Xanthogranulomatous Oophoritis – A Case Report
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Journal of Chalmeda Anand Rao Institute of Medical Sciences Vol 12 Issue 2 July - December 2016 ISSN (Print) : 2278-5310 104 Xanthogranulomatous Oophoritis – A Case Report Sravanthi Yerragolla 1 , Sumalatha Kasturi 2 , Renuka Barkam 3 , Vinuthna C 4 , Sravani Ramaa Modey 5 , Ravindra Thota 6 1 Associate Professor 2 Professor Department of Pathology Chalmeda Anand Rao Institute of Medical Sciences Karimnagar - 505 001 Telangana, India. CORRESPONDENCE : 1 Dr.Sravanthi.Y M.D. (Pathology) Post graduate student. Department of Pathology Chalmeda Anand Rao Institute of Medical Sciences Karimnagar - 505 001 Telangana, India. E-mail: [email protected] Case Report ABSTRACT Xanthogranulomatous inflammation is an uncommon form of chronic inflammation that is destructive of normal tissue in affected organs. Most commonly affected organs are kidney and gallbladder followed by anorectal area, bone, stomach and testis. This inflammation most commonly affects the endometrium in the female genital tract but involvement of vagina, cervix, fallopian tube and ovary may also occur. Only 16 cases have been reported in literature involving the ovary. We report a case of 25 year old pregnant women presented with pain abdomen since 4 days at 35 weeks of gestation. Emergency LSCS was done along with left ovarian cystectomy. Histopathological examination revealed xanthogranulomatous oophoritis. This case is of interest in view of its rarity also due to its clinical and radiological resemblance to neoplastic lesion. Keywords: Xanthogranulomatous oophoritis , oophorectomy, chronic oophoritis INTRODUCTION Xanthogranulomatous inflammation is an uncommon form of chronic inflammation in which the affected organ is destroyed and is replaced by large number of lipid- containing macrophages with an admixture of lymphocytes, plasma cells and multinucleated giant cells. [1] Most commonly affected organs are kidney followed by gall bladder. [2] Other organs in which xanthogranulomatous inflammation has been reported are stomach, anorectal area, bone, urinary bladder, testis, epididymis and female genital tract. [4] Only 16 cases have been reported in the literature involving the ovary, [2,4] which will clinically presents as mass in the pelvic cavity and mimics an ovarian tumour clinically, radiologically and macroscopically. CASE REPORT A 25 year old G2P1L1 pregnant women presented with lower abdominal pain since 4 days at 35 weeks gestation. Physical examination showed tenderness in left iliac fossa. Ultrasound scan revealed 14x7x6 cms size biloculated hypoechoic mass suggestive of left complex ovarian cyst[Figure 1]. Patient underwent emergency LSCS along with left ovarian cystectomy. Intraoperatively dense adhesions with omentum , bowel wall, uterus posterior wall and peritoneum were noted. Cyst wall rupture yielded 400ml of thick purulent fluid which was sent for culture and reported as positive for Escherichia coli.
Transcript of Xanthogranulomatous Oophoritis – A Case Report
Journal of Chalmeda Anand Rao Institute of Medical Sciences Vol 12 Issue 2 July - December 2016 ISSN (Print) : 2278-5310 104
Xanthogranulomatous Oophoritis – A
Case Report
Sravanthi Yerragolla1, Sumalatha Kasturi2, Renuka Barkam3, Vinuthna
C4, Sravani Ramaa Modey5, Ravindra Thota6
1 Associate Professor2 ProfessorDepartment of PathologyChalmeda Anand RaoInstitute of Medical SciencesKarimnagar - 505 001Telangana, India.
