Xanthogranulomatous inflammation is an uncommon form of chronic inflammation that is destructive to affected organs; it is characterized by the presence of lipid-filled macrophages with admixed lymphocytes, plasma cells, and neutrophils. Only a few cases of xanthogranulomatous oophoritis have been reported to date. We describe a rare case of xanthogranulomatous oophoritis with involvement of omentum

Bafna, UD

Devi, UK

Gouda, G

Kalloli M, M

Mukherjee, G

Rathod, PS

English

Cognitive Sciences ePrint Archive

 Case Report
A Rare Xanthogranulomatous Oophoritis Presenting as Ovarian Cancer
Authors
Mahesh Kalloli, Uttam D Bafna, Geetashree Mukherjee, Uma Devi K, Gurubasavangouda, Praveen S Rathod,
Department of Gynaecologic Oncology, Kidwai Memorial Institute of Oncology, Bangalore
Address for Correspondence
Dr. Mahesh Kalloli,
No. 5, AB Type, Block-1,
Kidwai Memorial Institute of Oncology campus,
Dr MH Marigowd Road,
Bangalore-29. 
E-mail: mahesh.kalloli@gmail.com
Citation
Kalloli M, Bafna UD, Mukherjee G, Devi UK, Gurubasavangouda, Rathod PS. A Rare Xanthogranulomatous Oophoritis Presenting as 
Ovarian Cancer. Online J Health Allied Scs. 2012;11(2):11. Available at URL:http://www.ojhas.org/issue42/2012-2-11.htm
Open Access Archives
http://cogprints.org/view/subjects/OJHAS.html
http://openmed.nic.in/view/subjects/ojhas.html
Submitted: Apr 20, 2012; Accepted: Jul 3, 2012; Published: Jul 25, 2012
Abstract: Xanthogranulomatous inflammation is an uncommon 
form  of  chronic  inflammation  that  is  destructive  to  affected 
organs;  it  is  characterized  by  the  presence  of  lipid-filled 
macrophages  with  admixed  lymphocytes,  plasma  cells,  and 
neutrophils. Only a few cases of xanthogranulomatous oophoritis 
have  been  reported  to  date.  We  describe  a  rare  case  of 
xanthogranulomatous oophoritis with involvement of omentum.
Key Words: Xanthogranulomatous Oophoritis; Ovarian Cancer; 
Omentum
Introduction
Xanthogranulomatous inflammation is a special form of chronic 
inflammation  that  is  destructive  to  normal  tissue  of  affected 
organs.1 This  is  an  uncommon  process  mostly  affecting  the 
kidney.2 However,  other  organs  in  which xanthogranulomatous 
inflammation  has  been  reported  are  the  gallbladder,  stomach, 
anorectal area, bone, urinary bladder, testis,  epididymis, vagina 
and  endometrium.  Xanthogranulomatous  inflammation  of  the 
female genital  tract is unusual and is essentially limited to the 
endometrium.3Only a few cases involving the ovary have been 
reported.3,4 It  is  characterized  by  a  massive  infiltration  of  the 
tissues  by  lipid-laden  histiocytes  admixed  with  lymphocytes, 
plasma  cells  and  polymorphonuclear  leucocytes.  There  is  no 
minimal  amount  requirement  for  the  histiocytes  to  make  this 
diagnosis. The pathogenesis is unclear. The proposed causes are 
infection,  ineffective  antibiotic  therapy,  abnormality  in  lipid 
metabolism,  endometriosis,  or  ineffective  clearance of  bacteria 
by  phagocytes.  A  combination  of  these  factors  may  be 
responsible. This lesion occurs in patients with recurrent pelvic  
inflammatory disease.4
Clinically  and  radiologically,  Xanthogranulomatous 
inflammation  is  mimicking  tumor  of  the  ovary  and  fallopian 
tube.  Kunakemakorn  was  the  first  to  report  inflammatory 
pseudotumor  in the pelvis  in 1976.5 To date,  only 13 cases of 
xanthogranulomatous  inflammation  in  the  ovary  and  fallopian 
tubes.6 In  this  paper  we  report  a  rare  case  of 
xanthogranulomatous  oophoritis  with  involvement  of  omentum 
with review of the literature on this subject.
Case Report
A 45 year  old woman,  married and a mother  of  two children, 
presented  with  3  months  history  of  intermittent  severe  sharp 
abdominal pain localized to the left suprapubic region and low 
backache. She had had an intrauterine device (IUD) for 4 years  
and experienced an episode of pelvic inflammatory disease after 
