Recurrence of Severe Hemoperitoneum in a Patient on Peritoneal Dialysis

Perit Dial Int 29(5): 583-585
2009
© 2009 International Society for Peritoneal Dialysis

Recurrence of Severe Hemoperitoneum in a Patient on Peritoneal Dialysis

R. Ikee^1,*, J. Branch², K. Honda¹, K. Ishioka¹, M. Oka¹, K. Maesato¹, H. Moriya¹, S. Hidaka¹, T. Ohtake¹ and S. Kobayashi¹

Department of Nephrology and Kidney & Dialysis Center¹ Department of General Internal Medicine² Shonan Kamakura General Hospital Kanagawa, Japan
^* e-mail: ryota.ikee@tokushukai.jp
Editor:
A peritoneal dialysis (PD) catheter is a window connecting the inside of the abdominal cavity to the outside. The diagnosis of hemoperitoneum can therefore be easily made by visualizing bloody peritoneal effluent. The most common cause of bloody peritoneal effluent is related to the menstrual cycle, resulting in minor bleeding with pink-tinged dialysate (1). Severe hemoperitoneum requiring blood transfusion or surgical intervention is rare. We report a rare case of recurrent severe hemoperitoneum in a PD patient.
A 38-year-old man with end-stage renal failure of unknown origin had been treated with continuous ambulatory PD (2 L 1.5% dextrose-containing solution, 4 exchanges/day) since July 2004. In March 2005, he presented acutely with abdominal pain and bloody peritoneal effluent. On admission, vital signs were blood pressure 138/60 mmHg, heart rate 84 beats/minute, and body temperature 36.3°C. Palpebral conjunctiva was anemic. Cardiac and pulmonary examinations were un-remarkable. The abdomen was distended, with tenderness around the umbilicus but no muscular defense or rebound tenderness. Complete blood cell count showed hemotocrit 19.6% (28.9% on the day before admission), white blood cell count 15.1 x 10⁹/L, and platelet count 144 x 10⁹/L. The results of coagulation tests were normal. Abdominal radiography showed no abnormal findings and the PD catheter tip was located in the rectovesical pouch. However, contrast computed tomography (CT) showed fluid retention in the abdominal cavity and a mass with heterogeneous density adjacent to the stomach, indicating hemoperitoneum and hematoma. Extravasation of radiographic contrast medium was not observed.
A peritoneotomy was performed and a large amount of bloody fluid and hematoma in the omental bursa were noted. Two aneurysms (measuring 2 cm in diameter) in the right gastroepiploic artery (GEA) were found. The aneurysms were resected along with a partial omentectomy. However, hemorrhagic lesions were not evident in a formalin-fixed specimen, which might be attributable to the dissolution of hemorrhagic vessels in the course of formalin fixation. Microscopic evaluation of other vessels did not show vasculitis or atherosclerosis. Magnetic resonance angiography (MRA) of the cranial vessels and selective abdominal angiography utilizing three-dimensional CT did not reveal any other aneurysms. Two weeks after the surgery, the patient was discharged from our department. During the next month, the PD catheter was removed because of refractory peritonitis; three-times per week hemodialysis was commenced.
In September 2007, the patient presented acutely with hypotension (systolic blood pressure 70 mmHg) and mild pain in the left hypochondrium during a hemodialysis session. Complete blood cell count showed hematocrit 25.3% (32.8% 9 days before). Abdominal contrast CT revealed an aneurysm and fluid retention in the abdominal cavity (Figure 1). A peritoneotomy was performed again and an aneurysm measuring 2 cm in diameter in the left GEA was resected. Pathological evaluation of the aneurysm showed that smooth muscular cell layer of the media had thinned and disappeared and adventitial fibroplasia was seen. Inflammatory cell infiltration and atherosclerosis were not present. These pathological findings were compatible with fibromuscular dysplasia and the medial changes were characteristic of segmental mediolytic arteriopathy (SMA). Repeat cranial MRA and selective abdominal angiography utilizing three-dimensional CT did not reveal other aneurysms. Twelve days after the surgery, the patient was discharged from our department and continues out-patient hemodialysis.

View larger version (7K):[in this window] [in a new window] Figure 1 — Abdominal contrast computed tomography in the second presentation showed an aneurysm (arrowhead) and hemoperitoneum.