CORRESPONDENCE :
1 Dr.Sravanthi.YM.D. (Pathology)Post graduate student.Department of PathologyChalmeda Anand RaoInstitute of Medical SciencesKarimnagar - 505 001Telangana, India.E-mail:[email protected]
Case Report
ABSTRACT
Xanthogranulomatous inflammation is an uncommon form of chronic inflammation thatis destructive of normal tissue in affected organs. Most commonly affected organs arekidney and gallbladder followed by anorectal area, bone, stomach and testis. Thisinflammation most commonly affects the endometrium in the female genital tract butinvolvement of vagina, cervix, fallopian tube and ovary may also occur. Only 16 caseshave been reported in literature involving the ovary.
We report a case of 25 year old pregnant women presented with pain abdomen since 4days at 35 weeks of gestation. Emergency LSCS was done along with left ovarian cystectomy.Histopathological examination revealed xanthogranulomatous oophoritis. This case is ofinterest in view of its rarity also due to its clinical and radiological resemblance to neoplasticlesion.
Keywords: Xanthogranulomatous oophoritis , oophorectomy, chronic oophoritis
INTRODUCTION
Xanthogranulomatous inflammation is an uncommonform of chronic inflammation in which the affected organis destroyed and is replaced by large number of lipid-containing macrophages with an admixture oflymphocytes, plasma cells and multinucleated giantcells.[1] Most commonly affected organs are kidneyfollowed by gall bladder.[2]
Other organs in which xanthogranulomatousinflammation has been reported are stomach, anorectalarea, bone, urinary bladder, testis, epididymis and femalegenital tract.[4] Only 16 cases have been reported in theliterature involving the ovary,[2,4] which will clinicallypresents as mass in the pelvic cavity and mimics an
ovarian tumour clinically, radiologically andmacroscopically.
CASE REPORT
A 25 year old G2P1L1 pregnant women presented withlower abdominal pain since 4 days at 35 weeks gestation.Physical examination showed tenderness in left iliac fossa.Ultrasound scan revealed 14x7x6 cms size biloculatedhypoechoic mass suggestive of left complex ovariancyst[Figure 1]. Patient underwent emergency LSCS alongwith left ovarian cystectomy. Intraoperatively denseadhesions with omentum , bowel wall, uterus posteriorwall and peritoneum were noted. Cyst wall ruptureyielded 400ml of thick purulent fluid which was sent forculture and reported as positive for Escherichia coli.
Sravanthi Yerragolla et. al
Journal of Chalmeda Anand Rao Institute of Medical Sciences Vol 12 Issue 2 July - December 2016 105
Figure 1: Ultrasound examination revealed 14.1 x 6.7cms size
biloculated hypoechoic lesion suggestive of left complex ovarian
cyst.
Figure 2: Gross examination showing enlarged ovary showing
nodular & cystic external surface.
Figure 3: Cut surface showing solid and cystic multiloculated mass.
The solid areas are yellowish in colour.
Figure 4 : x100, H&E, Histological sections from solid areas show
dense infiltration of inflammatory cells with foci of necrosis
Figure 5: x400, H&E, High power image showing sheets of foamy
histiocytes.
We received a specimen of ovarian mass measuring12x7x5cms. Gross examination showed grey brown togrey white nodular cystic mass [Figure 2]. Cut surfacewas partly solid and cystic with multiple loculae andyellowish solid areas [Figure 3]. Histologically sectionsfrom solid areas show dense infiltration of inflammatorycells [Figure 4], predominantly composed of foamyhistiocytes [Figure 5] neutrophils, lymphocytes andoccasional multinucleated giant cells and areas ofnecrosis. There was no evidence of malignancy in thesections studied.
DISCUSSION
Xanthogranulomatous inflammation is a form of chronicinflammation that is destructive to the normal tissue ofaffected organs.[3] Xanthogranulomatous inflammation is
a well documented entity in gall bladder and kidney.However, Xanthogranulomatous inflammation of thefemale genital tract is unusual and essentially limited tothe endometrium. Only a few cases involving theovary,fallopian tubes and vagina have been reported .[4,5]
According to the literature 16 cases involving the ovaryhave been reported until 2012.[6] Kunakemakorn was thefirst to describe xanthogranulomatous inflammation ofserosa of the uterus, left fallopian tube and ovary in hisreport of inflammatory pseudotumor in the pelvis in 1976.[7] The average age of occurance is 31 years and theyoungest case reported was of 18 years.
