CASE REPORT OPEN ACCESSInternational Journal of Surgery Case Reports 5 (2014) 290293

Contents lists available at ScienceDirect

International Journal of Surgery Case Reports

j ourna l h om epage: www.caserepor ts .com

Phyllodes tumor metastasis to the tonsil with synundifferentiated carcinoma

Rui Sano shiMichihira Department ob Department o , Japanc Department od Department o

a r t i c l

Article history:Received 27 JaReceived in reAccepted 16 MAvailable onlin

Keywords:Anaplastic carMultiple primNeoplasm metastasisPhyllodes tumorTonsillar neoplasm

sis told woastasnsillaent rbecam

ledgesizing to the tonsil. Furthermore, morphological and immunohistochemical study revealed that the righttonsillar tumor was irrelevant to the phyllodes tumors.CONCLUSION: We report a case of phyllodes tumor metastasis to the left tonsil which developed undif-ferentiated carcinoma in the other side of tonsil later. Breast tumor metastasis to the tonsil is rare but itshould be considered as a possible diagnosis.

2014 The Authors. Published by Elsevier Ltd. on behalf of Surgical Associates Ltd. This is an open

1. Introdu

Metastafor only 0involved mgastric adeneoplasms.inate from androblastopancreatic gall bladdermary tumohave been rmetastasis

Tonsillarnosis of themetastasis

CorresponHealth and W8530, Japan. T

E-mail add

http://dx.doi.o2210-2612/ (http://creativaccess article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/3.0/).

ction

sis to the tonsil is extremely rare, accounting.8% of all tonsillar tumors.1 The cases reportedetastasis from cutaneous or mucosal melanoma,2,3

nocarcinoma,4,5 hypernephroma,6,7 and various lung8 The less common metastatic tonsillar tumors orig-mesothelioma,9 hepatocellular carcinoma,10 ovarianma,11 seminoma,12 anaplastic thyroid carcinoma,13

adenocarcinoma,14 prostatic adenocarcinoma,15 and carcinoma.16 Breast carcinoma is one of the rarest pri-rs that can metastasize to the tonsil; only eight caseseported in the literature,17 and only one case of tonsillarof breast sarcoma has been reported.18

metastasis rarely becomes apparent before the diag- primary neoplasm, and only a few cases of tonsillaras a presenting feature have been reported in the

ding author at: Department of Otorhinolaryngology, Japan Laborelfare, Chubu Rosai Hospital, 1-10-6 Komei, Minato-ku, Nagoya 455-el.: +81 52 652 5511; fax: +81 52 653 3533.ress: ruiruisano@gmail.com (R. Sano).

literature.19 In most of these cases, the presenting symptoms weresore throat, an oral globus sensation, and different degrees ofdysphagia and odynophagia. In other cases, patients were asymp-tomatic, and their tonsillar neoplasm was detected incidentallyduring a routine oral examination. Tonsillar metastasis can be bilat-eral or unilateral depending on the nature of the primary neoplasm.The prognosis for patients with tonsillar metastasis is rather poor.6

In this report, we present a case of a phyllodes tumor that metas-tasized to the tonsil; we have included the clinical features andpathological ndings.

2. Presentation of case

A 54-year-old woman underwent right mastectomy for a breasttumor 3 years before she was referred to our department. The tumorwas diagnosed pathologically as a malignant phyllodes tumor.There was no evidence of axillary metastasis. Histopathologicalexamination showed a tumor larger than 10 cm in diameter thathad slightly invaded the surrounding tissues and was character-ized by moderate nuclei pleomorphism, moderate cell density, andmoderate mitosis (Fig. 1A and B). The nal pathology revealed thatno tumor was identied on the margin of the resected specimen.

