(7) Upsala J Med Sci 111 (2): 227–230, 2006 Malacoplakia and Spermatic Granuloma Complicating Vasectomy 1Margrét Agnarsdóttir, 2Birgitta Carlén and 1Roger Willén 1Pathology and Cytology, Centre for Laboratory Medicine, Uppsala University Hospital, S-75185 Uppsala, Sweden. 2Department of Pathology, Lund University Hospital, 22185 Lund, Sweden. ABSTRACT Malacoplakia is a granulomatous disease with a histiocytic infiltrate containing cal- cified structures called Michaelis-Gutmann bodies. These structures are considered to represent an abnormal response to infection involving defective lysosomes and abnormal microbubular assembly. The disease most frequently involves urinary and genital tracts, but has also been described from most other organs. Here we present the first case of malacoplakia only involving the vas deferens. INTRODUCTION Malacoplakia is a granulomatous disease, which most frequently involves the urinary and genital tract but has also been described from most other organs [1,2]. The disease is characterized microscopically by a histiocytic infiltrate (the so-called von Hanse- manns cells) containing calcified structures named Michaelis-Gutmann bodies. These can be seen both intra- and extracellularily and are considered to represent an abnormal response to infection involving defective lysosomes and abnormal microtubular assem- bly [3]. The first article describing the disease was published in 1902 [4]. Urinary organs as targets usually involve the urinary bladder and testis with sometimes engage- ment of the epididymis [5]. Sole engagement of the epididymis without concurrent involvement of the testis is reported in 9 cases [5]. We have not been able to find a case of malacoplakia solely targeting the vas deferens. We thus find it of interest to report one case, as besides spermatic granuloma the firm fibrous lump palpated clini- cally might simulate tumour or other forms of specific inflammatory states. MATERIALS AND METHODS The surgical specimens were fixed in formalin and routinely processed. Paraffin sections were stained with hematoxylin and eosin (HE), periodic acid-Schiff stain (PAS) for mucin, Prussian blue for iron and von Kossa´s stain for calcium. 227 Received 17 May 2005 Accepted 17 August 2005 For electron microscopy paraffin-embedded, formalin-fixed material was used. Using light microscopy the area in question was marked and the corresponding field on the paraffin block taken out. A very thin slice (0.25mm) was deparaffinized in xylene for 1.5h in a 60oC oven, thereafter rinsed in fresh xylene again and kept in room temperature for another 30 min. The deparaffinized tissue was cut into small cubes and immersed in 100% “blue” ethanol (0.5% methyleneblue) then in 99.5% ethanol followed by propy- lene oxide for 30 min each. The tissue was then infiltrated in propylene oxide/resin 1:1 for 2h and in pure resin for 1.5h, polymerized over night in a 60oC oven. Ultrathin sec- tions were cut and stained with 4% uranyl acetate dissolved in distilled water for 30 min in 40oC, washed with distilled water and then stained with Reynolds lead citrate [6] for 2 min in room temperature. The grids were examined in a Philips CM10 electron micro- scope at 60kV. CASE REPORT A 39-year old man presented with a tumour in the right vas deferens, which he had noted since 1998. Before that the patient had undergone ligation of the vas defer- ens. The tumour was removed in 2004. Macroscopically a cystic lesion was present with a diameter of 2 cm. The cyst was filled with a brownish material. Microscopic examination showed a benign cyst in the vas deferens covered with a fibrotic cap- sule. In the cyst wall a spermatic granuloma was observed containing sperms and a granulomatous inflammatory reaction was also present. Focally, an area in the outer part of the wall contained granulated histiocytes (Fig. 1). These granules stained positive with iron (Fig. 2a), PAS (Fig. 2b) and von Kossa´s staining (Fig. 2c). Elec- tron microscopy confirmed the presence of the characteristic Michaelis-Gutmann bodies (Fig. 3a and 3b). 228 Fig 1. The area in the wall of the cyst containing granulated histiocytes. DISCUSSION Malacoplakia of the vas deferens seems to be a rare disease as we were not able to find a published case solely targeting the vas deferens. The histopathological appearance is characteristic and is easily recognized. As malacoplakia is a disease that can affect 229 Fig 2. The Michaelis-Gutmann bodies staining positive with iron (a), PAS (b) and von Kossa´s (c) staining. Fig 3. An electron microscopic picture showing the characteristic Michaelis-Gutmann bodies (a and b). many organs it is important to know that this entity exists. Not only for histopatholo- gists but also for clinicians as the macroscopic appearance can easily simulate a malig- nant process and specific infections as e.g. tuberculosis. Spermatic granuloma is a well-known complication after the vasectomy procedure [7, 8], but it is also important to notice whether malacoplakia is present as those lesions represent a septic component of the granuloma lesion. ACKNOWLEDGEMENTS We are grateful to Frank Bittkowski for his skilled photographic help. The Uppsala University Hospital foundation for clinical research (ALF) and the Björnssons foun- dation supported this study. REFERENCES 1. Stanton M-J, Maxted W (1981). Malacoplakia: a study of the literature and current concepts of pathogenesis, diagnosis and treatment. J Urol 125:139-146. 2. McClure J (1983). Malakoplakia. J Pathol. Aug; 140: 275-330. 3. Dasgupta P, Womack C, Turner A-G, Blackford H-N (1999). Malacoplakia: von Hansemann´s disease. BJU Int 84:464-469. 4. Michaelis L, Gutmann C (1902). Über Einschlüsse in Blasentumoren. Ztschr Klin Med 47:208- 215. 5. Dieckmann K-P, Henke R-P, Zimmer-Krolzig G (1995). Malacoplakia of the epididymis. Report of a case and review of the literature. Urol Int 55:222-225. 6. Reynolds ES (1963). The use o lead citrate in high pH as an electron-opaque stain in electron microscopy. J Cell Biol 17:208-212. 7. Gade J ,Brasso K (1990). Sperm granulomata. Ugeskr Laeger 152:2228-2284. 8. Davis JE (1992). Male sterilization. Curr Opin Obstet Gynecol 4:522-526. Corresponding author: Roger Willén MD, PhD Pathology and Cytology Centre for Laboratory Medicine Uppsala University Hospital S-75185 Uppsala Sweden Phone: +46-18-6119408 E-mail: roger.willen@akademiska.se 230