32

CASE REPORT

DOI 10.4070 / kcj.2009.39.1.32 Print ISSN 1738-5520 / On-line ISSN 1738-5555

Copyright ⓒ 2009 The Korean Society of Cardiology

Spontaneous Retroperitoneal Hemorrhage and Hemothorax After Intravenous Heparin Treatment Do-Hoei Kim, MD1, Seung Jin Lee, MD2, Ung Jeon, MD2, Sang-Ho Park, MD2, Se-Hwan Lee, MD2, Won-Yong Shin, MD2 and Dong-Kyu Jin, MD2 1Division of Cardiology, Department of Internal Medicine, Cheongju St. Mary’s Hospital, Cheongju, 2Division of Cardiology, Department of Internal Medicine, Soonchunhyang University Cheonan Hospital, Cheonan, Korea ABSTRACT

Spontaneous retroperitoneal hemorrhage is a rare complication after percutaneous coronary intervention (PCI). The patient can be in danger if bleeding is not stopped immediately. However, it is not easy to control the bleeding completely because the bleeding foci can be multiple and there is a rich network of collateral circulation. We report a case of spontaneous retroperitoneal hemorrhage successfully treated using multiple microcoils. One year later, pan-hypopituitarism occurred as a likely consequence of the accompanying hypovolemic shock. (Korean Circ J 2009;39:32-36) KEY WORDS: Retroperitoneal; Hemorrhage; Hemothorax; Heparin; Panhypopituitarism.

Introduction

Spontaneous retroperitoneal hemorrhage occurs in a

variety of clinical circumstances, including tumors, he-modialysis, polycythemia vera, percutaneous interven-tions, and anticoagulation treatment.1-4) Surgical treat-ment is indicated if the patient remains unstable despite adequate fluid and blood resuscitation. Recently, there have been many reports of successful treatment by en-dovascular intervention using a variety of techniques and devices, and this is now considered to be a better treat-ment option than surgery, when possible. A case report involving spontaneous retroperitoneal hemorrhage in a patient with myocardial infarction receiving intravenous heparin has been reported;5) however, the patient also received intravenous urokinase. We report a case of suc-cessful endovascular intervention using microcoils and gelform in a patient with spontaneous retroperitoneal hemorrhage after intravenous heparin treatment compli-cated by panhypopituitarism due to hypovolemic shock.

Case

A 57-year-old female with a history of hypertension

for 4 years presented to our hospital complaining of chest pain at rest. She did not have a history of bleeding and did not bruise easily. The physical examination on admission revealed the following vital signs: blood pres-sure, 110/70 mmHg; and heart rate, 70/min. Laboratory testing revealed a hemoglobin level of 10.6 g/dL; the platelet count, PT, and aPTT were all within normal li-mits. Coronary angiography revealed an 80% stenosis of the mid-left circumflex artery and a 50% stenosis of the proximal right coronary artery. We prescribed aspirin (200 mg) and clopidogrel (300 mg) as a loading dose and enoxaparin (6,000 IU subcutaneously twice a day). Two days later, we successfully deployed a paclitaxel-eluting stent (TaxusTM; Boston Scientific, Natick, MA, USA) at the left circumflex lesion using a right femoral approach, with unfractionated heparin (5,000 IU intra-venously) just before the percutaneous coronary inter-vention (PCI). Several hours later, the patient complain-ed of right lower quadrant abdominal pain and we co-uld see that the abdomen was distended with tenderness. The patient’s vital signs revealed that the blood pressure had decreased to 70/40 mmHg and the heart rate had increased to 100/min. The laboratory findings showed that the hemoglobin level had dropped to 7.3 g/dL. An abdominopelvic CT scan showed a huge retroperitoneal hemorrhage, including an active bleeding focus (Fig. 1). An urgent angiography was performed and revealed that the right access site was not remarkable, but two active bleeding foci, branches of the right lumbar artery and the right circumflex iliac artery, were visualized (Fig. 2). All bleeding foci, including collateral vessels, were suc-

