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Pelvic splenosis mimicking endometriosis, causing low abdominal mass and pain. Peer Zitzer, Moti Pansky, Ron Maymon1,. Rami Langer, Ian Bukovsky and Avi ...
Human Reproduction vol.13 no.6 pp.1683–1685, 1998

CASE REPORT

Pelvic splenosis mimicking endometriosis, causing low abdominal mass and pain

Peer Zitzer, Moti Pansky, Ron Maymon1, Rami Langer, Ian Bukovsky and Avi Golan Department of Obstetrics and Gynaecology, Assaf Harofeh Medical Centre (affiliated with Sackler Faculty of Medicine, Tel Aviv University), Zerifin 70300, Tel Aviv, Israel 1To

whom correspondence should be addressed

Splenosis is the heterotopic autotransplantation of splenic tissue that usually follows traumatic splenectomy. The clinical significance of these splenic implants and the need for surgical removal is debatable. A case of a 35 year old woman, with post-traumatic splenectomy presenting with low abdominal pain and pelvic mass is reported. Laparoscopic removal of the pelvic splenic implants dispelled all complaints. Key words: abdominal pain/endometriosis/laparoscopy/pelvic splenosis/pelvic tumour

Introduction Splenosis is the heterotopic autotransplantation of splenic tissue that usually follows traumatic rupture of the spleen. The description of heterotopic splenic nodules in the human peritoneal cavity dates from the beginning of this century (Faltin, 1911), and the terminology was first used by Buchbinder and Lipkopf in 1939. The clinical significance of this condition and the need for surgical treatment is debatable. We describe a case in which a woman presented with low herehere abdominal pain and pelvic masses that were eventually diagnosed as splenosis. Case report A 35 year old Caucasian female (gravid 4, parous 3) was admitted to the gynaecological department complaining of chronic pelvic pain, dysmenorrhoea, and deep dyspareunia. She had a past history of splenectomy due to traumatic rupture during a car accident. Displaying similar symptoms 6 years ago, the patient underwent a laparoscopy elsewhere, was diagnosed as having moderate endometriosis, and was unsuccessfully medically treated with Danazol for 6 months. At the present admission, results of routine physical and laboratory tests were unremarkable. On pelvic examination, the uterus was found to be retroverted, normal in size, and tender. The left ovary was enlarged to 436 cm and also tender. The right ovary and cul-de-sac were normal. On transvaginal © European Society for Human Reproduction and Embryology

ultrasound scanning, a 334 cm solid mass attached to the left ovary was demonstrated. The other pelvic structures appeared normal. Doppler flow study of this tumour showed a resistance index (RI) of 0.9 and the serum CA 125 value was 4 IU/ml. Further exploration of the tender pelvic mass was required, because an exact diagnosis and final treatment could not be determined. Before the operation, the patient was advised about the various surgical options and it was agreed that if the diagnosis was not definitive during surgery or if there was the slightest doubt, then salpingo–oophorectomy would be performed. This concurs with recommendations for indications and procedures for laparoscopy (Chapron et al., 1996). Thus on laparoscopy, a normal size uterus with two small bluish– red masses of 1 cm in diameter with soft consistency were observed on its fundus (Figure 1). On the left ovary, a similarly coloured 334 cm mass was found (Figure 2). The cul-de-sac was crowded with seven of the same coloured masses ranging in diameter from 0.5 to 2 cm (Figure 2). Two additional masses were noticed on the serosal surface of the rectosigmoid. The rest of the abdominal cavity was normal, with no pelvic adhesions nor any other evidence of either endometriosis or ascites. Since the exact nature of the macroscopic lesions could not be determined, as previously agreed with the patient, a meticulous laparoscopic left salpingo–oophorectomy was performed using bipolar seizers, and the adnexa was placed intact inside an endoscopic bag before being extracted. Following the procedure, extensive pelvic lavage was performed and samples of the fluid were sent for cytological analysis. Frozen section revealed non-malignant tissue whose origin could not be identified at that stage. All other masses were then laparoscopically removed using the same technique. Final histopathological examination revealed splenic tissue, and diagnosis of pelvic splenosis was determined. The patient made an uneventful recovery, and 1 year following surgery, she was free of abdominal pain. Discussion Road accidents with abdominal trauma and its sequelae are one of the major medical problems in our society. Fewer than 100 cases of splenosis have been reported in the literature, and only the minority of these in the gynaecological literature (Overton, 1982; Auerbach et al. 1985; Belhassen et al., 1987; Stovall and Ling, 1988; Griggs et al., 1990; Higgins and Crain et al., 1995; Matonis and Luciano et al., 1995). Autotransplantation of splenic nodules following spleen trauma has been described in all intraperitoneal and some extraperitoneal sites 1683

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Figure 1. Arrows pointing at splenic nodules (S) on uterine fundus (U).

