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Coexistence of sclerosing polycystic adenosis and dysgenetic polycystic disease of parotid, Report of a case

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24.07.2020

Coex stence of scleros ng polycyst c adenos s and dysgenet c polycyst c d sease of parot d, Report of a case :[PAUTHORS], Ind an…

www. jpmonl ne.org/pr ntart cle.asp? ssn=0377-4929;year=2020;volume=63; ssue=1;spage=109;epage=111;aulast=Kahraman

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CASE REPORT

Year : 2020 | Volume : 63 | Issue : 1 | Page : 109--111

Coex stence of scleros ng polycyst c adenos s and dysgenet c polycyst c d sease of parot d, Report of a case

Devr m Kahraman1, Pýnar Yalavaç1, Eylem Akar2, Özgür Özen3, Ömer Günhan1, 1 Department of Pathology, TOBB ETU School of Med c ne, Ankara, Turkey 2 Department of Pathology, Ac badem Hosp tal, İstanbul, Turkey 3 Department of Rad ology, Memor al Ankara Hosp tal, Ankara, Turkey Correspondence Address:

Devr m Kahraman

TOBB ETU Hosp tal Yaşam Caddes No: 5, PK: 06510, Ankara Turkey

Abstract

Scleros ng polycyst c adenos s (SPA) s a rare ben gn sal vary gland les on. Dysgenet c polycyst c d sease (DPD), wh ch s a h stolog cally s m lar les on, may cause a latt ce-l ke gross appearance w th b lateral enlargement of the ent re sal vary glands. In th s report, we present a case of SPA n the r ght parot d and coex stent DPD nvolv ng the both parot d.

How to c te th s art cle:

Kahraman D, Yalavaç P, Akar E, Özen Ö, Günhan Ö. Coex stence of scleros ng polycyst c adenos s and dysgenet c polycyst c d sease of parot d, Report of a case.Ind an J Pathol M crob ol 2020;63:109-111

How to c te th s URL:

Kahraman D, Yalavaç P, Akar E, Özen Ö, Günhan Ö. Coex stence of scleros ng polycyst c adenos s and dysgenet c polycyst c d sease of parot d, Report of a case. Ind an J Pathol M crob ol [ser al onl ne] 2020 [c ted 2020 Jul 24 ];63:109-111

Ava lable from: http://www. jpmonl ne.org/text.asp?2020/63/1/109/277388

Full Text

Introduct on

Scleros ng polycyst c adenos s (SPA) s a rare ben gn sal vary gland les on wh ch s character zed by morpholog cal s m lar ty to f brocyst c changes, scleros ng adenos s, and ntraductal ep thel al prol ferat ons of the breast. SPA occurs over a w de age spectrum and s mostly un focal.[1],[2] The les ons are well c rcumscr bed and are composed of densely sclerot c lobules w th prom nent cyst c, metaplast c, and hyperplast c changes n the ductal and ac nar elements. The d fferent al d agnos s of SPA ncludes a var ety of non-neoplast c and neoplast c sal vary gland les ons also nclud ng dysgenet c polycyst c d sease (DPD).[3] The presence of cyst cally d lated ducts and apocr ne metaplas a are the most frequent shared features.

DPD s a congen tal d sease, frequently seen n females and s character zed by mult ple ep thel al-l ned cyst c spaces ar s ng from ac n and ntercalated ducts.[4],[5] In th s paper, we present a coex stence of SPA of the parot d w th b lateral DPD wh ch were not documented prev ously.

Case Report

A 35-year-old woman appl ed to hosp tal w th a slowly grow ng, pa nless, hard parot d mass, not ced recently w thout other mean ngful cl n cal symptoms. Cl n cal h story revealed that she had been succesfully treated for acute lymphoblast c leukem a w th chemotheraphy and rad otheraphy to the head of poss ble bra n nvolvement. Then total thyro dectomy was performed for thyro d pap llary carc noma and th s was followed by rad oact ve od ne treatment.

Ultrasonograph c and magnet c resonance mag ng [F gure 1] exam nat ons revealed a sol d, hypoecho c les on 2.3 cm n largest d ameter w th n the deep poster or part of the rregular look ng r ght parot d gland. In magnet c resonance mag ng small cyst c changes and calc f ed dens t es w th n both enlarged parot d glands [F gure 1]. Add t onally, small

m crocalc f cat ons were dent f ed n the r ght and left parot d parenchyma. A r ght total parot dectomy was performed.{F gure 1}

Grossly, the parot dectomy spec men measured 6 × 3 × 2.5 cm n s ze. The cut surface showed a f rm, well demarcated nodular les on 2.3 cm n t's largest d ameter w th a rubber cons stency. The sal vary gland around the mass had a sponge l ke parenchyma and latt ce-l ke appearance.

M croscop cally; the sol tary les on was a well encapsulated nodule cons st ng of prol ferat ng ducts n a sclerot c collagenous stroma [F gure 2]. The duct prol ferat ons were composed of scleros ng adenos s-l ke closely packed areas, cr br form structures, sol d slands, cyst c changes and apocr ne metaplas a. It was d agnosed as a representat ve example of SPA. H stolog cally, s m lar to gross apparence the sal vary gland parenchyma outs de the les on demonstrated mult ple lobules of cyst c spaces n vary ng s ze w th a latt ce-l ke appearance. The cyst c changes nvolved the ac n more frequently than the ducts and were l ned by cubo dal or flattened ep thel al cells w th vacuolated cytoplasm rem n scent of lactat onal change of the breast. Cyst c ducts of the DPD commun cates w th the normal lobular structures of the sal vary glands. The lum na of these cysts conta ned pale eos noph l c secret on, globular dens spherules w th concentr c lamellat on, m neral zat on, large s alol ths, and macrophages [F gure 3]. Th s coex stent w despread cyst c change was cons stent w th a d agnos s of DPD. Abdom nal MRI also revealed splen c subcapsular cysts of vary ng s ze.{F gure 2}{F gure 3}

Immunoh stochem cally there was d ffuse CK 7 mmunoreact v ty n the ductal ep thel al cells and smooth muscle act n and p63 pos t v t es n the myoep thel al layer around the ducts wh ch were h ghl ghted the presence of b phas c pattern. Weak estrogen receptor pos t v ty was found w th n the prol ferat ng ducts and K -67 mmunoreact v ty was less than 2%.

