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l,fess -\ts/- carcin0nra, malignant melanoma and endothelial cell lined tumors. A diagnosis of carcino-sarcoma arising in SCAP was made, as the tumor cells were infiltrating into the deep dermis, atypical cells were observed along rvith the characteristics of SCAP, with immunopositivig for EMA which shows carci- noma component, and dermal tumor cells showed gradual transition into spindle cell forms arranged in intersecting bundles that was positive for vimentin, suggestive of sarco- matous component. Conclusions SCACP is a rare varieg of shn firmor. The pres- ence 0fsatellite nodule and rapid growth indicates malignant hansformation. Sarcomatous transfor- mation can occur in SCACP Histopatholog along with immunohistochemis$ helps in establishing diagnosis of carcinGsarcoma in SCAP Wde exci- sion is the beatnent of choice. More studies are required for understanding the development of these lesions. References 1. Mammino JJ, Vidmar DA. Syringocy- stadenoma papilliferum. Int J Dermatol 1991:30:763-6. 2. lkt fi DV, Requena ! Kukrer H, et al. /nipf rfr gic aiversity of syringoqntadenocar- ,{.iiima n+illiferum based on a clinic pat}re \rf lo$c shtdy of 6 cases and review of the litera ture. Am J Dermatopathol 2010;32:340-7. 3. lshida-Yamamoto A Sato IL Wada T, et al. Syringocystadenocarcinoma papilliferum: case report and immuno histochemical com- parison with iS benign counterpart J Am Acad Dermatol 2001 ;45:75$9. 4. Dissanalalre RV, Salm R Sweat4land carci- nomas: progrosis related to hishlogical tne. Histopathologi 1980;4 :44F66 . 5. Park SH, Shin YM, Shin DH, et al. Syringo cystadenocarcinoma papilliferum: a case report J Korean Med Sci 2007p2:762-5. 6. Leeborg N, Thompson M, Rossmiller S. Diagnostic pi$alls in syringo cystadenocarci- noma papilliferum: case report and review of the literaturc. Arch Pathol lab Med 20I0J34: 120t9. 7. Requena I. Kirru H, Ackerman AB. Ackerman's histologic diagnosis of neoplastic ' skin alisease: a method by pattein analysis: neoplasms with apocrine differentiation. Philadelphi4 PA Uppincott- Raven; 1998. pp 66S75. 8. Chi CC, Tsai RY, Wang SH. Synngoqsta- denocarcinoma papilliferum: successfully treated with Mohs mioographic surgery. Dermatol Surg 20M;30:46&71. 9. Seco Navedo Md Fresno Forcellerlo M, Ordufla Domingo A" et al. Syringocy- ' stadenoma papilliferum with malignant ew lution: presentation of a case [in []enchl. Ann Dermatol Venereol 1982;109: 68$9. 10. Bonadi R, Urso C. Syringocystadenocar- cinoma papilliferum. Histopathology 1996'p8: 47*7. 11. Arai Y, Kusakabe H, Kiyokane K A case of syringocystadenocarcinoma papilliferum in sifu occuning partially in syringoqrtadeno ma papilliferum. J Dermatol 2003;30:146-50. Figure 1. Ctinical photograph (superior and lateral view). Figure 3. Immunohistochemistry: positive for: A) Pancytokeratin (400X); B) Epithelial membrane antigen; C) Vimentin. Figure 2. Histology: A) Classic sl.ringocystadenoma papilliferum (SCAP) (H&E stain, 100X magnification); B) Classic SCAP (H&E itain, 400X magnification); C) Carcinomatous change in SCAP (H6aE Stain 100X magniffcation); D) Carcinomatous change in SCAP (H&E Stain 400X magniffcation)i E) Sarcomatous change in SCAP (H&E Stain 1O0X magniffcation); F) Sarcomatous change in SCAP (H&E-Stain 400X magniffcation), o.r*&*o.ruu IClinics and Practice 20121 2:e7l) [page 179] I I, ;,Wi.r I

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Page 1: vedantaa.institutevedantaa.institute/wp-content/uploads/2019/06/IMG_0020.pdfCreated Date: 5/29/2019 2:18:04 PM

l,fess-\ts/-

carcin0nra, malignant melanoma and

endothelial cell lined tumors. A diagnosis ofcarcino-sarcoma arising in SCAP was made,

as the tumor cells were infiltrating into the

deep dermis, atypical cells were observed

along rvith the characteristics of SCAP, with

immunopositivig for EMA which shows carci-noma component, and dermal tumor cellsshowed gradual transition into spindle cellforms arranged in intersecting bundles thatwas positive for vimentin, suggestive of sarco-matous component.

