A Very Rare Case of XanthogranulomatousSalphingitis in One Tube and Pseudoxanthomatous Salphingitis in Contralateral Tube in a Patient 209 Case Report 1 2 3 Saranya.B , Jayanthi Kamakshi.N , Thamilselvi.R ABSTRACT Xanthogranulomatous salphingitis and pseudoxanthomatous salphingitis are rare forms of chronic inflammation of the fallopian tube with many etiologic factors implicated in its pathogenesis. A 26 year old female was operated for a right ovarian cyst. Ovarian cyst with bilateral fallopian tubes were removed since both the fallopian tubes were dilated and adherent to the adjacent tissues. One tube showed features of Xanthogranulomatous salphingitis and other pseudoxanthomatous salphingitis. Ovarian cyst was a corpus luteal cyst. Although the exact etiology for this case was not clear, endometriosis can be thought to have a contributory role in the pathogenesis as the patient had severe dysmenorrhoea and severe adhesions were noted intraoperatively. Both Xanthogranulomatous salphingitis and pseudoxanthomatous salphingitis were reported separately by many authors. But, both these conditions in one patient to my knowledge is not yet reported in literature. Hence, this case is reported for its rarity. INTRODUCTION: anthogranulomatous salphingitis is a rare form of chronic inflammation which xcauses destruction to the normal tissue of many organs. On histology, it is characterised by the presence of numerous lipid containing macrophages with an admixture of lymphocytes, plasma cells and neutrophils. Multinucleated giant (1-4) cells may be present . Kidney is the most commonly affected organ followed by gall bladder. Other organs affected are stomach, anorectal region, bone, urinary bladder, testis and epididymis. Female genital tract involvement is relatively uncommon and it is mostly confined to the endometrium. However, rare involvement of vagina, cervix, fallopian tube and ovary have been (5-7) documented. As elsewhere in the body, the mechanism of this form of inflammatory response is not clear. Predisposing conditions for the development of XGS include Pelvic Inflammatory Disorder, the presence of an IUD, endometriosis, and inborn error in lipid metabolism by macrophages, unsuccessful antibiotic therapy, and radiotherapy. Pseudoxanthomatous salphingitis is another rare form of chronic inflammation characterised by the presence of pigmented histiocytes within the lamina propria. It is associated with long standing endometriosis. Only a few cases of Xanthogranulomatous salphingitis and pseudoxanthomatous salphingitis have been reported in literature separately by authors. But Xanthogranulomatous salphingitis and pseudoxanthomatous salphingitis in one patient is not yet reported to the best of my knowledge. Address for correspondence: Dr.Saranya.B ,MayilIllam, Puduchatram , Namakkal , Tamilnadu - 637018 , India. E- mail:[email protected] No. 9965655499 1 2 3 Assistant Professor, Assistant Professor, Professor and HOD, Department of pathology, Vinayaka Missions Kirupanandavariyar Medical College and Hospitals, Vinayaka Missions Research Foundation - Deemed to be University, Salem , Tamilnadu – 636308. National Journal of Basic Medical Sciences | Volume 8 | Issue 4 | 2018 http://dx.doi.org/10.31975/NJBMS.2018.8406
6
Embed
xrare form of chronic inflammation which · Ovarian cyst showed cyst wall lined by luteinised ... neoplastic and neoplastic conditions. Non neoplastic conditions include tuberculous
This document is posted to help you gain knowledge. Please leave a comment to let me know what you think about it! Share it to your friends and learn new things together.
Transcript
A Very Rare Case of XanthogranulomatousSalphingitis in One Tube and
Pseudoxanthomatous Salphingitis in Contralateral Tube in a Patient
1 2 3Assistant Professor, Assistant Professor, Professor and HOD,Department of pathology, Vinayaka Missions Kirupanandavariyar Medical College and Hospitals,Vinayaka Missions Research Foundation - Deemed to be University, Salem , Tamilnadu – 636308.