rg/10.1016/j.ijscr.2014.03.0142014 The Authors. Published by Elsevier Ltd. on behalf of Surgical Associates Ltd. This is an open access article under the CC BY-NC-ND licenseecommons.org/licenses/by-nc-nd/3.0/).a,b,, Eisuke Satob, Tetsuya Watanabec, Hisakazu Oo Masakib, Tsutomu Nakashimaa

f Otorhinolaryngology, Graduate School of Medicine, Nagoya University, Nagoya, Japanf Otorhinolaryngology, Japan Labor Health and Welfare, Chubu Rosai Hospital, Nagoyaf Surgery, Chita City Hospital, Chita, Japanf Otorhinolaryngology, Chita City Hospital, Chita, Japan

e i n f o

nuary 2014vised form 5 March 2014arch 2014e 25 March 2014

cinomaary

a b s t r a c t

INTRODUCTION: Breast tumor metastaPRESENTATION OF CASE: A 54-year-otumor and later presented with metlobectomy and resection of the left tonoma of the right tonsil. She underwstarted. The central venous catheter Finally, she died.DISCUSSION: To the best of our knowchronous

mad, Atsushi Andob,

the tonsil is extremely rare.man underwent resection of a breast malignant phyllodesis to the lung and the left tonsil. She underwent left lowerr tumor. She subsequently developed undifferentiated carci-esection of the right tonsillar tumor and chemotherapy wase infected with methicillin-resistant Staphylococcus aureus.

, this is the rst reported case of a phyllodes tumor metasta-

CASE REPORT OPEN ACCESSR. Sano et al. / International Journal of Surgery Case Reports 5 (2014) 290293 291

Fig. 1. (A) The breast surgical specimen shows spindle cell proliferation with evident stromal overgrowth and numerous bizarre cells (HE stain, 40). (B) Higher magnicationshows malignant spindle cell proliferation arranged in long intersecting fascicles. The cells show evident pleomorphism, hyperchromasia, and mitotic activity (HE stain, 200).

Free margin was 900 m. Immunostaining showed that the tissuewas negative for cytokeratin AE1/AE3, MNF116, and CAM5.2.

Although she was informed of possible recurrence, she stoppedambulatory care 4 months after the operation. Two years and 10months aftePhysical exathe lung tumwas diagnoquently undPathology athe malignayears and 1 to our depaswelling. Phsuring 20 ma left tonsigeal constriPathology aof the malirence was ithe left tonsillar tumornuclei (Fig. negative fothe patholoto those of diagnosed aoperative clater.

Three months after the left tonsil resection, she presented withright tonsil swelling. Physical examination revealed a right tonsillartumor. Biopsy of the right tonsillar tumor showed undifferentiatedcarcinoma of the oropharynx. One week after the biopsy, she under-

esectrightd atyy bree tiss2. In steinonsilphagnosrecurhad bcludi

caths aud. Fin

cussi

llodets fond moma

mal

Fig. 2. (A) Sec(HE stain, 40r the operation, she presented with cough and dyspnea.mination revealed a tumor in the left lung. A biopsy ofor was performed with bronchoscopy, and the sample

sed as metastasis of the phyllodes tumor. She subse-erwent left lower lobectomy for the lung metastasis.nalysis of the lung specimen indicated metastasis ofnt phyllodes tumor with incomplete resection. Threemonth after the lung operation, the patient was referredrtment for throat pain and presented with left tonsilysical examination revealed a left tonsillar tumor mea-m in diameter. Computed tomography images showedllar tumor that did not spread outside the pharyn-ctor muscles. No neck lymphadenopathy was present.nalysis of the biopsy of the left tonsil showed metastasisgnant phyllodes tumor of the breast. No tumor recur-dentied in the left lung. She underwent resection ofsillar tumor. Histopathological analysis of the left ton-

showed growth of spindle cells, necrosis, and atypical2A and B). Immunostaining showed that the tissue wasr cytokeratin AE1/AE3, MNF116, and CAM5.2. Becausegical features of the left tonsillar tumor were similarthe primary breast tumor, the left tonsillar tumor wass a metastasis of the primary phyllodes tumor. The post-ourse was favorable, and she was discharged 1 week

went rof the sis, anprimarthat thCAM5.for Epright tthe orothe dia

No lesion apy, invenouslococcustoppeapy.