Conclusions

SCACP is a rare varieg of shn firmor. The pres-

ence 0fsatellite nodule and rapid growth indicates

malignant hansformation. Sarcomatous transfor-

mation can occur in SCACP Histopatholog along

with immunohistochemis$ helps in establishing

diagnosis of carcinGsarcoma in SCAP Wde exci-

sion is the beatnent of choice. More studies are

required for understanding the development of

these lesions.

References

1. Mammino JJ, Vidmar DA. Syringocy-

stadenoma papilliferum. Int J Dermatol

1991:30:763-6.

2. lkt fi DV, Requena ! Kukrer H, et al.

/nipf rfr gic aiversity of syringoqntadenocar-

,{.iiima n+illiferum based on a clinic pat}re

\rf lo$c shtdy of 6 cases and review of the literature. Am J Dermatopathol 2010;32:340-7.

3. lshida-Yamamoto A Sato IL Wada T, et al.

Syringocystadenocarcinoma papilliferum:

case report and immuno histochemical com-

parison with iS benign counterpart J Am

Acad Dermatol 2001 ;45:75$9.4. Dissanalalre RV, Salm R Sweat4land carci-

nomas: progrosis related to hishlogical tne.Histopathologi 1980;4 :44F66 .

5. Park SH, Shin YM, Shin DH, et al. Syringo

cystadenocarcinoma papilliferum: a case

report J Korean Med Sci 2007p2:762-5.

6. Leeborg N, Thompson M, Rossmiller S.

Diagnostic pi$alls in syringo cystadenocarci-

noma papilliferum: case report and review ofthe literaturc. Arch Pathol lab Med 20I0J34:

120t9.

7. Requena I. Kirru H, Ackerman AB.

Ackerman's histologic diagnosis of neoplastic' skin alisease: a method by pattein analysis:

neoplasms with apocrine differentiation.

Philadelphi4 PA Uppincott- Raven; 1998. pp

66S75.

8. Chi CC, Tsai RY, Wang SH. Synngoqsta-denocarcinoma papilliferum: successfully

treated with Mohs mioographic surgery.

Dermatol Surg 20M;30:46&71.

9. Seco Navedo Md Fresno Forcellerlo M,

Ordufla Domingo A" et al. Syringocy-' stadenoma papilliferum with malignant ewlution: presentation of a case [in []enchl.Ann Dermatol Venereol 1982;109: 68$9.

10. Bonadi R, Urso C. Syringocystadenocar-

cinoma papilliferum. Histopathology 1996'p8:

47*7.

11. Arai Y, Kusakabe H, Kiyokane K A case ofsyringocystadenocarcinoma papilliferum insifu occuning partially in syringoqrtadeno

ma papilliferum. J Dermatol 2003;30:146-50.

Figure 1. Ctinical photograph (superior and lateral view).

Figure 3. Immunohistochemistry: positive for: A) Pancytokeratin (400X); B) Epithelialmembrane antigen; C) Vimentin.

Figure 2. Histology: A) Classic sl.ringocystadenoma papilliferum (SCAP) (H&E stain,100X magnification); B) Classic SCAP (H&E itain, 400X magnification); C)Carcinomatous change in SCAP (H6aE Stain 100X magniffcation); D) Carcinomatouschange in SCAP (H&E Stain 400X magniffcation)i E) Sarcomatous change in SCAP(H&E Stain 1O0X magniffcation); F) Sarcomatous change in SCAP (H&E-Stain 400Xmagniffcation),

o.r*&*o.ruu IClinics and Practice 20121 2:e7l) [page 179]

I

I,

;,Wi.r

I

Page 2: vedantaa.institutevedantaa.institute/wp-content/uploads/2019/06/IMG_0020.pdfCreated Date: 5/29/2019 2:18:04 PM

