal hemorrhage with myeloproliferative disorder was made. Conservative management with back up of laparotomy in case of worsening shock was planned. She received hydroxyurea, allopurinol and hemostatics (Tranexamic acid). On day 7 hemoperitoneum resolved completely and need for laparotomy was obviated. Patient was referred to our haem-oncology unit.

Reverse transcriptase Polymerase-chain-reaction testing of peripheral-blood RNA revealed Bcr-abl rearrangement confirming chronic myeloid leukemia (CML). Patient was started on imatinib with contraceptive advice and disease was under remission. She conceived while on imatinib due to failed barrier contraception and presented to our antenatal clinic at 28 weeks. As she was past teratogenic age, imatinib was continued. Level 2 sonography ruled out congenital anomalies in foetus. She went into spontaneous labor and delivered healthy term female baby vaginally.

Chronic myeloid leukemia (CML) is a clonal myeloproliferative disorder arising from single pluripotent hemopoietic stem cell due to acquired genetic change namely Bcr-abl gene rearrangement, owing to balanced reciprocal translocation involving chromosomes 9 and 22 [1]. Fifty percent of patients have splenomegaly. Thrombotic and hemorrhagic complications such as atherothrombosis, venous thrombosis, rarely gastrointestinal hemorrhage and splenic hemorrhage do occur in CML. Spontaneous hemoperitoneum is rare in CML. It is defined as blood within peritoneal cavity without traumatic cause. Imaging is required for diagnoses. Spontaneous splenic rupture has been reported worldwide as a cause of massive hemoperitoneum in CML patients leading to acute abdomen with varying degrees of shock and even death [2].

Ovarian cyst hemorrhage in CML had been first described by Valentsik in 1951 [3]. However, no data regarding corpus luteal hemorrhage in CML have been reported till date. Ruptured corpus luteal cyst is a common in women of reproductive age group. Symptoms range from being asymptomatic to features of an acute abdomen. In normal menstrual cycle, a mature ovarian follicle ruptures, releasing an ovum. Occasionally, this rupture site may bleed, causing acute abdominal pain with varying degrees of shock. Etiology is unknown, although abdominal trauma and congenital bleeding disorders may increase the risk. Diagnosis is by pelvic ultrasound. Treatment can be either conservative or surgical [4]. This is a first reported case of spontaneous hemoperitoneum following corpus luteal haemorrhage in CML patient till date, best to our knowledge. Women with CML if presents with spontaneous hemoperitoneum, corpus luteal hemorrhage should be a differential diagnoses apart from splenic hemorrhage. Our patient belonged to reproductive age group who was managed conservatively without surgical intervention. She conceived spontaneously and had live birth without congenital anomalies while on imatinib.

Corpus luteal cyst hemorrhage should be considered as a possible etiology in women of reproductive age group with CML presenting with spontaneous hemoperitoneum in absence of positive urine pregnancy test. Conservative approach prevents morbidity associated with CML and is successful. Imatinib may not cause congenital anomaly if women conceive while on therapy though more studies are needed to substantiate it [5].

Conflict of Interest

No conflicts to disclose.

Funding Source

None.

1. Mughal TI, Goldman JM .Chronic Myeloid Leukemia: The Value of Tyrosine Kinase Inhibition. American Journal of Cancer. 2003; 2(5): 305-311.
   doi
2. Bulus H, Koyuncu A, Yildiz M, Coskun A, Koklu S, Akbal E. Chronic myeloid leukemia presenting with spontaneous splenic rupture. Am Surg. 2012;78(1):E28-29.
   pubmed
3. Valentsik F. [Profuse ovarian hemorrhage in chronic myelocytic leukemia]. Bratisl Lek Listy. 1951;31(3-4):416-421.
4. Raziel A, Ron-El R, Pansky M, Arieli S, Bukovsky I, Caspi E. Current management of ruptured corpus luteum. Eur J Obstet Gynecol Reprod Biol. 1993;50(1):77-81.
   doi
5. Apperley J. Issues of imatinib and pregnancy outcome. J Natl Compr Canc Netw. 2009;7(10):1050-1058.
   pubmed

This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Journal of Hematology is published by Elmer Press Inc.