Received: July 28, 2008

Revision Received: August 11, 2008 Accepted: September 5, 2008

Correspondence: Seung Jin Lee, MD, Division of Cardiology, Department of

Internal Medicine, Soonchunhyang University Cheonan Hospital, 23-20

Bongmyeong-dong, Cheonan 330-721, Korea

Tel: 82-41-570-3673, Fax: 82-41-574-5762

E-mail: drlsj@paran.com

Do-Hoei Kim, et al.·33

cessfully embolized using 7 microcoils (Fig. 3) and 5 pints of packed red blood cell were transfused. However, the blood pressure did not increase above 90/60 mmHg and the heart rate was 110/min. The repeat hemoglobin level was 8.1 g/dL. Fortunately, the patient’s urine out-put was preserved and the creatinine remained within normal limits. A follow-up CT scan 4 days after the em-bolization showed a larger hemorrhage with another bleeding focus (Fig. 4A) along with newly developed hydronephrosis in the right kidney due to ureteral com-pression (Fig. 4B). An additional angiography revealed multiple bleeding foci from branches of the right ova-rian artery (Fig. 5A). Due to the tortuosity of the right ovarian artery, we could not advance the guide wire, so we decided to use gelform and successfully occluded the right ovarian artery at the ostial portion (Fig. 5B). The patient complained of chest tightness with shortness of breath and a chest radiograph demonstrated a pleural effusion in both lungs. A chest CT scan and diagnostic thoracentesis showed a bilateral hemothorax (Fig. 6A), so we inserted a chest tube into the right pleural space. A total of 350 cc of blood was initially drained. After this intervention, the patient’s vital signs stabilized and

no bleeding foci were found in the follow-up abdomin-opelvic CT scan. Both hemothoraces also improved (Fig. 6B). Seven days later, we were able remove the chest tube. Before discharge, we confirmed that there was no evidence of platelet or coagulation disorders. Blood tests were as follows: fibrinogen, 350 mg/dL; protein C acti-vity, 94%; protein C antigen, 72.4%; protein S antigen, 40.6%; von Willebrand factor activity, 243%; factor as-say VIII, 112%; factor assay V, 96%; antinuclear anti-body, (-); anti-double stranded deoxyribonucleic acid (ds DNA) antibody, (-); and antiplatelet antibody (-). One month after the initial CT scan, a follow-up CT scan showed that the hemorrhage had increased slightly in size (Fig. 7A) and was more mature. Furthermore, the hydronephrosis had progressed with a deterioration in renal function (Fig. 7B). However, the 3 month follow-up CT scan revealed a much smaller volume of hemor-rhage (Fig. 8A) and an improvement in the hydroneph-rosis with a corresponding improvement in renal func-tion (Fig. 8B). The hemorrhage was almost completely resolved by the 7 month follow-up (Fig. 9) and there was no evidence of infection. One year later, the patient pre-sented to our hospital complaining of anorexia and loss of appetite. The patient’s serum sodium and potassium levels were 117 mEq/L and 3.3 mEq/L, respectively. The laboratory findings were consistent with panhy-popituitarism; specifically, endocrine profile was as fol-lows: prolactin, 3.7 ng/mL; growth hormone, 0.15 ng/ mL; luteinizing hormone (LH), 2.01 mIU/mL; follicle stimulating hormone (FSH), 7.24 mIU/mL; estradiol, 5.02 pg/mL; T3, 0.195 ng/mL; free T4, 0.179 ng/dL; thyroid stimulation hormone (TSH), 1.95 μIU/mL; cor-tosol at 8 am, 4.0 μg/dL; and adrenocorticotropin hor-mone (ACTH), 13.48 pg/mL. The sella MRI showed an empty sella (Fig. 10). The patient reco vered after once daily doses of oral prednisolone (5 mg) and levothyro-xine (0.05 mg).

Fig. 2. Initial finding of the angiography. The urgently performed angiography showed two active bleeding foci (arrows): branches of the right circumflex iliac artery (A) and right lumbar artery (B).

A B

Fig. 1. Initial abdominopevic CT scan. A huge hematoma in theretroperitoneal space was found, and there was contrast enhan-cement leakage inside of the mass (arrow) implying active ble-eding. CT: computed tomography.

34·Spontaneous Retroperitoneal Hemorrhage With Heparin

Fig. 3. Coil embolization. A total of 7 microcoils were used to eliminate all bleeding foci, including all collateral arteries. After coil em-bolization, a final angiography was performed and comfirmed that there was no contrast agent leakage.

A B

A B

Fig. 5. Another bleeding focus. A: the angiography revealed multiple bleeding foci (dark arrows) from branches of the right ovarian artery. B:after gelfoam injection, angiography showed a totally occluded right ovarian artery at the ostium portion (dotted arrow).

A B

Fig. 6. Both hemothoraces. A: a chest CT scan revealed bilateral hemothoraces, confirmed by diagnostic thoracentesis. A chest tube in-sertion was performed. B: 9 days later, a follow-up chest CT scan showed a much improved hemothorax. CT: computed tomography.