3. Dis

Phyaccounrecur aand strline, ortion from the left tonsillar tumor covered by stratied squamous epithelium shows remn). (B) Higher magnication shows brisk mitotic activity and multinucleated tumor giant cion of the right tonsillar tumor. Histopathology analysis tonsillar tumor showed growth of spindle cells, necro-pical nuclei, which differed from the ndings for theast phyllodes tumor (Fig. 3). Immunostaining showedue was positive for cytokeratin AE1/AE3, MNF116, andsitu hybridization showed that the tissue was negativeBarr virus. The pathological features of the resected

were similar to those of undifferentiated carcinoma ofrynx. Three histopathologists consulted and agreed onis.rence of the tonsillar tumors or deterioration of the lungeen observed, but the breast tumor regrew. Chemother-ng doxorubicin and ifosfamide, was started. The centraleter became infected with methicillin-resistant Staphy-reus during chemotherapy, and the chemotherapy wasally, she died 1 month after the cessation of chemother-

on

s tumor is an uncommon breast neoplasm thatr less than 1.0% of all breast tumors and has the ability toetastasize.20 Phyllodes tumor comprises both epitheliall elements. Phyllodes tumor is graded as benign, border-ignant according to a set of histological data includingants of lymphoid tissue and wide areas of hemorrhage and necrosisells (HE stain, 200).

CASE REPORT OPEN ACCESS292 R. Sano et al. / International Journal of Surgery Case Reports 5 (2014) 290293

Fig. 3. The hisspindle cells, nbreast tumor:

stromal celovergrowthtrative andthe literaturThe overallmost comm

The treaalthough mtectomy is noptimal trePhyllodes tuand radiatio

This casknowledge,metastasizireports of pregion, inclmandibularmandible.27

tasis of theadenoma.28

to the tonsiSecond,

right tonsila metastatithat the riglodes tumoas undifferediffered froplasms occone paper.3

patients ascer, laryngcolon cancecinoma. Budiagnosis ofsial. Anothetumor (i.e.elements). metastasis

Breast tuconsidered

4. Conclusion

report a case of phyllodes tumor metastasis to the left tonsil developed undifferentiated carcinoma in the other side ofater.sider

t of

e de

g

e.

l app

s was. Wrlicatritt

journ

nces

ford 979;1amurnasopogan Lil. Laro A, Pocarcto I, Ae stomnson

tine tosain SS8;67(on DJ. J R Sor T, Dil. Otoes F, wing ;67(7kov Vil. J Sukov Bringolar T, Mtopathological ndings of the right tonsillar tumor show growth ofecrosis, and atypical nuclei. These differed from the ndings for theHE stain 200.

lularity, stromal cell atypia, mitotic activity, stromal, necrosis, and whether the margin is rounded or inl-

shows malignant heterologous elements. A review ofe suggests that malignancy occurs in 1450% of cases.21

incidence of metastasis of phyllodes tumor is 312%,only to the lung.22

tment of choice is excision with a 10-mm margin,alignant lesions require mastectomy. However, mas-ot effective in preventing metastatic disease,22 and theatment of metastatic disease has not been established.mors are thought to be resistant to both chemotherapyn.23

e is extremely rare from two perspectives. First, to our this is the rst reported case of a phyllodes tumorng to the tonsil. A review of the literature found severalhyllodes tumors metastasizing to the head and neckuding the right ramus,24 posterior right maxilla,25 left

posterior quadrant,26 and left angle and ramus of theOther cases involved gingival metastasis and metas-

temporomandibular region, parotid grand, thyroid31 There is no report of phyllodes tumor metastasizingl.

Wewhichtonsil lbe con

Conic

Non

Fundin

Non

Ethica

Thireviewfor pubof the wof this

Refere

1. CrawAm 1

2. Ramthe

3. Aydtons

4. Galladen

5. Beniof th

6. Browpala

7. HusJ 198

8. Seddlung

9. Hefetons

10. Llanfollo1996

11. Bychtons

12. Sinianola

13. Hadthis is the rst reported case of secondary primarylar tumor which was developed in the patient withc left tonsillar tumor. The clinical course suggestedht tonsillar tumor should be a metastasis of a phyl-r. However, the right tonsillar tumor was diagnosedntiated carcinoma because the histopathology clearlym those of the left tonsillar tumor. Secondary neo-urring in cases of phyllodes tumor were reported in2 In that report, eight tumors were diagnosed in 6/32

secondary tumors of duodenal cancer, cervical can-eal cancer, alveolar cell adenocarcinoma, metastaticr, astrocytoma, esophageal cancer, and lung adenocar-t these cases were not simultaneously occurred. The

the right tonsillar tumor in this case remains controver-r hypothesis is anaplastic transformation of phyllodes, stromal elements may have changed to epithelialFurther investigation is necessary to understand theprocess in cases of phyllodes tumor.mor metastasis to the tonsil is rare but it should beas a possible diagnosis.