CafCinO-SafCOma in a CaSe which had rapidly increased in size over trvo

-i

or syringocystadenoma. H',1i;fl,lJi;H'ljJlll;ifXiil,lXfill,,i'; i ;,:1Ti:HH:;, ff:l-l;,,:xll',li;,,#fti.J: I

papilliferum: a rare entity ri.,, in".onrirt.ncy with u gr*urur rr;ru.l. I ;oh;r,il;;;;;;ilj#;:"""'' " "" "" i

Erosions and crustations were seen over its Cell: +9l.9g202lglgg. I

Girish D. Bakhshi,l Kishor R. Wankhede,l surface. She had associated satellite lesion 2 ] E-*ait' [email protected]

I

MukundB.Tayade,lSachinS.Shenonr cminfrontof theprimarylesionmeasuring ln*,,.,^.r.."^-^i-^^^-,--- I

sandeep T. Gore,rArvind G. vatandj 2xi cm with similar morphology (Figure lf i l:J'.ifi::il:"'t0-sarc0ma'svrrng.cvstaden.ma I

Departments of lsurgery and 2pathology, There was no associaled ll,rnphadenopathy. i ' '

I

Grant Hedical College & Sir J J Group of The rest of the examination was within normal i Received for publication: 28 March 2012. I

Hospita,s, ,',umbai, ,ndia l,.Tl'lli,iil,;Hl*,HPl..,i'JilffiXTil i ffiffi [[ffifl;1,,il,",1,Ti; ,,,, I

into adenocarcinoma. She underwent wide I I

locaIexcisionofthetumor,.,c*'l.:I]1lIlH'll*i,*?,Tl,J[1i:Tiiff?uii:|Abstract :#j*::_.i:':.,r,,,,.t;l,ffirdiffi:H:;l l;ilffi:" '*"' I

Slringocvstadenocarcinoma ,."p,'1ir:yT i' :ii,:[l,:ili:1ff;1X[1H1?[l?:",f ffl lf*X::l;:,:;::k!;J;et at' 2012

|arare formof adenocarcinomaof the_1kin. It,is rnorginr *.r. free of tumor cerrr rnol'rls..- | i*^"*irrri,*'iii; 2:e7r Ithe malignant counterpart of syringocystade- tion]lmmunonistochemistry was poritiue io, I doi:l0.408ucp20t2.eit

' I

il jtJS:ti,ilii,-TJ;ilJJi1["':l?:ii6il,.fi .Ht'] jl*m,f

:-1,U,1y.JfiedclinicallyasSCAP'lkelvecases.of sytingo- n.gutiu. for CD3l, CDi4, 5-100, CKi/d, ;; increas-e S :L. or. number, ulceration,

a{pr'***

Correspondence: Girish D. Bakhshi, Demeeti,Plo!61, Sector-7, Koper Khairane, Navi Mumbai-400709, Il'laharashtra, India.

Cell: +91.9820218198.

E-mail: [email protected]

Key words: carcino-sarcoma, syringocystaden0mapapillilerum.

Received for publication:28 March 2012.Revision received: l2 June 2012.

Accepted for publication: 29 June 2012.

This work is licensed under a Creative CommonsAttribution NonCommercial 3.0 License (CC BY-

NC 3.0).

@Copyright G.D. Bakhshi et al., 2012Licensee PAGEPras, ItolyClinics and Proctice 2012;2:e7ldoi: I 0.408l/cp.20I 2.e7I

cystadenocarcinoma papilliferum.h:u. b.:t, ir,isas.Adiagnosisof carcino-sarco;;;- dr$1agi, pruritus, pain, and cosmesis. Thereportedinliterature,withlimitedinformation ing in SCAp was made. No arljuvant tt"r.p,_ltt$fr treatment for syringocystadenomaregarding its clinical and histological charac- was given. patient is disease free ."a ,.riiOyolves complete surgical excision to the sub-teristics. Sarcomatous change in a case of rence-freeafterlyearof followup. 12$,c-utaneous layer to remove all the adenexalSCAP has not been reported in literature till \'? structure. The tissue defect is usually closedrLAr nas not oeen reporreo ln llterature trll \-J structure. 'l'he tissue defect is usually closeddate to the best of our knowledge. We present \ \)- primarily, but a skin graft may sometimes bethe first case of syringocystadenocarcino-sar- u\li - required. On histopathologr, the tumor showscoma papilliferum in a middle-aged female Discussion .^f t deepepidermalcysticinvaginationscontainingwitnasatp,iteresionoverthescarp. --