Fig. 4. Follow-up chest CT scan 4 days after the initial embolization. A: the size of the hematoma increased, but no suspicious bleeding sites were found. B: a hydronephrosis in the right kidney developed, probably caused by a right ureteral compression by the mass. CT:computed tomography.

A B

Do-Hoei Kim, et al.·35

Discussion

Access site bleeding is a common minor complication after PCI and it can usually be controlled easily with manual compression. However, retroperitoneal hemor-rhage from the puncture site can be lethal because it is difficult to stop bleeding by external compression. The worldwide incidence of retroperitoneal hemorrhage fol-lowing cardiac catheterization is 0.15%.6) The clinical presentation is varied and may be subtle, therefore early detection can be delayed. There are often no obvious stigmata of an underlying expanding hematoma or cuta-neous bruising. Retroperitoneal hemorrhage should be suspected in patients with significant flank pain, groin

pain, or relative hypotension and tachycardia that tran-siently improve with fluid resuscitation, and a fall in hemoglobin level following a procedure. Most hemody-namically stable patients can be managed with fluid re-suscitation, correction of coagulopathy, and blood trans-fusion. But if the patient is unstable, conservative man-

A B

Fig. 7. One month after the initial chest CT scan. A: the size of the hematoma slightly increased without any bleeding points and the wall of the hematoma was more densely enhanced, indicating that the hematoma had matured. B: the hydroneprosis of the right kidney becamemore aggravated. CT: computed tomography.

A B

Fig. 8. Three month follow-up chest CT scan. A: the size of the hematoma was decreased. B: the hydronephrosis of the right kidney hadnearly resolved. CT: computed tomography.

Fig. 9. Seven month follow-up chest CT scan. The hematoma wasnearly resolved. CT: computed tomography.

Fig. 10. The sella MRI. A T1-weighted image revealed that the pi-tuitary gland was thin and flat and the sella was empty (arrow).MRI: magnetic resonance imaging.

36·Spontaneous Retroperitoneal Hemorrhage With Heparin

agement should be avoided.7) In the past, open surgical repair has been considered the only option in uncon-trollable hemodynamic collapse,8) but removal of the he-matoma may increase rebleeding by releasing the tam-ponade effect.9) Recently, many reports have discussed successful endovascular interventions using microcoils, gelform, or stent grafts. Microcoils are probably the most widely used and safest form of treatment, but it should be remembered that proximal coiling of the bleeding artery may not be sufficient when there is a rich net-work of collateral arteries and new arteries may develop after embolization of the bleeding artery.10) It is impor-tant to place coils both proximally and distally to the bleeding site to prevent rebleeding. In the patient pres-ented herein, we used seven microcoils in the bleeding site, including all collaterals.

Retroperitoneal hemorrhage can occur spontaneously in the absence of an obvious underlying pathology or trauma. It is most commonly seen in patients who have been anticoagulated, have bleeding abnormalities, or are receiving hemodialysis. Several sporadic cases have been reported, but the exact pathologic mechanism is not yet known. One possible hypothesis is diffuse occult vascu-lopathy and arteriosclerosis of the small vessels in the retroperitoneum.11) A second suggested hypothesis is hep-arin-induced immune micriangiopathy.12) It can be as-sumed that our patient was initially exposed to unfrac-tionated heparin by heparin mixed saline during the diagnostic coronary angiography, in which case antibo-dies to heparin were produced, so that when the patient was exposed to heparin again during PCI, there was an antigen-antibody reaction, resulting in microvascular damage. The final possible hypothesis is that an unrecog-nized minor trauma in the microcirculation during phy-sical activity and vomiting or coughing may have induced sustained bleeding in the absence of clotting factors.13)

An abdominal compartment syndrome is a rare com-plication that acutely increases intra-abdominal pressure by massive retroperitoneal hemorrhage, which can cause a decrease in cardiac output, anuria, or worsening of re-nal failure, intestinal ischemia, and respiratory failure.14) In our case, hydronephrosis and acute renal failure de-veloped due to an extrinsic compression of the retroperi-toneal hemorrhage. However, renal function gradually improved as the size of the hematoma decreased. There-fore, if the acute bleeding is controlled and vital signs are stabilized, we think it is a wise choice to wait and monitor the patient’s condition despite the enormous size of the hematoma, as in the case presented. In our

patient, panhypopituitarism developed one year after the initial bleeding, which is the first report of empty sella caused by acute spontaneous retroperitoneal bleeding.

In conclusion, although our case featured an extre-mely large retroperitoneal hemorrhage, we successfully stopped the acute bleeding through endovascular inter-vention and eventually the mass spontaneously resolved without surgical intervention.

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