Xanthogranulomatous inflammation of myometrium with uterine perforation Available online at www.sciencedirect.com ScienceDirect Taiwanese Journal of Obstetrics & Gynecology 52 (2013) 428e430 www.tjog-online.com Research Letter Xanthogranulomatous inflammation of myometrium with uterine perforation Chi-Yuan Liao a,*, Cheng-Hui Chiu b , Fuh-Jinn Luo c a Department of Obstetrics and Gynecology, Mennonite Christian Hospital, Hualien, Taiwan b Department of Radiology, Mennonite Christian Hospital, Hualien, Taiwan c Department of Pathology, Mennonite Christian Hospital, Hualien, Taiwan Accepted 17 December 2012 Xanthogranulomatous inflammation of the female genital tract is unusual and is essentially limited to the endometrium [1,2] and the fallopian tubes [3e6]. We report a rare case of xanthogranulomatous inflammation of the myometrium. This case is of interest because the xanthogranulomatous inflam- mation destroyed the myometrium and further extended to the pelvis with uterine perforation. The findings were first seen on ultrasonographic images and computed tomography (CT) with pathological confirmation after a surgical operation. A 66-year-old married female, gravid 2, parity 2, with a history of poorly controlled diabetes, presented to the emer- gency room with complaints of lower abdominal pain, flank pain, and an intermittent fever of 1 week duration. She was febrile with a temperature of 38.2 �C. Physical examination showed a mildly enlarged uterus with tenderness on the right adnexa. Ultrasonography revealed an enlarged uterus with pyometra, suspected uterine perforation, and a right tuboovarian abscess (Fig. 1B). Abdominal CT revealed pyometra and a pelvic abscess with a suspected uterine wall defect (Fig. 1A). The laboratory data showed the following detailsdwhite blood cell count, 23.63 THSD/mL; neutrophil, 86.6%; C-reactive protein, 18.30 mg/dL; and glucose spot, 536 mg/dL. The patient was admitted and received an antibiotic, Cefmetazole (Daiichi Sankyo, Tokyo, Japan) 2 gm IV Stat, followed by 1 gm IV Q6H. She then underwent an endometrial aspiration and Foley catheter drainage for her pyometra, which was acutely inflamed with numerous polymorphonuclear leukocytes and necrotic debris. Accordingly, she received a laparoscopic surgery. A thick adhesion mass about 5 cm in diameter on the * Corresponding author. Department of Obstetrics and Gynecology, Mennonite Christian Hospital, Hualien, Number 44, Minchuan Road, Hualien City 970, Taiwan. E-mail address: mchliaochiyuan@yahoo.com.tw (C.-Y. Liao). 1028-4559/$ - see front matter Copyright � 2013, Taiwan Association of Obstetri http://dx.doi.org/10.1016/j.tjog.2012.12.003 right adnexadaffecting the sigmoid colon, omentum, right tube, and ovarydwas found. Pus was found, and a culture was conducted. Adhesion lysis was performed, and a hole approximately 2.5 cm in diameter was observed on the right uterine fundus. A laparoscopy-assisted vaginal hysterectomy was performed smoothly. The culture reports from the blood, pyometra, and right adnexa were all negative. The patho- logical report of the uterus revealed localized xanthogranu- lomatous inflammation at the right uterine fundus with perforation (Fig. 2A). Postoperative antibiotic with Cefme- tazole 1 gm IV Q6H was administered. Drainage using the JacksonePratt drain was carried out. The patient recovered without incident. Xanthogranulomatous inflammation is rare. It mainly in- volves the kidneys or the gall bladder [1,4e7]. Primary xan- thogranulomatous inflammation that involves the female genital organ has been reported in only a few cases, and it usually af- fects the endometrium or fallopian tubes. However, to date no case of myometrium involvement with uterine perforation has been reported. Xanthogranulomatous inflammation is a form of chronic inflammation that is destructive to the affected tissue, as in the case reported here, in which the myometrium was replaced with lipid-laden, foamy macrophages (xanthoma cells) and polymorphonuclear leukocytes, lymphoplasma cells (Figs. 2A and B). The etiology of the disease has not been fully elucidated. The proposed causes include occlusion of the cer- vical canal, hemorrhage, and infection [7,8]. Multiple factors may be responsible. In the case considered herein, cervix obstruction and infection with tissue necrosis occurred, fol- lowed by the release of cholesterol and other lipids. Therefore, phagocytosis by the macrophage may explain the formation of foam cells that is associated with xanthogranulomatous inflammation. In some cases, bacteria including Proteus mir- abilis or Escherichia coli have been identified [9]. However, very often, neither organism [10] is identified, as in our case. cs & Gynecology. Published by Elsevier Taiwan LLC. All rights reserved. mailto:mchliaochiyuan@yahoo.com.tw http://crossmark.crossref.org/dialog/?doi=10.1016/j.tjog.2012.12.003&domain=pdf www.sciencedirect.com/science/journal/10284559 http://dx.doi.org/10.1016/j.tjog.2012.12.003 http://dx.doi.org/10.1016/j.tjog.2012.12.003 http://dx.doi.org/10.1016/j.tjog.2012.12.003 http://www.tjog-online.com Fig. 1. (A) Oblique-sagittal reformation imaging showed dilated uterine cavity (arrowhead). Presence of a defect of myometrium at right anterior aspect (bold arrow) and abscess formation (arrow). (B) Ultrasonography revealed a perforation hole, about 2.5 cm in diameter, that was enclosed with an echogenic, septate mass 4 cm � 2.5 cm in diameter on the fundal region. 