Ovarian Vein Thrombosis: AnExceedingly Rare and Elusive

Postpartum Complication

Katherine Routson, OMS-IIIA.T. Still University School of Osteopathic Medicine in Arizona

5850 E. Still CircleMesa, AZ 85206

sa200318@atsu.edu

Dr. Davinder Mann, MD, MPHObstetrics and Gynecology

Adelante Healthcare Goodyear13471 W. Cornerstone Blvd

Goodyear, AZ 85395dmann@adelantehealthcare.com

ABSTRACT_______________________________________________

Ovarian vein thrombosis is a rare postpartum complication that typically presentswithin the first 10 days after birth as nondescript symptoms of abdominal or flank painas well as fever and leukocytosis which can mimic other acute abdominal pathologiesincluding appendicitis, renal colic, pyelonephritis, and endometritis.1 It is estimated thatpain will occur on the right side 90% of the time due to length of the right ovarian vein ascompared to the left.2 Furthermore, the estimated postpartum occurrence following avaginal delivery is a mere 0.05-.18% with the frequency being higher following cesareandelivery.2 Based on the rarity, this can be a difficult diagnosis to make. Being thatendometritis is much more common, ovarian vein thrombosis may go undiagnosed. Thiscan be problematic as patients with ovarian vein thrombosis are at risk of pulmonaryembolism.

This case involves a 35 year old G5 P2 A2 woman who was diagnosed with aright-sided ovarian thrombosis two days after an uncomplicated spontaneous vaginaldelivery. The patient had routine prenatal care as well as antenatal testing due toadvanced maternal age. The location and severity of pain did not correlate with typicalpain after a vaginal delivery. Taking this into consideration as well as the presence offever, an abdominal CT was performed which clearly showed the diagnosis (Figure 1).

An important discussion arises from this case in regards to treatment. Thereremains a debate as to whether or not to anticoagulate women with ovarian veinthrombosis because the thrombus can resolve on its own. However, there are numerousadverse and sometimes even fatal outcomes associated with ovarian vein thrombosiswhich makes the decision of anticoagulation a critical choice.

INTRODUCTION____________________________________________

Aside from being seen in the postpartum period, ovarian vein thrombosis (OVT)can also be associated with malignancy, recent surgical procedures, and pelvicinflammatory disease.3 Over the years several theories have evolved as to why awoman might be at increased risk of developing an OVT in the postpartum period.These theories include but are not limited to the following: venous stasis which iscommonly seen in pregnancy, endometritis, and an increase in Von Willebrand factorand clotting factors.3 Furthermore, it has been noted that the ovarian vein becomesthree times as large in diameter during pregnancy. After delivery of the baby, the ovarianvein blood flow decreases which can contribute to venous stasis. 3

Although the most common presentation of OVT is a triad of vague abdominalpain, fever, and potentially an abdominal mass, many other symptoms can occurincluding: tachycardia, tachypnea, nausea, vomiting, hypotension, and rarely, positive

blood cultures.3 The current literature suggests that a significant number of patients whodevelop OVT may have a tendency to clot from disorders including Factor V Leidenmutation, protein S deficiency, and antiphospholipid syndrome.3 These disorders arealso causes of recurrent pregnancy loss.

The best modality for diagnosis of OVT has also been a long-standing debatewith CT, MRI, and laparotomy all being viable options.3 Laparotomy remains the goldstandard but is also the most invasive technique and therefore is less frequently used.In this case a CT scan was utilized which clearly demonstrated the thrombus.Osteopathic tools that may aid in diagnosis and decision making include viscerosomaticreflexes and Chapman's points. It would be expected to find tissue texture changes atthe levels of T10-T11 which correspond to the ovaries.4 A small palpable area of roundtissue texture abnormality would most likely be palpated anteriorly at the superior pubicramus just lateral to the pubic symphysis as well as posteriorly at T10 which bothcorrespond to the ovaries.5

Making the accurate diagnosis of ovarian vein thrombosis is crucial to thepatient’s health because OVT is associated with many adverse outcomes. Suchoutcomes include: pulmonary embolism, infiltration of the inferior vena cava, ovarianinfarct, ovarian abscess, uterine necrosis, septic thrombophlebitis, and ureteralcompression.3 Current literature suggests that the rate of associated pulmonaryembolism may be as high as 13-25%.3

This case merits reporting to further the discussion of when/if to anticoagulateand how to best make that decision while also empowering the patient to participate intheir care.