National Journal of Basic Medical Sciences | Volume 8 | Issue 4 | 2018
http://dx.doi.org/10.31975/NJBMS.2018.8406
210
CASE REPORT :
A 26 year old female patient P L , both LSCS 2
presented to the Gynaecology department of
Vinayaka missions Kirupananda Variyar Medical
College and Hospitals with complaints of
abdominal pain on and off and white discharge per
vaginum. She had regular menstrual cycles with
severe dysmenorrhoea during cycles. There were
no history tuberculosis & contraceptive usage. On
examination, she had pallor. Per abdominal
examination was normal. Per speculum and per
vaginal examination revealed healthy cervix and
vagina except for white discharge. Ultra Sonogram
abdomen showed normal uterus and a cyst was
observed in the right ovary measuring 4 X 2 cms
with normal left ovary. Per operatively, extensive
adhesions were noted and hence adhesiolysis was
done. Since, both fallopian tubes were dilated and
adhered to the adjacent tissue, they were removed
along with the ovarian cyst. Ovarian cyst and both
fallopian tubes were sent for histopathological
examination.
PATHOLOGY:
Received both fallopian tubes in the same container
larger tube measuring 3 X 1.5 X 0.5 cms and
smaller tube measuring 2 X 2 X 0.5cms. Both tubes
were congested. C/S of larger tube showed dilated
and thinned out wall. C/S of the smaller tube
showed thickened wall filled with brownish
haemorrhagic material. Right ovarian cyst
measured 4.5 X 2.5 X 0.5 cms with irregular
congested external surface. C/S of the cyst was
irregular with trabeculations. Cyst wall showed no
bladder, testis, epididymis, vagina, endometrium, (10)ovary, and fallopian tube .
A review of the literature revealed fourteen cases of (2, 10 –15)
fallopian tube involvement reported to date .
Patients with XGS ranged from 23 to 72 years old.
Initial presentation most often included lower
abdominal or suprapubic pain, fever, menorrhagia
and vaginal bleeding. Physical examination often (2,
showed a pelvic mass with adnexal tenderness14–18).
The exact pathogenesis of xanthogranulomatous
inflammation remains debated. Many unrelated
disorders may have the same mechanism of foam
cell production. Proposed causes are infection,
National Journal of Basic Medical Sciences | Volume 8 | Issue 4 | 2018
Saranya.B, et.al : Xanthogranulomatoussalphingitis and pseudoxanthomatoussalphingitis in a patient
211National Journal of Basic Medical Sciences | Volume 8 | Issue 4 | 2018
A. Gross picture of fallopain tubes. Pointed area shows thickened wall B. Gross picture of the ovarian cyst. Cyst wall shows trabeculations with no solid areas C. Corpus luteal cyst. Right lower area shows haemorrhage and left upper area shows luteinised Granulosa and theca cells(40 X) D. Thinned fallopian tube shows tubal plicae distended by xanthomatous cells (10 X) E. High power view of the xanthomatous cells (40 X) F. Thickened fallopian tube shows fused tubal plicae forming follicle like structures (Salphingtis follicularis) with inflammatory cells and pigmented histiocytes in the lamina propria (10 X) G.High power view of pigmented histiocytes (40 X)
Saranya.B, et.al : Xanthogranulomatoussalphingitis and pseudoxanthomatoussalphingitis in a patient
212National Journal of Basic Medical Sciences | Volume 8 | Issue 4 | 2018
ineffective antibiotic therapy, abnormality in lipid
metabolism, endometriosis, and ineffective
clearance of bacteria by phagocytes.
Predisposing conditions for the development of
XGS include PID, the presence of an Intrauterine
Device, endometriosis, and inborn error in lipid
metabolism by macrophages, unsuccessful (8–11)
antibiotic therapy, and radiotherapy. A whole
spectrum of changes may exist in this lesion and
probably represent a specialised form of tissue
reaction to multiple etiologies. For example,
bleeding and obstruction may predispose to
infection, and tissue necrosis may occur, followed
by the release of cholesterol and other lipids and
phagocytosis by macrophages.
Xanthogranulomatous salphingitis is often
misdiagnosed by pathologists if they don't keep this
entity in mind. This may be due to the rarity of the
c o n d i t i o n . D i f f e r e n t i a l d i a g n o s i s f o r
xanthogranulomatous salphingitis includenon
neoplastic and neoplastic conditions. Non
neoplastic conditions include tuberculous and
fungal infections which can be ruled out by doing
special stains and culture. For this case,
Ziehlneelsen stain and Periodic Acid Schiff stain
were negative. If the lesion contains mainly focal
scattered lymphocytes, it may be misdiagnosed as
secondary lymphoma or leukemia. If the
lymphocytes are scattered diffusely and foam cells
are seldom, adiagnosis 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 (6,20)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), κ and λ (both positive in (19)polyclonal B lymphocytes) .