429C.-Y. Liao et al. / Taiwanese Journal of Obstetrics & Gynecology 52 (2013) 428e430 In our case, the histological involvement of xanthogranu- lomatous inflammation is present only in the myometrium with transmural extension to the peritoneal cavity. However, the endometrium and fallopian tubes were simply present with acute and chronic inflammation (Fig. 2A). This characteristic, Fig. 2. (A) Xanthogranulomatous inflammation of the uterus. The myometrium disp inflammation. (hematoxylineeosin, original magnification, �40). (B) High magnifi and neutrophils (hematoxylineeosin; original magnification, �400). (C) Periodic ac �400). pathological phenomenon associated with uterine perforation is similar to the formation of fistula in xanthomatous pyelo- nephritis and xanthomatous cholecystitis. Most cases of xan- thogranulomatous endometritis resolve spontaneously or after antibiotic treatment [7,8,11]. In the case presented here, lays xanthogranulomatous inflammation and overlying endometrium with usual cation reveals abundant foamy histiocytes, admixture of lymphoplasma cells id Schiff staining reveals no MichaeliseGutmann body (original magnification 430 C.-Y. Liao et al. / Taiwanese Journal of Obstetrics & Gynecology 52 (2013) 428e430 xanthogranulomatous inflammation destroyed the myome- trium and extended to the pelvis with uterine perforation, as revealed by the ultrasonographic images and CT scan, which is an exceptional occurrence. Whether the patient’s poorly controlled diabetes is a contributing factor is not known. The most important differential diagnosis of xanthogra- nomatous inflammation is malakoplakia. The histopatholog- ical diagnosis of malakoplakia depends on the presence of intracellular and extracellular laminated inclusions, the so- called calcispherites or MichaeliseGutmann bodies, and spe- cial foamy histiocytes, which are called von Hansemann cells [12]. In the case reported here, the lack of a Michae- liseGutmann body in histochemical periodic acid Schiff staining almost excludes this diagnosis (Fig. 2C). In conclusion, a rare case xanthomatous inflammation of the myometrium with uterine perforation associated with fe- male genital organ infection is presented. A poorly controlled diabetes with a very high level of spot sugar (536 mg/dL) was noted. Whether it was a causal factor is uncertain. To the best of our knowledge, this is the first report of a xanthomatous inflammation of the myometrium with uterine perforation in a patient with poorly controlled diabetes. Surgeons will recog- nize the possibility of xanthomatous inflammation in pelvic inflammatory disease with pyometra, especially in patients with uncontrolled diabetes. Acknowledgment The authors thank Yinju Chen, MHS, Department of Medical Research and Education, Mennonite Christian Hos- pital, Hualien, for the writing and editing assistance in this manuscript. References [1] Noack F, Briese J, Stellmacher F, Hornung D, Horny HP. Lethal outcome in xanthogranulomatous endometritis. APMIS 2006;114:386e8. [2] Lopez JI, Nevado M. Exuberant xanthogranulomatous-like reaction following endometrial curettage. Histopathology 1989;15:315. [3] Yener N, Ilter E, Midi A. Xanthogranulomatous salpingitis as a rare pathologic aspect of chronic active pelvic inflammatory disease. Indian J Pathol Microbiol 2011;54:141e3. [4] Howey JM, Mahe E, Radhi J. Xanthogranulomatous salpingitis asso- ciated with a large uterine leiomyoma. Case Rep Med 2010;2010:970805. [5] Idrees M, Zakashansky K, Kalir T. Xanthogranulomatous salpingitis associated with fallopian tube mucosal endometriosis: a clue to the pathogenesis. Ann Diagn Pathol 2007;11:117e21. [6] Gray Y, Libbey NP. Xanthogranulomatous salpingitis and oophoritis: a case report and review of the literature. Arch Pathol Lab Med 2001;125:260e3. [7] Ladefoged C, Lorentzen M. Xanthogranulomatous inflammation of the female genital tract. Histopathology 1988;13:541e51. [8] Russack V, Lammers RJ. Xanthogranulomatous endometritis. Report of six cases and a proposed mechanism of development. Arch Pathol Lab Med 1990;114:929e32. [9] Barua R, Kirkland JA, Petrucco OM. Xanthogranulomatous endometritis: case report. Pathology 1978;10:161e4. [10] Buckley CH, Fox H. Histiocytic endometritis. Histopathology 1980;4:105e10. [11] Blanco C, Fernández F, Buelta L, Garijo F, Val-Bernal JF, Sánchez S. Xanthomatous endometritis. Appl Pathol 1989;7:273e6. [12] Chou SC, Wang JS, Tseng HH. Malacoplakia of the ovary, fallopian tube and uterus: a case associated with diabetes mellitus. Pathol Int 2002;52:789e93. Xanthogranulomatous inflammation of myometrium with uterine perforation Acknowledgment References