Generally, hemoperitoneum is a benign complication of PD. Tse et al. followed 549 patients for more than 10 years and reported 116 episodes of hemoperitoneum in 46 patients (2). During follow-up, ultrafiltration failure was not induced by hemoperitoneum and the causes of death in 4 patients were unrelated to hemoperitoneum. Greenberg et al. followed 424 patients for 11 years and reported 30 episodes of hemoperitoneum in 26 patients (1). In 24 of the 30 cases, hemoperitoneum was due to benign pathogenesis, such as retrograde menstruation. In the remaining 6 cases, significant bleeding occurred and the causes included rupture of an ovarian cyst, post-splenectomy, and encapsulating peritoneal sclerosis. Rupture of aneurysms of splenic and omental arteries have also induced significant bleeding (3,4).
In our patient, recurrent rupture of GEA aneurysms led to hemoperitoneum and 1 episode of hemorrhagic shock. Unfortunately, in the first episode of hemoperitoneum, pathologic evaluation of hemorrhagic lesions could not be performed because the hemorrhagic vessels dissolved in the course of formalin fixation. Two years after the first episode of hemoperitoneum, severe hemoperitoneum recurred. The second episode was due to rupture of a left GEA aneurysm, which had not been found in abdominal contrast CT in the first episode. In the second episode, pathologic findings of the left GEA aneurysm were compatible with fibromuscular dysplasia. Fibromuscular dysplasia is a non-atherosclerotic and noninflammatory arterial disease commonly affecting the renal or carotid arteries but rarely affecting the visceral arteries (5). Based on the medial changes of the aneurysm, we made a diagnosis of SMA.
Segmental mediolytic arteriopathy was first defined as segmental mediolytic arteritis by Slavin and Gonzalez—Vitale in 1976 (6). Nowadays, however, it is clear that inflammatory responses are not generally recognized in this disease, and segmental mediolytic arteriopathy, or segmental arterial mediolysis, is used as a better term. Some authorities proposed that SMA is a precursor or a variant of fibromuscular dysplasia (7). SMA is initiated by lytic change of the outer media, which can expand to involve the mid and inner media (7). Transmural mediolysis results in arterial wall gap frequently complicated by dissecting hematomas and aneurysms. SMA commonly affects visceral small and medium-sized arteries and rarely affects cerebral, coronary, or pulmonary arteries (8). Takagi et al. reviewed the SMA literature and showed that 9 of 19 patients had died from rupture of aneurysms due to SMA (8). It is noteworthy that multiple aneurysms are often found and repeated bleeding has been reported (9). Although SMA is considered a rare disease, case reports will increase with recognition of SMA as a distinct disease. In fact, case reports of SMA have been increasing in Japan (10).
Hemoperitoneum is a benign complication in most cases involving PD. However, SMA should be recognized as a recurrent and fatal cause of hemoperitoneum.
DISCLOSURE
No financial conflict exists.
REFERENCES

1. Greenberg A, Bernardini J, Piraino BM, Johnston JR, Perlmutter JA. Hemoperitoneum complicating chronic peritoneal dialysis: single-center experience and literature review. Am J Kidney Dis1992; 19:252 -6.[Medline]
2. Tse KC, Yip PS, Lam MF, Li FK, Choy BY, Chan TM, et al. Recurrent hemoperitoneum complicating continuous ambulatory peritoneal dialysis. Perit Dial Int 2002;22 : 488-91.[Abstract/Free Full Text]
3. Macia M, Pulido-Duque JM, Hortal L, Vega N, Garcia-Medina J. Ortiz E, et al. Percutaneous embolization of splenic artery pseudoaneurysm as a treatment of hemoperitoneum in a CAPD patient. Perit Dial Int 1993; 13:157 -9.[Free Full Text]
4. Park DJ, Oh KH, Kim SJ, Joo KW, Han JS, Kim S, et al. True aneurysm rupture of omental artery leading to hemoperitoneum and shock in a CAPD patient. Nephrol Dial Transplant2005; 20:2292 .[Free Full Text]
5. Yamaguchi R, Yamaguchi A, Isogai M, Hori A, Kin Y. Fibromuscular dysplasia of the visceral arteries. Am J Gastroenterol1996; 91:1635 -8.[Medline]
6. Slavin RE, Gonzalez-Vitale JC. Segmental mediolytic arteritis. A clinical pathologic study. Lab Invest1976; 35:23 -9.[Medline]
7. Slavin RE, Saeki K, Bhagavan B, Maas AE. Segmental arterial mediolysis: a precursor to fibromuscular dysplasia? Mod Pathol 1995; 8:287 -94.[Medline]
8. Takagi C, Ashizawa N, Eishi K, Ashizawa K, Hayashi T, Tanaka K, et al. Segmental mediolytic arteriopathy involving celiac to splenic and left renal arteries. Intern Med 2003;42 : 818-23.[Medline]
9. Rosenfelder NA, Taylor-Robinson SD, Jackson JE, Stamp GW. Segmental mediolytic arteriopathy in a patient with intraperitoneal bleeding. Eur J Gastroenterol Hepatol 2006;18 : 295-7.[Medline]
10. Inada K, Maeda M, Ikeda T. Segmental arterial mediolysis: unrecognized cases culled from cases of ruptured aneurysms of abdominal visceral arteries reported in the Japanese literature. Pathol Res Pract 2007; 203:771 -8.[Medline]