-clq n{.x::*i{:r$:*:*}::i.*:iSyringocystaden.r"rahrl- papilliferum these c]4stic cavities are connected to the skin

(SCACp) is a rare fdfro-f aaenocareinoma of surface through funnel-shaped structures lined

ttresr.in;th';isthe-mJi;".'t;;,t.il;ibyinfundibularepithelium'TheupperpartofSCAp. lt is saifio diveto-o in , tons_r;ai* invaginations is made up of keratinizing squa-

syringocystadenomapapiriferum (SCAp) is ;tT.Itff*,:;lmn'1ru:llil:H* [*l,H=*r,il|i'.i,T,T..."'ilil1-il#la proliferating skin tumor with apocrine differ- tracl?th-distant metastasis.i However in our epithelium. Cells of the inner layer have colum-entiation. 0ccasionally eccrine differentiation q6iit '** not so. only 12 cases of SCACp nar with oval nuctei and abundant eosinophiliccan be seen. Majority are seen on the face and TFave'been reported in the literature till daie cytoplasm and decapitation on the luminal sur-scalp of young adults, often increasing in siz{after it was first described in l9g0.a However a face, while the cells of the outer layer are small,atoraroundpuberty'rThelesionmlVbgnrgs-.-case of sarcomatous change in a case of cuboidalorflattenedandhavescantycytoplasment at birth or appear in early chililhood-!'ut SCACp has not been reported in literature till and oval nuclei.t occasionally, numerous nor-may also appear at a later agfln ftneral, date. SCAP is usually seen in the middle aged . mal apocrine glands can be found.s However,these tumors have a characteristic ;ducrlike and elderly people with maximum incidencJ in SCACP can be differentiated from SCAP in tha!structure covered by two layers of glandular 4l-?4 years with equal incidence in both the it has an asymmetric structure with ill definedepithelial cells, located in the upper dermis, sexes.s The most common sites are scalp an<t boundaries. The tumor cells often infiltrate thewith a varying degree of papillomatosis. The neck.6 Lesions involving the extremities and deep dermis or subcutaneous faL The neoplas-deeper dermis has tumor cells arranged in perineum have also been described.3s tic cells exhibit crowded nuclei, with variablesheets and cord like pattem along with areas Cfinically, the patient presents with skin col- degree of nuclear atypia and mitotic figures.zrofnecrosis. These tumors comprise ofspindle ored or yellowish papules or nodules of vari- our case also had findings consistent withshaped cells, epithelioid cells, round cells or able rir. (2.5-i3- cm), which remain SCACP and in adrlition had transition to thepolygonal cells.z We report a caseof SCAP hav- unchanged for manyyears but enlarge sudden- spindle cell variant of a sarcoma. On immuno.ing characteristic histology which had turned Iy. There may be blee<ting or ulceiation and histochemistry the tumor is usually positive forinto a carcinoma with a sarcomatous compo- associated new satellite nodules.as The lesion cytokeratin, carcino.embryonic antigen andnent over a short period of time. is usually covered with crusts, which are secre- gMAr,ro,u In our case the immunohistochem-

tions ofapocrine epithelial cells.T The differen- istry was positive for pancgokeratin and EMA

tialdiagnosisincludessquamouscellcarcino.confirmingittobeSCACP.Inadditionthe

;__- :__ - - ma,basalcellcarcinoma,malignantmelanoma tumorwasalsopositiveforvimentinwhichwas

Lase KePort or tumors arising from the endothelial lining consistentwith the sarcomatous change of thelike an angioma or angiosarcoma.3 tumor. other markers like CD31, CD34, 5-100,

A 4S-year-old female presented with cauli- Indications for surgical excision include, CK5/6, HMB45 were negative hence ruling outflower like growth over scalp for one year, malignant change characterized by rapid other differential diagnosis like squamoui cell

[page 178] [ctinics and practice 20r2;2:e7r] oou* ffio..rr

Clinics and Practice 2012; yolume 2:e7l