the  IUD was  first  inserted.  The patient’s  medical  history  was 
otherwise  unremarkable.  Her  physical  examination  revealed  a 
large mass in left iliac fossa with restricted mobility.
Laboratory  investigation  revealed  a  normal  hemogram  and 
normal liver and renal function and normal levels of CEA and 
CA-125. Pelvic ultrasound showed a large lobulated lesion in left 
adnexa. MRI abdomen and pelvis shown a large 8.7×5.1×7.7 cm 
enhancing mass lesion involving the left ovary with peri ovarian 
enhancing  fascial  thickening  involving  the  adjescent  uterus, 
bladder  and  peri  rectal  fascia  with  left  sided 
hydroureteronephrosis which was thought to represent either an 
inflammatory mass or an ovarian neoplasm.
Patient  was  taken  for  exploratory  laparotomy.  During  the 
surgery,  it  was  noted  that  the  left  adnexal  structures  were 
involved with an 8 to 8 cm mass adherent to omentum. Frozen 
section  report  came as  inflammatory  lesion  with  fibrosis.  The 
patient underwent a total abdominal hysterectomy and bilateral 
salpingooophorectomy with total omentectomy. The patient had 
an uneventful recovery and was discharged on the postoperative 
day 6.
The  histopathology  of  the  surgical  specimens  revealed 
Xanthogranulomatous  inflammation  of  ovary  and  omentum 
(Figure  1  and  2).  Microscopy  showed  Patient  was  referred to 
urologist  for  left  ureteric  stenting.  Now  patient  is  on  regular 
follow up.
1
This work is licensed under a
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No Derivative Works 2.5 India License
Online Journal of Health and Allied Sciences
Peer Reviewed, Open Access, Free Online Journal
Published Quarterly :  Mangalore, South India : ISSN 0972-5997
Volume 11, Issue 2; Apr-Jun 2012
Figure 1: Microscopic picture showing ovarian structure in 
the background of dense chronic inflammatory cells 
(hematoxylin-eosin)
Figure 2: Lipid-containing macrophages and mixed 
inflammatory infiltrate (hematoxylin-eosin).
Discussion
Xanthogranulomatous inflammation is a special form of chronic 
inflammation  that  is  destructive  to  normal  tissue  of  affected 
organs.1 This  is  an  uncommon  process  mostly  affecting  the 
kidney.2 The  affected  organs  suffer  disorganization  and 
infiltration  with  focal  or  sheets  of  foam  cells  admixed  with 
chronic  inflammatory  cells such as  lymphocytes,  plasma cells, 
occasional neutrophils with or without multinucleated or Touton 
giant  cells.  Xanthogranulomatous  inflammation  occurring  in 
female  genital  tract  affects  endometrium,  fallopian  tubes  or 
ovaries  focally  or  entirely,  which  clinically  forms  mass  like  
lesion in the pelvic cavity and invades the surrounding tissues. 
Only  13  related  cases  of  Xanthogranulomatous  inflammation 
involving  fallopian  tube  or  ovary  have  been described so  far. 
Only a few cases have been reported from india.7 The clinical 
manifestation, imaging detection and macroscopic observation of 
xanthogranulomatous oophoritis are subject to be confused with 
ovarian malignancy. Eight reported cases as well as ours were all 
misdiagnosed as ovarian cancers.
The average age of patients with affected ovaries is 31 years. The 
involved  ovary  in  each  of  the  previously  reported  cases  was 
replaced  by  a  solid,  yellow,  lobulated  mass  that  was  well 
circumscribed  and  consisted  of  xanthogranulomatous 
inflammation.8 Presenting complaints  were  lower  abdominal  or 
suprapubic  pain  (sometimes  unilateral),  fever,  menorrhagia,  or 
vaginal  bleeding.  On physical  examination,  there  was  adnexal 
tenderness  and  a  pelvic  mass.  The  pathogenesis  of 
xanthogranulomatous  inflammation  remains  unclear.  Many 
unrelated disorders may have the same mechanism of foam cell  
production 9, proposed causes are infection, ineffective antibiotic 
therapy,  abnormality  in  lipid  metabolism,  endometriosis,  and 
ineffective clearance of bacteria by phagocytes.  A combination 
