Case Report

Spontaneous splenic rupture caused by splenic peliosis of a hemodialysis patient with chronic renal failure receiving erythropoietin

Abstract

Peliosis is a rare lesion characterized by widespread blood- filled cystic spaces, most often detected in the liver and the spleen. Spontaneous splenic rupture caused by splenic peliosis is often fatal. We report the case of a patient who underwent hemodialysis and received erythropoietin. The patient was admitted because of sudden onset of abdominal pain in the left side, and emergency laparotomy was performed. A ruptured enlarged spleen was found with a hematoma at the surface with 1600 mL of nonclotted blood in the peritoneal cavity. A splenectomy revealed an enlarged spleen weighing 500 g. Eleven days after admission, the patient recovered and was discharged from the hospital. This case report demonstrates that spontaneous splenic peliosis rupture needs to be considered in the differential diagnosis of chronic renal failure patients with abdominal pain in the left side who have been undergoing hemodialysis and receiving erythropoietin.

Spontaneous splenic rupture is rare, and some cases receiving anticoagulant or thrombolytic therapy have been reported [1]. Lund et al reported a case of atraumatic rupture of the spleen in a uremic, hemodialyzed patient [2]. The diagnosis of spontaneous splenic rupture is very difficult to make in a hemodialyzed patient. This case should remind physicians that spontaneous splenic rupture caused by splenic peliosis needs to be considered in the differential diagnosis of chronic renal failure patients with abdominal pain in the left side who have been undergoing hemodialysis and receiving erythropoietin. The possibility of spontaneous splenic rupture needs to be borne in mind because, in most cases, early diagnosis and treatment can avoid morbidity and Loss of life [3].

A 49-year-old man was admitted because of sudden onset of abdominal pain. The patient had a 14-year history of chronic hemodialysis and erythropoietin therapy for terminal uremia caused by chronic glomerulonephritis and anemia. Erythro- poietin was given intravenously 2 times weekly (total 6000 IU/ wk). There was no history of trauma or androgen therapy. Three days before admission, an episode of sudden severe abdominal pain in the left side occurred. There were no other associated symptoms. On initial examination at the hospital, upper abdominal pain in the left side with peritoneal signs was

observed. The abdomen was rigid and tender with no bowel sounds. On physical examination, his body temperature was 98.968F, blood pressure was 82/56 mm Hg, and pulse rate was 98/min. Hemoglobin and hematocrit were 5.6 g/dL and 0.181, respectively. An abdominal computerized tomographic scan revealed splenomegaly with splenic hematoma and fluid in the peritoneal cavity (Fig. 1). Prothrombin time and platelet count were 94% and 72000/mm³, respectively. Emergency laparot- omy was performed. A ruptured enlarged spleen was found with a hematoma at the surface with 1600 mL of nonclotted blood in the peritoneal cavity. There was no evidence of neoplasia or hematologic disorder. The liver was normal in appearance; but because a liver biopsy was not performed, the possibility of coexisting hepatic peliosis could not excluded. A splenectomy revealed an enlarged spleen weighing 500 g. Cross section showed that dark red blood-filled cavities had formed in the whole splenic parenchyma (Fig. 2A). Histopath- ologic examination of the spleen revealed numerous blood- filled cystic spaces, with focal dilation of sinusoids, scattered diffusely (Fig. 2B). There was no evidence of necrosis or significant hemosiderin deposition. The features were consis- tent with peliosis of the spleen. Eleven days after admission, the patient was discharged to continue hemodialysis.

Peliosis is a rare lesion characterized by widespread blood- filled cystic spaces, most often detected in the liver and the spleen but also occasionally found in other organs. Splenic peliosis is most commonly associated with steroid adminis-

Fig. 1 An abdominal computerized tomographic scan revealed splenomegaly with splenic hematoma and fluid in the peritoneal cavity (coronal image).

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109.e2 Case Report

Fig. 2 A, Resected specimen. Many round small blood-filled cavities are present on a cross section of the whole parenchyma. In the upper pole, there are large thrombosed cavities and slightly smaller blood-filled cavities 1 to 10 mm in diameter, with irregular margins close to the large cavities. B, histopathologic examination of the spleen revealed numerous blood-filled cystic spaces, with focal dilation of sinusoids, scattered diffusely (hematoxylin-eosin stain; original magnification, x40).

tration, especially anabolic steroids [4]. It has also been reported in association with neoplasms, tuberculosis, Acquired immunodeficiency syndrome, diabetes mellitus, chronic renal failure, Thorotrast, and immunosuppressive therapy [4]. Previous reports note inflammation, sinusoidal reticulin fiber destruction, and local deposits of immunoglobulin G and activated C3 in the surroundings of the lesions [5-7]. Nevertheless, the precise mechanism of its origin is still unknown. In our case, the patient had chronic renal failure while receiving erythropoietin; and a possible association between splenic peliosis with renal failure and erythropoietin therapy was suggested. Although recombinant erythropoietin is now an accepted therapy for treating anemia in chronic renal failure, adverse effects of erythropoietin include increased blood pressure, Generalized seizures, increased clotting of vascular access, and pure red cell aplasia [8]. The effect of erythropoietin on the cardiovascular system can lead to peliosis. Erythropoietin secretion is increased by androgens, and the latter are strongly associated with peliosis. Thus, erythropoietin may be the mediator responsible for causing peliosis. The effects of erythropoietin on the microvasculature of the spleen are unknown. In our case, there was endothelial lining noted in the blood cysts and there was no necrosis in the parenchyma. Thus, it is unclear whether erythropoietin acted through these mechanisms.

Although peliosis is often symptomless, fatal intraperito- neal hemorrhage may occur because of rupture. Slater et al reported a case of spontaneous splenic rupture in a hemodialysis patient with renal failure being treated with androgens [9]. Spontaneous splenic rupture needs to be considered in the differential diagnosis of chronic renal

failure patients with abdominal pain in the left side who have been undergoing hemodialysis and receiving erythropoietin.

Shigeru Hakoda MD, PhD Hiroshi Shinya MD, PhD Shun-ichiro Kiuchi MD, PhD

Department of Emergency Medicine, Kitano Hospital The Tazuke Kofukai Medical Research Institute

Osaka, Japan E-mail address: shakodajp@yahoo.co.jp

doi:10.1016/j.ajem.2007.03.025

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