D scuss on

SPA was grouped under the nonneoplast c ep thel al les ons of sal vary glands n the 2017 WHO class f cat on.[6] However, the presence of an un nterrupted th ck capsule, a centr fugal growth pattern and a totally d fferent h stolog cal pattern from the surround ng t ssue seen n the present SPA case may support the poss b l ty of neoplas a.[7] Rarely, SPA may be

(2)

24.07.2020

Coex stence of scleros ng polycyst c adenos s and dysgenet c polycyst c d sease of parot d, Report of a case :[PAUTHORS], Ind an…

www. jpmonl ne.org/pr ntart cle.asp? ssn=0377-4929;year=2020;volume=63; ssue=1;spage=109;epage=111;aulast=Kahraman

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assoc ated w th other sal vary gland les ons, though the coex stence of SPA and DPD as n our case has not been reported prev ously. In the present case, morpholog cally, t was not d ff cult to d st ngu sh the local zed SPA from DPD wh ch nvolved ent rely b lateral sal vary glands.

H stolog cal features of DPD may resemble those of polycyst c cond t ons affect ng the other organs. However, there s no report about the assoc at on of DPD nvolv ng other organs except for a s multaneous mult ple, major, and m nor sal vary gland nvolvement.[8]

Except splen c cysts n the present case, there s no reported assoc at on.[9] Immunoh stochem cal sta n ng results n our case showed a dual ep thel al and myoep thel al d fferent at on n SPA and DPA, cons stent w th the prev ous reports.[2] Th s f nd ng s s m lar to the prol ferat ve breast les ons.

The prev ous chemotheraphy and rad otheraph es may be contr but ng factors for the development of SPA n the present case. The coex stence of SPA and DPD n th s report may well suggest that the w despread cyst c changes n the both sal vary gland prov ded a ground for the format on of SPA. SPA cases were usually treated w th exc s on of the sal vary gland and the recurence rates were low.[10] We bel eve that the management of the DPD should be ta lored accord ng to the cl n cal symptoms. The cyst c les ons of the left parot d w ll be followed-up n the present case.

Eth cal approval

Th s art cle does not conta n any stud es w th an mals performed by any of the authors. Informed consent

The samples of the pat ents whose consent forms taken before were evaluated, retrospect vely. Declarat on of pat ent consent

The authors cert fy that they have obta ned all appropr ate pat ent consent forms. In the form the pat ent(s) has/have g ven h s/her/the r consent for h s/her/the r mages and other cl n cal nformat on to be reported n the journal. The pat ents understand that the r names and n t als w ll not be publ shed and due efforts w ll be made to conceal the r dent ty, but anonym ty cannot be guaranteed.

F nanc al support and sponsorsh p N l.

Confl cts of nterest

There are no confl cts of nterest.

References

1 Petersson F. Scleros ng polycyst c adenos s of sal vary glands: A rev ew w th some emphas s on ntraductal ep thel al prol ferat ons. Head Neck Pathol 2013;7(Suppl 1):97-106. 2 Carlos A, Esp nosa CA, Rua L, Torres HE, Valle AF, Fernandes R, et al. Scleros ng polycyst c adenos s of the parot d gland: A systemat c rev ew and report of new cases. J Oral

Max llofac Surg 2017;75:984-93.

3 Skalova A, Gnepp DR, S mpson RH, Lew s JE, Janssen D, S mar R, et al. Clonal nature of scleros ng polycyst c adenos s of sal vary glands demonstrated by us ng the polymorph sm of the human androgen receptor (HUMARA) locus as a marker. Am J Surg Pathol 2006;30:939-44.

4 M halyka EE. Congen tal b lateral polycyst c parot d glands. JAMA 1962;181:634-5.

5 F carra G, Sapp P, Chr stensen RE, Polyakov V. Dysgenet c polycyst c d sease of the parot d gland: Report of a case. J Oral Max llofac Surg 1996;54:1246-9.

6 Seethala R, Gnepp DR, Skalova A, Slater L, W ll ams MD. Non-neoplast c ep thel al les ons: Scleros ng polycyst c adenos s. In: El-Naggar AK, Chan JKC, Grand s JR, Takata T, Slootweg PJ, ed tors. World Health Organ sat on (WHO) Class f cat on of Head and Neck Tumours. Lyon: IARC Press; 2017. p. 195.

7 Mumtaz S, Al A, S ngh M. Scleros ng polycyst c adenos s of oral cav ty. Br J Oral Max llofac Surg 2018;56:753-4.

8 Sr kant N, Yellapurkar S, Boaz K, Bal ga M, Manaktala N, Sharma A, et al. Dysgenet c polycyst c d sease of m nor sal vary gland: A rare case report and rev ew of the l terature. Case Rep Pathol 2017;2017:5279025.

9 Brown E, August M, P lch BZ, Weber A. Polycyst c d sease of the parot d glands. AJNR 1995;16:1128-31.

10 Gnepp DR, Wang LJ, Brandwe n-Gensler M, Slootweg P, G ll M, H lle J. Scleros ng polycyst c adenos s of the sal vary gland: A report of 16 cases. Am J Surg Pathol 2006;30:154-64.

Fr day, July 24, 2020

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