Xanthogranulomatous oophoritis (XGO) has an uncertainetiopathogenesis. Various factors implicated are chronicbacterial infection with E.coli, Proteus and Staphylococcusaureus, inadequate antibiotic therapy, endometriosis,intrauterine device and Inborn errors of lipid metabolismin macrophages.[8] In the present case no predisposingfactors were identified except the history of chronic pelvicinflammatory disease. Chronic or recurrent forms ofovarian involvement by pelvic inflammatory disease maytake the form of chronic perioophoritis with peri ovarianand tubo ovarian adhesions as seen in the case of ourpatient.
Chronic ovarian abscess may result in a solid tumor likemass, variably designated as ovarian xanthogranuloma,Xanthogranulomatous oophoritis, or inflammatorypseudotumor. [9] XGO presents clinically as mass in thepelvic cavity and invades the surrounding tissues whichcan be misdiagnosed as neoplastic lesion and this mayalso be due to the rarity of the condition.[3,6]
The involved ovary in XGO is replaced by a solid , yellow,lobulated mass characterised macroscopically by a wellcircumscribed, solid, yellowish, lobulated mass and canalso present with cystic lesion at times due to liquefactivenecrosis, microscopically by foamy histiocytes(xanthomacells) admixed with multinucleated giant cells, plasmacells, fibroblasts, neutrophils, foci of necrosis andfibrosis.[3,9] The yellowish coloration of this condition isdue to the presence of foam cells [3,6,7]
Chronic infection leads to tissue necrosis andcontinuously releases cholesterol and other lipids fromdead cells, these cellular components are phagocytosedby macrophages, leading to xanthomatous process is alsoa possible explanation of this entity.[10,11]
Clinical presentations include anemia, anorexia, fever,menorrhagia and pain abdomen. Gynaecologicalexamination reveal adnexal mass with tenderness andradiologically mimics ovarian tumor.[3] In our case, patienthas presented with recurrent pain abdomen complicatingpregnancy. Shukla et al.have reported a case of XGO
associated with primary infertility and endometriosis.[12]
Singh et al. have reported premature ovarian failure as arare sequelae of XGO.[13] Punia et al. have reported a caseof XGO and salpingitis as a late sequelae of inadequatelytreated staphylococcal pelvic inflammatory disease.[14]
Cases of xanthogranulomatous inflammation of ovarywith ovarian hemangioma,[15] secondary todiverticulitis,[16] association with endometriosis anduterine leiomyoma,[17] secondary to talcum powder,[18]
presenting as an unusual complication of typhoid[19] andfollowing uterine artery embolization [20] have beenreported.
Treatment of choice for XGO is oophorectomy. Thoughcystectomy was attempted in our case during secondtrimester of pregnancy the recurrent ovarian mass wasprobably due to inadequate antibiotic therapy. Awarenessof this inflammatory lesion is important to the pathologistand treating surgeon to prevent extensive surgery andalso over diagnosis as malignancy.
CONCLUSION
Xanthogranulomatous oophoritis is a rare inflammatorylesion involving the ovary which usually presents withpelvic mass and abdominal pain. The clinical andradiological features may mimic an ovarian neoplasm,so it must be considered in the differential diagnosis ofovarian tumors. Since XGO is usually associated withpelvic inflammatory disease[PID], endometriosis ,intrauterine death etc., these patients should be followedup closely. Although a correct diagnosis is chiefly madethrough histology, a suggestive preoperative diagnosisof XGO could lead to less radical surgery.
CONFLICT OF INTEREST :The authors declared no conflict of interest
FUNDING : None
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