Figure 2. Arrows pointing at splenic nodules (S) on left ovary (O) and cul-de-sac.

as well (Skinner and Hurteau, 1957; Stovall and Ling, 1988). Although the true incidence of this event is unknown, a clue is given by Muller and Ruthlin (1995), who ultrasonographically followed patients who underwent post-traumatic splenectomy. They presumed splenosis occurred in one-third of their patients, but this was histologically unconfirmed (Muller and Ruthlin, 1995). Presumed diagnosis can be made using the patient history of prior splenectomy or at least severe abdominal trauma, absence of siderocytosis and Howell–Jolly bodies in the blood smear of a patient with history of splenectomy, and by scintigraphy using heat damaged Tc-99m-labelled autologous red blood cells (Atkins et al., 1980). At laparoscopy, these implants are bluish in colour and have no hilus, supplied by local arteries that penetrate their fibrotic capsule. Splenosis may mimic endometriosis, as happened initially in both this and other cases (Matonis and Luciano, 1995). A major difference 1684

between these two entities may be the discrepancy between peritoneal spread and the lack of adhesions in cases of splenosis. Furthermore, splenoses differ in colour and consistency from malignant tumours and in consistency from fibroids. Accessory spleen, which is a common finding in autopsies, is usually single, and is supplied by a branch of the splenic artery (Cohen, 1954). The development of high frequency transvaginal scanning has facilitated the diagnosis of relatively small pelvic masses, and the advancement of laparoscopic surgical techniques may increase the frequency of diagnosing splenosis. Before surgery, an estimation of the potentially malignant nature of the organ should be performed using high frequency transvaginal and transabdominal sonography, Doppler flow waveform measurements, and serum biomarker determinations (Chapron et al., 1996). During laparoscopic surgery, adnexectomy is mandatory whenever there is the slightest suspicion of malignancy. To

Pelvic splenosis mimicking endometriosis

avoid any contamination or peritoneal dissemination if a malignancy is missed, special care must be paid to avoid any cysts, which should then be placed intact inside an endoscopic bag before being extracted, immediately followed by a frozen section examination. Provided that all the above mentioned stages from preoperative assessment and the clinical diagnostic phase during endoscopic surgery are meticulously carried out, laparoscopic surgery is reliable both for diagnosis and management of a benign pelvic mass (Chapron et al., 1996) and the occasional case of pelvic splenosis. However, the most controversial issue is what to do with these splenic nodules, since it is not clear whether they may still provide a protective role against post-splenectomy sepsis. It has been reported that the overall incidence of sepsis and mortality is significantly higher in cases of incidental splenectomy than in post-traumatic splenectomy cases (Brewster, 1973; Singer, 1973). Therefore, we may argue that symptomatology is the key issue. When splenosis is incidentally diagnosed in an asymptomatic patient, complete surgical removal is not indicated. However, the surgical approach is recommended in cases of low abdominal pain or uncertain diagnosis. It is in this respect that operative laparoscopic resection is a new attractive alternative. References Atkins, H.L., Goldman, A.G., Fairchild, R.G. et al. (1980) Splenic sequestration of Tc-99m labeled, heat treated red blood cells. Radiology, 136, 501–503. Auerbach, R.D., Kohorn, E.I. and Cornelius, E.A. (1985) Splenosis: a complicating factor in total abdominal hysterectomy. Obstet. Gynecol., 65, 673–685. Belhassen, A., Aubineau, J.M., Bauverne, B. and Gautier, P. (1987) Splenosis: an unrecognized cause of pelvic pain. J. Gynecol. Obstet. Biol. Reprod. (Paris), 16, 335–338. Brewster, D.C. (1973) Splenosis: report of two cases and review of the literature. Am. J. Surg., 126, 14–19. Buchbinder, J.H. and Lipkopf, C.J. (1939) Splenosis: multiple peritoneal splenic implants following abdominal injury. Surgery, 6, 927–930. Chapron, C., Dubuisson, J., Fritel X. and Rambaud, D. (1996) Diagnosis and management of organic ovarian cysts: indication and procedures for laparoscopy. Hum. Reprod. Update, 2, 435–446. Cohen, E.A. (1954) Splenosis: review and report of subcutaneous splenic implants. Arch. Surg., 69, 777–781. Faltin, R. (1911) Milzartige bildungen in peritoneum, beobachet Ca, 6 jahre snce nach wegen milzuptur vorgenommen splenekfomie. Deutsch. Z. Chir., 110, 160–163. Griggs, J.A., Rudoff, J. and Codington, C.C. (1990) Mayer–Rokitansky– Kuster–Hauser syndrome with splenosis: a case report. J. Reprod. Med., 35, 821–823. Higgins, R.V. and Crain, J.L. (1995) Laparoscopic removal of pelvic splenosis: a case report. J. Reprod. Med., 440, 140–142. Matonis, L.M. and Luciano, A.A. (1995) A case of splenosis masquerading as endometriosis. Am. J. Obstet. Gynecol., 173, 971–973. Muller, U. and Ruthlin, M. (1995) Milzneubildungen nach traumatisch bedingter splenektomie-diagnostik und funktion. Swiss Surg., 5, 230–233. Overton, T.H. (1982) Splenosis: a cause of pelvic pain. Am. J. Obstet. Gynecol., 143, 969–970. Singer, D.B. (1973) Post-splenectomy sepsis. Perspect. Pediatr. Pathol., 1, 285–311. Skinner, E.F. and Hurteau, W. (1957) Autotransplantation of splenosis into the thorax. J. Thorac. Surg., 33, 807–809. Stovall, T.G. and Ling, F.W. (1988) Splenosis: report of a case and review of the literature. Obstet. Gynecol. Surv., 43, 69–72. Received on October 30, 1997; accepted on March 25, 1998

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