CASE PRESENTATION________________________________________________

A 35 year old G5 P2 A2 woman with a history of obesity, PCOS, and two priorspontaneous abortions who is otherwise healthy presented to an emergencydepartment complaining of excruciating right-sided lower abdominal pain with radiationto the right groin two days after spontaneous vaginal delivery at 38.3 weeks withoutpostpartum hemorrhage or other complications. She was febrile in the emergency room.Unfortunately, the facility was not a location that her obstetrician group has privileges at.She was therefore transferred to another facility where her obstetrician saw her in theemergency room. On the initial exam in the second emergency room, the patient wasextremely uncomfortable and appeared in moderate distress. She was no longer febrile,but was receiving Tylenol. Vital signs were otherwise within normal limits. Acardiovascular exam demonstrated regular rate and rhythm, no murmurs and normalS1/S2. Pulmonary exam showed normal effort, no adventitious breath sounds, andgood air movement throughout. Abdominal exam revealed extreme tenderness to lightpalpation of the right lower quadrant and diffuse tenderness throughout. There was no

erythema, warmth, or swelling of the skin. An exam of the right thigh and groin showedno color changes, warmth, or edema. The CT scan showed multiple pelvic varices withdilatation of the right ovarian vein, inflammation surrounding the ovarian vein withlow-density area near its junction with the inferior vena cava suspicious for thrombus(Figure 1). The patient was admitted to labor and delivery for observation, treatment,and pain control. She was also evaluated by the medicine team and started onLovenox. Admission lab data revealed an elevated white blood cell count at 12.9 withneutrophil count of 9.5, normal LFTs, normal creatinine, urine with 10-20 red blood cellsper high power field, INR of 0.96, PT 10.0, and PTT 22.5. Patient was discharged thefollowing day and eventually transitioned to Coumadin as an outpatient. She was seenin the office for follow-up two weeks later at which time her pain had improved and shewas feeling well. She had no issues with breast-feeding, no evidence of mastitis,abnormal vaginal bleeding, urinary symptoms, or bowel issues. She was scheduled tofollow-up with her PCP for Coumadin management.

The patient then returned to the clinic for obstetric follow-up the next weekbecause she reported recurrence of right lower quadrant pain, however, she noted thatit was much less severe than when she was originally diagnosed with an OVT. Shedenied fevers, chills, shortness of breath and chest pain. Her most recent INR wassubtherapeutic at 1.5 therefore Coumadin was increased from 5 mg to 7.5 mg. Anabdominal CT with intravenous contrast was ordered to assess for any changes in thethrombosis. Results of the abdominal CT showed midline anteverted uterus with mildthickening of the endometrial lining that likely represented secretory endometriummeasuring approximately 17 mm in the uterine fundus, normal adnexal structures, 1.2mm nonobstructing left interpolar renal calculus, and miniscule size umbilical hernia thatcontained fat. No evidence of ovarian thrombus was visualized. Patient continued onCoumadin.

She was last seen for routine postpartum examination two weeks later at whichtime a postpartum depression screening was completed and was negative. The patientdenied any issues with mood or risk for domestic violence as well as feeding difficulties,nursing difficulties, bowel or bladder issues, or breast issues. She will continueCoumadin until the end of March, 2021.

The patient had regular prenatal care without complications during thispregnancy. Her routine first trimester screening was unremarkable. Patient’s anatomyscan at 18.6 weeks revealed a single viable intrauterine pregnancy, normal anatomy, noplacenta previa, and adequate cervical length. Patient did not have evidence ofgestational diabetes. Past surgical history included an ovarian cystectomy. Patient hasno known history of clotting disorders. She is a former smoker with a 1.5 pack yearhistory. Prior to this pregnancy, the patient had two prior full-term spontaneous vaginaldeliveries along with the two spontaneous miscarriages. She did not undergo additional

workup after the second miscarriage. Current daily medications include Coumadin 7.5mg and Ortho Micronor 0.35 mg tablet. Patient has no known drug allergies.

DISCUSSION____________________________________________

This diagnosis is not only interesting based on its rarity and evasiveness, butalso because there is no standard treatment protocol to date. Making the diagnosisrequires an astute clinician that includes OVT on their differential diagnosis for acuteabdominal pain in females as it often mimics other pathology. Possible treatmentoptions include anticoagulation, antibiotics, thrombectomy, ovarian vein ligation, ovarianvein excision, and inferior vena cava (IVC) filter.3 Furthermore, there are no firmrecommendations on how long to continue anticoagulation, however, some sourcessuggest continuation for at three months if the cause of the thrombus is identified, andlonger if it is idiopathic.3 Acceptable antibiotic regimens if a septic thrombophlebitis issuspected includes ampicillin-sulbactam, piperacillin-tazobactam, ticarcillin-clavulanate,and ceftriaxone + metronidazole.3 The primary goal of anticoagulation is to prevent thedevelopment of a pulmonary embolism (PE), although the rate of subsequent PEdevelopment is also widely debated in the literature with estimates ranging from 4% to25%.3

