This case report describes an unusual case of spontaneous iliac intramuscular hematoma after a heart transplant secondary to valvular cardio­myopathy. A spontaneous retroperitoneal hematoma occurring after a heart transplant is rare, but can occur when the patient is on immunosuppressive medications. This could be secondary to the medications’ ability to suppress the immune system and cause antiplatelet therapylike effects. Spontaneous retroperitoneal hematomas can cause significant patient morbidity, sometimes even causing immobility; however, a high index of suspicion with early recognition and immediate intervention can reduce the sequelae. Although the occurrence of a spontaneous retroperitoneal hematoma after a heart transplant is rare; there should be a high index of suspicion in cases where the patient is elderly, has experienced rejection episodes, and is taking immunosuppressants, or in cases where the patient is additionally on anticoagulation and antiplatelet therapy.

Key words : Heart transplantation, Spontaneous bleed

Introduction

Heart transplants are used to treat end-stage cardiomyopathy and coronary artery disease.¹ However, there is a myriad of debilitating com­plications that can occur after a heart transplant that have a negative effect on the recipient’s quality of life. These complications include infection, rejection, accelerated graft atherosclerosis, malignancy, mediastinitis, bleeding complications, as well as aortic dissection, and thromboembolism.¹ Unnoted in literature, a potential rare complication is a spontaneous retroperitoneal hematoma (SRH). Spontaneous retroperitoneal hematomas are potentially lethal, uncommon diagnosis that often have nonspecific presentations, which can lead to misdiagnosis and delayed treatment. Spontaneous retroperitoneal hematomas are defined as bleeding into the retroperitoneal space without associated trauma or iatrogenic manipulation. Because there is the potential of fatality associated with SRHs, they require immediate recognition and intervention.² Thus, it is important to have a high index of suspicion to reduce the sequela associated with an SRH. Here, we present an unusual case of spontaneous iliac intramuscular hematoma after a heart transplant.

Case Report

A 68-year-old woman received a heart transplant secondary to valvular cardiomyopathy. She had undergone a mitral valve repair 25 years earlier for a myxomatous mitral valve. Despite undergoing induction with antithymoglobulin for a positive
B-cell crossmatch, she developed an acute cellular and humoral rejection 2 weeks after undergoing a heart transplant that required readmission and rejection treatment including intravenous immuno­globulin and plasmapheresis. She developed pancytopenia from her immunosuppression regimen and 4 weeks after the heart transplant, she developed acute right hip and thigh pain. On physical examination, she was noted to have decreased neuromuscular function of her right lower extremity. A computed tomography scan of her abdomen and pelvis revealed a 4.2 × 8.4 cm intramuscular collection of high density in her right iliac compatible with an intramuscular hematoma (Figure 1). She underwent an additional 30-day inpatient stay at a physical rehabilitation center and was discharged home with the use of a cane for ambulation.

Discussion

Spontaneous retroperitoneal hematomas have been reported in the literature after abdominal organ transplant but not after a heart transplant. However, the most frequent causes of SRHs are renal tumors, vascular disease, and anticoagulant treatments.³ Of interest to this case report is the association with anticoagulation therapy. According to Simsek and associates, anticoagulation and antiplatelet therapy can cause SRH in elderly patients taking multidrug medications.³ Although heart transplant patients may not be on anticoagulation and antiplatelet therapy specifically, the immunosuppressants the patients are medicated with can cause similar effects as the anticoagulation and antiplatelet therapy; thus, making SRH a possible complication for certain patients.

In general, immunosuppressants are associated with increased susceptibility to infection and risk of malignancy, with specific agents having consequences of hypertension, bone marrow suppression, leukopenia, and rarely thrombocytopenia.⁴ Because of their ability to suppress the immune system and cause antiplatelet therapy effects, SRHs can occur as a complication of immunosuppressant use as seen in this case presentation. This patient experienced acute cellular and humoral rejection, which then required the use of more immunosuppressants, whose negative effects may be seen by the pancytopenia that developed in the patient. As pancytopenia results in some of the same effects of antiplatelet therapy, with a reduction in platelet count, it is likely that the SRH that occurred was because of the immuno­suppressants that originally caused the pancytopenia.

Knisely and associates indicate 2 cases which after a difficult transplant died because of retroperitoneal hemorrhage after heparin treatment for pulmonary embolism, a complication of their initial heart transplant.¹ While this supports the association of SRHs with anticoagulation therapy, these patients could have been at an increased risk for SRHs because of the compounding effects of the immuno­suppressants they were on causing even greater antiplatelet and anticoagulant therapy effects. Therefore, SRHs should have been suspected. As SRHs are a rare but serious complication, it is essential to have a high index of suspicion, so that the SRHs may be immediately recognized in cases where they are possible.

Spontaneous retroperitoneal hematomas can lead to a debilitating postoperative course and neurologic sequelae if not caught early and treated aggressively. Although the presentation of SRH is nonspecific, the most common symptoms are abdominal, leg, hip, and back pain. Spontaneous retroperitoneal hematomas can be then subsequently diagnosed using computed tomography or magnetic resonance imaging scanning.² In this case report, the patient presented with both leg and hip pain and then had a positive CT scan (Figure 1), which confirmed the SRH diagnosis. Spontaneous retroperitoneal hematomas occurring after a heart transplant are rare, but can occur in certain cases where immuno­suppressants are used. There should be a high index of suspicion for SRH in cases where the patient is elderly, has experienced rejection episodes, and is taking immunosuppressants, or in cases where the patient is additionally on anticoagulation and antiplatelet therapy. As SRH can cause significant patient morbidity and immobility, it is crucial to detect these early and begin aggressive intervention.

References:

1. Knisely BL, Mastey LA, Collins J, Kuhlman JE. Imaging of cardiac transplantation complications. Radiographics. 1999;19(2):321-339; discussion 340-341.
   CrossRef - PubMed
2. Sunga KL, Bellolio MF, Gilmore RM, Cabrera D. Spontaneous retroperitoneal hematoma: etiology, characteristics, management, and outcome. J Emerg Med. 2012;43(2):e157-e161.
   CrossRef - PubMed
3. Simsek A, Ozgor F, Yuksel B, et al. Spontaneous retroperitoneal hematoma associated with anticoagulation therapy and antiplatet therapy: two centers experiences. Arch Ital Urol Androl. 2014;86(4):266-269.
   CrossRef - PubMed
4. Min DI, Monaco AP. Complications associated with immunosuppressive therapy and their management. Pharmacotherapy. 1991;11(5):119S-125S.
   PubMed