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Peliosis of the Spleen: Possible Association with Chronic Renal Failure and Erythropoietin Therapy

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Peliosis of the Spleen: Possible Association with Chronic Renal Failure and Erythropoietin Therapy Peliosis of the spleen 493 Postgrad Med J: first published as 10.1136/pgmj.71.838.493 on 1 August 1995. Downloaded from Peliosis of the spleen: possible association with chronic renal failure and erythropoietin therapy KY Lam, ACL Chan, TM Chan Summary of gastrointestinal blood loss. The levels of Splenic peliosis was identified at necropsy iron, ferritin, and aluminium were within nor- in a 62-year-old woman receiving con- mal limits, while that of parathyroid hormone tinuous ambulatory peritoneal dialysis was elevated to 298 pg/ml (normal range for end-stage renal failure, and ery- 10-50). She was started on erythropoietin thropoietin therapy for uraemia and therapy (2000 units subcutaneous injection anaemia. The immediate cause of death twice weekly) in July 1993. In August 1993, she was arrhythmia related to ischaemic was admitted because of abdominal intramus- heart disease, following an episode of cular haematoma after minor trauma. Com- intramuscular haematoma (secondary to plete blood picture showed a haemoglobin level platelet dysfunction). The unusual assoc- of 5.4 g/dl, white cell count of 8.7 x 1O9/1 and iation between peliosis and renal failure, platelet count of 1 15 x 109/1. Prothrombin time and possibly erythropoietin therapy, is and activated partial thromboplastin time were discussed. within the normal range. She had not received any medication that could predispose to Keywords: peliosis, spleen, erythropoietin bleeding diathesis. She was transfused and her haemodynamic condition was stable. While being investigated for her intramuscular Peliosis is a rare lesion characterised by wide- haemorrhage, she developed arrhythmia with spread blood-filled cystic spaces, which is most sudden cardiac arrest shortly after admission. often detected in the liver and the spleen,'23 but also occasionally detected in other organs Post mortem findings including lung, lymph node, bone marrow, Autopsy examination showed a 20-cm intra- kidney, adrenal gland, gastrointestinal tract muscular haematoma within the anterior and parathyroid gland. 2,8,16,18,24,25 The condition abdominal wall. Both kidneys were small and is often clinically silent, but can predispose to nodular hyperplasia of the parathyroid glands rupture of the liver or spleen.7"0"2'20'22'23 was present. There was concentric left ven- http://pmj.bmj.com/ We report an incidental finding of peliosis of tricular hypertrophy and coronary athero- spleen in a patient with end-stage renal failure sclerosis with 75%O obstruction of the right and receiving erythropoietin therapy for coronary artery. There was no evidence of uraemia and anaemia. The possible association myocardial infarction. The cause of death was between peliosis and erythropoietin therapy is arrhythmia, probably related to ischaemic discussed. heart disease following an episode of intramus- cular haematoma. The bleeding tendency was on September 25, 2021 by guest. Protected copyright. Case report probably due to mild thrombocytopenia and platelet dysfunction related to chronic renal A 62-year-old Chinese woman presented in failure. June 1990 with end-stage renal failure and The spleen was slightly enlarged and bilateral small kidneys. Except for a three-year weighed 260 g. Numerous blood-filled cyst- history of gouty arthritis, her past health was like spaces were scattered throughout the cut unremarkable. At presentation, she had nor- surface. These cysts measured from 1 to 5 mm mochromic normocytic anaemia with a haemo- in diameter. They were round, oval or some- globin level of 6.3 g/dl, white cell count of what irregular and were filled with partially or 6.2 x 109/l, and platelet count of 195 x 109/l. totally clotted blood (figure). Microscopic She was started on continuous ambulatory examination showed that these cystic spaces peritoneal dialysis, with serum urea and lacked endothelial lining. This was confirmed Queen Mary Hospital, creatinine levels Hong Kong, maintained at around 25 by negative immunostaining of the 'cyst wall' Department of mmol/l (70.0 mg/dl) and 1100 ltmol/l (12.4 mg/ for endothelial markers, factor VIII related Pathology, dl), respectively. She remained uneventful for antigen (at a dilution of 1:300 from Dakopatts, KY Lam two years, with stable serum biochemistry and Glostrup, Denmark) and CD31 (at a dilution of ACL Chan haemoglobin levels, not requiring any blood 1:20 from Dako, Kyoto, Japan). There was no Department of transfusion. Thereafter, she developed pro- evidence of Medicine necrosis or significant TM Chan gressive anaemia, necessitating blood trans- haemosiderin deposition. The features were fusion with increasing frequency up to one unit consistent with peliosis ofthe spleen. The other per month. Investigations including gastro- organs including the liver showed no evidence Accepted 24 February 1995 scopy and sigmoidoscopy showed no evidence of peliosis. 