( 2 0 )Walther presumed malakoplakia and
xanthogranulomatous inflammation were identical
chronic inflammatory disease. In malakoplakia, the
deposition that leads to permanent bridging (22)between mucosal folds .
Radiologically, Salphingitis follicularis may mimic
an adnexal neoplasm with cystic component.
Histopathologic examination of the material should
be done to make a final diagnosis. Another
differential diagnosis of these follicle like
structures is Salphingitis isthmica nodosa , which is
a diverticular disease of the fallopian tubes that
Saranya.B, et.al : Xanthogranulomatoussalphingitis and pseudoxanthomatoussalphingitis in a patient
213National Journal of Basic Medical Sciences | Volume 8 | Issue 4 | 2018
causes infertility. The etiology of this condition is
unknown. The possible explanations are post
inflammatory distortion and adenomyosis like
process. However in contrast to salphingitis
follicularis, this typically appears as yellow white
nodular swellings located at the isthmic portion of
the tube. Microscopically, they represent the
diverticula like structures that lie within the
hyperplastic smooth muscle with scant (23,24)surrounding inflammation .
Our patient presented with symptoms of severe
dysmenorrhoea and per operatively adhesions were
seen. The patient did not have any history of a lipid
metabolic disease nor her history was suggestive of
immunodefeciency. There was no evidence of
endometriosis histologically, however with the
history of dysmenorrhoea along with adhesions
seen peroperatively between fallopian tube and
adjacent structures, endometriosis can be
considered of as a contributory factor in the
pathogenesis of these conditions presenting as
xanthogranulomatous salphingitis in one tube and
pseudoxanthomatous salphingitis in the other tube.
CONCLUSION:
Xanthogranulomatous sa lphingi t i s and
pseudoxanthomatous salphingitis in one patient is a
very rare entity and we have described the possible
etiologic relationship of these conditions with
endometriosis. It should be borne in mind that both
xan thogranu lomatous sa lph ing i t i s and
pseudoxanthomatous salphingitis may cause
follicle like structures and may be pre operatively
diagnosed as adnexal neoplasm. Proper history,
r ad io log ica l examina t ion and p rec i se
histopathological examination may enlighten the
inflammatory pathology of the lesion and helps in
preventing misdiagnosis because of the rarity of
these conditions.
REFERENCES:
1. Davis.M ,Whitley.ME, and Haque.AK, “Xanthogranulomatous abscess of a mullerian duct remnant. A rare lesion of the rectum and anus,” Diseases of the Colon and Rectum, 1986; vol. 29, no. 11, pp. 755–759.
2. Franco.V, Florena.AM, Guarneri.G, and G a rg a n o . G , “ X a n t h o g r a n u l o m a t o u s salpingitis. Case report and review of the literature,” Acta Europaea Fertilitatis, 1990; vol. 21, no. 4, pp. 197–199.
3. Guarino.M, Reale.D, Micoli .G, and Cristofori.PE , “Xanthogranulomatous g a s t r i t i s : a s s o c i a t i o n w i t h xanthogranulomatouscholecystitis,” Journal of Clinical Pathology,1993; vol. 46, no. 1, pp. 88–90.
4. Cozzu t to .C , “Xan thogranu lomatous osteomyelitis,” Archives of Pathology and Laboratory Medicine, 1984; vol. 108, no. 12, pp. 973–976.
5. Zhang X, Dong H , Zhang L, Desouki MM, Zhao C. Xathogranulomatous Inflammtion of the female genital tract: Report of three cases. Journal of Cancer.2012;3:100-6
6. Date S, SulhyanK, BadgeS, GosaviA, R a m t e e r t h k a r N . Xanthogranulomatousoophoritis : Case report. Indian Journal of Basic and Applied Medical Research 2013;7(2):745-9
7. ShasikalaK, SharmilaPS, SushmaTA, Francis P. Ovarian haemangioma with synchronous inflammation - a rare pathological finding. Int J Health Sci Res.2013;3(5):116-9
8. Herrera.GA, Reimann.BEF, Greenberg.HL, and Miles.PA, “Pigmentosistubae, a new entity: light and electron microscopic study,” Obstetrics and Gynecology, 1983;vol. 61, no. 3, pp. 80S–83S.