of  factors  may  be  responsible.  For  example,  bleeding  and 
obstruction may predispose to infection, tissue necrosis  occurs, 
followed  by  the  release  of  cholesterol  and  other  lipids  and 
phagocytosis by macrophages.9
Wather10 presumed  malakoplakia  and  xanthogranulomatous 
inflammation  were  identical  chronic  inflammatory  disease.  In 
malakoplakia,  the  cytoplasmic  concentric  calcific  bodies 
(Michaelis-Gutmann bodies)  are phagolysosome  of  phagocytes 
which calcifies after phagocytes phagocytise E. coli but fail to 
digest it. Malakoplakia occurs mainly in the urinary system, but 
xanthogranulomatous inflammation occurs mainly in the genital 
system.
Presence of  nonenhancing intramural  nodules  in  the  thickened 
wall of an ovarian cystic mass may be a unique MR indicator of 
xanthogranulomatous oophoritis. Although a correct diagnosis is 
made chiefly through histology. Xanthogranulomatous oophoritis 
is  often  misdiagnosed  by  pathologists  if  they  don’t  keep  this 
entity in mind. This may be due to the rarity of the condition. If  
the  lesion  is  mainly  focal  scattered  lymphocytes,  it  may  be 
misdiagnosed  as  secondary  lymphoma  or  leukemia.  If  the 
lymphocytes are scattering diffusely and foam cells are seldom, a 
diagnosis  of  malignant  small  cell  tumor  with  stromal 
luteinization  may  be  rendered.  If  there  are  small  amount  of 
obvious fibrosis and foam cells, a diagnosis of sclerosing stromal 
tumor may be made. However, the right diagnosis is possible to 
be made as long as pathologists elevate vigilance and master the 
pathological features. Immunohistiochemical stains are helpful in 
establishing the diagnosis, including CD68 (foam cells positive), 
CD3 (T lymphocyte marker), CD20 (B lymphocytes marker), ? 
and ? (both positive in polyclonal B lymphocytes).
The treatment of choice for Xanthogranulomatous oophoritis is 
oophorectomy. Since xanthogranulomatous oophoritis is usually 
associated  with  pelvic  inflammatory  disease  (PID), 
endometriosis,  intrauterine  death etc.,  these  patients  should  be 
followed up closely. Although a correct diagnosis is made chiefly 
through  histology,  a  suggestive  preoperative  diagnosis  of 
xanthogranulomatous  oophoritis  could  lead  to  less  radical 
surgery.
References
1. Franco  V,  Florena  AM,  Guarneri  G,  Gargano  G. 
Xanthogranulomatous  salpingitis:  case  report  and review 
of the literature. Acta Eur Fertil. 1990;21:197–199.
2. Goodman  M, Curry T,  Russell  T. Xanthogranulomatous 
pyelonephritis  (XGP):  a  local  disease  with  systemic 
manifestations:  report  of  23  patients  and  review  of 
literature.Medicine. 1979;58:171–181.
3. Jung  SE,  Lee  JM,  Lee  KY,  Han  KT,  Hahn  ST. 
Xanthogranulomatous  oophoritis:  MR  imaging  findings 
with  pathologic  correlation. Am  J 
Roentgenol 2002;178:749–751.
4. Gray Y, Libbey NP. Xanthogranulomatous salpingitis and 
oophoritis: a case report and review of the literature. Arch 
Pathol Lab Med 2001;125:260–263.
5. Kunakemakorn  P,  Ontai  H.  Pelvic  inflammatory 
pseudotumor:  A  case  report. Am  J  Obstet  Gynecol. 
1976;26:286-287.
6. Zhang XS, Dong HY, Zhang Ll,  Desouki  MM, Zhao C. 
Xanthogranulomatous Inflammation of the Female Genital 
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7. Shukla  S,  Pujani  M,  Singh  SK,  Pujani  M. 
Xanthogranulomatous  oophoritis  associated with  primary 
infertility  and  endometriosis. Indian  J  Pathol  
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8. Clement  PB.  Nonneoplastic  lesions  of  the  ovary.  In: 
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genital  tract,  4th ed. New York:  Springer-Verlag,  1994. 
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A Rare Xanthogranulomatous Oophoritis Presenting as Ovarian Cancer

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A Rare Xanthogranulomatous Oophoritis Presenting as Ovarian Cancer

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