494 Lam, Chan, Chan Features/causes of peliosis Features: Postgrad Med J: first published as 10.1136/pgmj.71.838.493 on 1 August 1995. Downloaded from * an uncommon lesion * multiple blood-filled cyst-like spaces * in liver, spleen, lung, lymph node, bone marrow, kidney, adrenal glands, gastrointestinal tract and parathyroid gland Proposed causes: * idiopathic * admission of steroids * neoplastic diseases * tuberculosis * AIDS Figure Cut surface of the spleen with numerous * diabetes mellitus blood-filled cystic spaces measuring up to 5 mm in * chronic renal failure diameter (white arrows) * Thorotrast * immunosuppressive therapy Box 1 Discussion Splenic peliosis refers to the macroscopic and liver, cervix and colon, lymphoma, chronic microscopic findings of multiple blood-filled myelomonocytic leukaemia and paragang- cystic spaces in the spleen usually without an lioma),49"11-13,21 tuberculosis,l acquired im- endothelial lining. The entity must be differ- munodeficiency syndrome,'5 diabetes mel- entiated from congestion, vascular tumours litus,1 chronic renal failure,4 Thorotrast,'4 and and tumour-like vascular lesions of the spleen immunosuppressive therapy.7 Nevertheless, including haemangioma, angiosarcoma, Kap- the aetiology is still unknown. In the present osi's sarcoma and bacillary angiomatosis. Con- case, the patient had chronic renal failure, but it gestion ofthe spleen is diffuse rather than focal, has been suggested that chronic renal failure and the cordal-sinusoidal relationship remains per se was probably not responsible for peliosis normal. Cavernous haemangioma is the most as it is commonly found after renal transplanta- common benign vascular tumour of the spleen, tion.27 Thus, the possible role oferythropoietin but the vascular spaces are lined by endothelial should also be considered in this case. cells and are separated by bands of connective Erythropoietin is a circulating glycoprotein tissue. Angiosarcoma is characterised by anas- composed of 165 amino acids and four oligosac- tomosing vascular channels lined by atypical charide chains.28 The gene for the hormone has endothelial cells with frequent tufting. been cloned and recombinant erythropoietin Kaposi's sarcoma has the characteristic inter- has become an accepted therapy for the treat- lacing pattern of spindle cells with cleft-like ment of anaemia of chronic renal failure with spaces and hyaline bodies. Bacillary well-documented beneficial effects.29 However, http://pmj.bmj.com/ angiomatosis in immunocompromised patients there are several possible adverse effects in is caused by the bacteria Rochalimaea hen- using erythropoietin therapy. These include an selae.26 It is distinguished from peliosis by the increase in blood pressure, generalised sei- presence of neutrophils and bacterial clumps, zures, and possible increased clotting of vas- the latter can be demonstrated by Warthin cular access. It has never been associated with Starry stain. peliosis. However, the effect of erythropoietin Forty cases of splenic peliosis have been on the cardiovascular system might lead to on September 25, 2021 by guest. Protected copyright. documented in the English literature (table peliosis. Also, erythropoietin secretion is in- 1).i-23 The condition has a wide age distribu- creased by androgens and the latter are strongly tion (ranging from 14 to 82) with a mean of 53 associated with peliosis. Thus, erythropoietin and a male to female ratio of 1.7:1. Although may be the mediator responsible for causing most of the cases were found incidentally, peliosis in this group ofpatients. The proposed splenic peliosis was not just a pathological theories for the pathogenesis ofpeliosis include curiosity because splenic rupture was seen in congenital malformations, vascular varico- 20% (eight cases). Many cases were associated sities, ruptured vessels, primary necrosis fol- with hepatic peliosis, but 23 cases of isolated splenic peliosis were identified. However, many of these cases of isolated splenic peliosis Properties of erythropoietin were from surgical specimens and the status of the liver might not be properly assessed. Thus, * a circulating glycoprotein it was difficult to prove that the liver was not * composed of 165 amino acids and four involved except in reports documenting aut- oligosaccharide chains opsy findings and these included a total of 11 * secreted by the kidney * used for treatment of anaemia associated with cases,13'15'16 including the present case. chronic renal failure Splenic peliosis has been mostly associated * adverse effects include increased blood with the administration of steroids, especially pressure, generalised seizures,
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