9. Seidman.JD, Oberer.S, Bitterman.P, and A i s n e r . S C , “ P a t h o g e n e s i s o f
Saranya.B, et.al : Xanthogranulomatoussalphingitis and pseudoxanthomatoussalphingitis in a patient
214National Journal of Basic Medical Sciences | Volume 8 | Issue 4 | 2018
pseudoxanthomatous salpingiosis,” Modern Pathology, 1993; vol. 6, no. 1, pp. 53–55.
10. G r a y . Y a n d L i b b e y . N P , “Xanthogranulomatous salpingitis and oophoritis,” Archives of Pathology and Laboratory Medicine, 2001; vol. 125, no. 2, pp. 260–263.
11. Punia.RS, Aggarwal.R, and Amanjit.R, “Xanthogranulomatousoophoritis and salpingitis: late sequelae of inadequately treated staphylococcal PID,” Indian Journal of Pathology and Microbiology, 2003; vol. 46, no. 1, pp. 80–81.
12. Furuya.M, Murakami.T, Sato.O et al., “ P s e u d o x a n t h o m a t o u s a n d xanthogranulomatous salpingitis of the fallopian tube: a report of four cases and a literature review,” International Journal of Gynecological Pathology,2002; vol. 21, no. 1, pp. 56–59.
13. Kostopoulou.E, Daponte.A, Kallitsaris. A et al., “Xanthogranulomatous salpingitis: report of three cases and comparison with a case of pseudoxanthomatous salpingitis,” Clinical and Experimental Obstetrics and Gynecology, 2008; vol. 35, no. 4, pp. 291–294.
14. L a d e f o g e d . C a n d L o r e n t z e n . M , “Xanthogranulomatous inflammation of the female genital tract,” Histopathology, 1988; vol. 13, no. 5, pp. 541–551.
15. Daniel.C and Babe's.A, “E' tudesur le x a n t h o m e d e l a t r o m p e u t e ' r i n e (salpingitexanthomateuse),” PresseMédicale, 1923; vol. 31, pp. 1073–1074.
16. Scagl ione.V, “Tubal Xanthomatos is (Strawberry Fallopian Tube),” Minerva Ginecologica, 1964; vol. 16, pp. 352–354.
17. Kunakemakorn.P, Ontai.G, and Balin.H, “Pelvic inflammatory pseudotumor: a case report,” American Journal of Obstetrics and Gynecology, 1976; vol. 126, no. 2, pp. 286–287.
18. Shalev.E, Zuckerman.H, and Rizescu.I, “Pe lv ic in f lammatory pseudotumor ( x a n t h o g r a n u l o m a ) , ” ActaObstetriciaetGynecologicaScandinavica, 1982; vol. 61, no. 3, pp. 285–286.
19. McEntee.GP, Coughlan.M, Corrigan.T, and D e r v a n . P, “ P e l v i c i n f l a m m a t o r y pseudotumour: problems in clinical and histological diagnosis. Case report,” British Journal of Obstetrics and Gynaecology, 1985; vol. 92, no. 10, pp. 1067–1069.
20. G u p t a N , G u p t a C . X a n t h o g r a n u l o m a t o u s o o p h o r i t i s - masquerading as ovarian neoplasm: Report of two cases.Annals of Pathology and Laboratory medicine2015:2(1):C 24-7
21. W a l t h e r M , G l e n n J F , Ve l l i o s F . X a n t h o g r a n u l o m a t o u s c y s t i t i s . J Urol,1985;134:745-6
22. Wheler JE. Disease of the fallopian tubes. In: KurmanRJ,editor. Blausein's Pathology of the Female Genital Tract. 5th ed. Springer:New York,2002;p 624
23. Chawla N, KudesiaS,AzadS,SinghalM,Rai SM. Salphingitisisthmicanodosa. Indian J PatholMicrobiol 2009;52:434-5
24. Creasy JL. Clark RL,CuttinoJT,Groff TR. Salphingitisisthmicanodosa: Radiologic and clinical aspects. Radiology 1985;154:597-600
Saranya.B, et.al : Xanthogranulomatoussalphingitis and pseudoxanthomatoussalphingitis in a patient
Received on : 18.04.2018 Revised on : 02.05.2018 Accepted on: 05.06.2018