Perhaps the largest study investigating treatment of OVTs was a retrospectivestudy of the Penn State Medical center in which both inpatient and outpatient recordsfrom 2010-2015 were searched for any diagnosis that included a thrombus andanalyzed to identify ovarian vein thrombosis specifically. A total of 50 patients from 1436qualifying records were found to have OVT. 30 of the patients were treated withanticoagulation alone, three were treated with anticoagulation and antibiotics, two hadIVC filters placed, one was treated with aspirin, 12 were left untreated, and two wereunknown. The average length of follow-up was 23.7 months and the average length ofanticoagulation was 13.2 weeks. 12 of the patients had follow-up imaging, with 10patients showing radiologic resolution after anticoagulation. Two patients had resolutionwithout treatment. Seven of the patients were diagnosed with other deep veinthrombosis or pulmonary embolism at the time of OVT diagnosis.

The retrospective study concluded that there was no statistically significantrelationship between treatment and patient outcomes.5 Furthermore, the authorsdetermined that symptomatic OVT, septic thromboembolism, and cases of OVT withcoexisting DVT or PE may warrant anticoagulation, but OVTs that are discovered asincidental findings do not necessarily require anticoagulation.3 There have otherwisebeen no studies that have drawn a concrete conclusion as to whether or not patientswho are anticoagulated as a treatment for OVT have better outcomes than those whoare not. Furthermore, there are only a handful of studies examining treatment of OVT

with most being an extremely small sample size or individual care reports. Thus,treatment remains a topic that is debated among clinicians and is often decided basedon clinical judgement, the patient’s history/risk factors, as well as patient preference torespect autonomy.

OSTEOPATHIC CONSIDERATIONS____________________________________________

Although osteopathic manipulative techniques were not utilized due to thepatient's extreme pain with movement, the osteopathic tenets were utilized throughouther care. Respecting the first tenet of osteopathy which is to recognize that a person isa unit of body, mind and spirit was salient. This patient had given birth just days prior toher diagnosis of OVT and was subsequently temporarily separated from her newbornwhile receiving hospital care during a pandemic. Additionally, she was experiencingsignificant pain. Accounting for this major stressor, she was admitted to labor anddelivery where breast pumping accommodations are easily accessible and the patientwas already familiar with the nursing staff and unit. Additionally, the patient wasdischarged with close follow-up to monitor for any complications and to ensure she isbonding well with her baby and not having any issues with postpartum mood. Byappreciating the impact that physical pain can have on one’s mind as well as the fearthat may come from receiving a rare diagnosis, we can help the patient to feelwell-supported and empowered to play an active role in their healing process.

ACKNOWLEDGEMENTS____________________________________________Thank you very much to Dr. Davinder Mann, MD, MPH, for not only guiding my learningthroughout this case and helping me acquire the data for this presentation, but for beinga wonderful mentor who is invested in training the next generation of physicians.

ABBREVIATIONS____________________________________________OVT: ovarian vein thrombosisCT: computed tomographyMRI: magnetic resonance imagingPCOS: polycystic ovarian syndromeLFTs: liver function testsINR: international normalized ratioPT: prothrombin timePTT: partial thromboplastin timePCP: primary care provider

REFERENCES____________________________________________1. Ovarian Vein Thrombosis Diagnosis, Management Tips for Emergency Physicians.ACEP Now.https://www.acepnow.com/article/ovarian-vein-thrombosis-diagnosis-management-tips-emergency-physicians/. Published April 9, 2019. Accessed January 22, 2021.

2. Kodali N, Veytsman I, Martyr S, Lu K. Diagnosis and management of ovarian veinthrombosis in a healthy individual: a case report and a literature review. Wiley OnlineLibrary. https://onlinelibrary.wiley.com/doi/full/10.1111/jth.13584. Published February 6,2017. Accessed January 22, 2021.

3. Plastini T, Henry D, Dunleavy K. Ovarian vein thrombus: to treat or not to treat? Bloodadvances. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5728322/. Published June 22,2017. Accessed January 22, 2021.

4. Viscerosomatic Reflexes . American College of Osteopathic Physicians .https://www.acofp.org/acofpimis/acofporg/apps/omt/materials/downloads/sdofm_viscerosomatic_reflex_pocket_chart.pdf. Accessed January 22, 2021.

5. Doane CB. OPP Board Review for AOBFP Certification . MSU.Edu.https://scs.msu.edu/media/2013-02-06%20AOBFP%20Cert/OPP%20Board%20Review%20for%20AOBFP%20Certification-%20Handout%203%20slides%20a%20page%20and%20notes.pdf. Published February 6, 2013. Accessed January 22, 2021.

GRAPHIC ELEMENTS____________________________________________

Figure 1. Abdominal CT with IV contrast illustrating right ovarian vein thrombosis