tonsil. J La14. Feleppa AE

Nose Throa15. Millar EKA

tonsil a c16. Asami K,

palatine to17. Tueche SG

breast can18. Bar R, Netz

hemangioThroat J 20

19. Hurlstoneof gastric

20. McDivitt RJ 1967;120

21. Moffat CJCcopatholo

22. Kessinger case repor

23. Chen WHof the bre18594.

24. Cooney BMof the brethe literat599604. Breast tumor metastasis to the tonsil is rare but it shoulded as a possible diagnosis.

interest statement

clared.

roval

an institutional review board-approved retrospectiveitten informed consent was obtained from the patiention of this case report and accompanying images. A copyen consent is available for review by the Editor-in-Chiefal on request.

BE, Callihan MD, Corio RL, et al. Oral pathology. Otolaryngol Clin North2(1):2943.

thy L, Nassar WY, Hasleton PS. Metastatic melanoma of the tonsil andharynx. J Laryngol Otol 1995;109(3):2367.B, Myers JN, Myers EN, et al. Malignant melanoma metastatic to theyngoscope 1996;106(3):3136.escarmona E, Crupi J, et al. Bilateral tonsillar metastasis of gastricinoma. Head Neck 1992;14(1):557.lvarez-Gago T, Morais D. Tonsillar metastasis from adenocarcinomaach. J Laryngol Otol 1996;110(3):2913.

RJ, Lamonte SE, Jaques WE, et al. Hyper-nephroma metastatic to thensils. Ann Otol Rhinol Laryngol 1979;88(2):23540., Dalal VC. Tonsillar metastases from hypernephroma. Ear Nose Throat2), 1178, 121.. Tonsillar metastasis at presentation of small cell-carcinoma of thec Med 1989;82(11):688.

anino J, Joachims HZ, et al. Metastatic malignant mesothelioma to thelaryngol Head Neck Surg 1997;116(6):6848.SanzOrtega J, Suarez B, et al. Hepatocellular carcinomas diagnosedmetastasis to the oral cavity. Report of 2 cases. J Periodontol):7179., Ghosh L, Lundine M, et al. Ovarian androblastoma metastatic torg Oncol 1984;27(4):2759.S. A case of metastasis of seminoma to the palatine tonsil. Vestn Otori-

1963;25:99101.or C, Harel G, et al. Anaplastic thyroid-carcinoma metastatic to the

ryngol Otol 1987;101(9):9536., Ellison NM. Metastatic pancreatic adenocarcinoma of the tonsil. Eart J 1981;60(3):1368., Jones RV, Lang S. Prostatic adenocarcinoma metastatic to the palatinease-report. J Laryngol Otol 1994;108(2):17880.Yokoi H, Hattori T, et al. Metastatic gall-bladder carcinoma of thensil. J Laryngol Otol 1989;103(2):2113., Nguyen H, Larsimont D, et al. Late onset of tonsillar metastasis fromcer. Eur J Surg Oncol 1999;25(4):43940.er A, Ostrovsky D, et al. Abrupt tonsillar hemorrhage from a metastaticsarcoma of the breast: case report and literature review. Ear Nose11;90(3):11620.

DP, Sanders DS, Smith A, et al. Tonsillar metastasis: a rare presentationcarcinoma. Eur J Surg Oncol 2001;27(3):32830.W, Urban JA, Farrow JH. Cystosarcoma phyllodes. Johns Hopkins Med(1):3345., Pinder SE, Dixon AR, et al. Phyllodes tumors of the breast a clini-gical review of 32 cases. Histopathology 1995;27(3):20518.A, Foley JF, Lemon HM, et al. Metastatic cystosarcoma phyllodes: at and review of the literature. J Surg Oncol 1972;4(2):13147., Cheng SP, Tzen CY, et al. Surgical treatment of phyllodes tumorsast: retrospective review of 172 cases. J Surg Oncol 2005;91(3):

, Ruth GJ, Behrman DA, et al. Malignant cystosarcoma phyllodesast metastatic to the oral cavity report of a case and review ofure. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1988;66(5):

CASE REPORT OPEN ACCESSR. Sano et al. / International Journal of Surgery Case Reports 5 (2014) 290293 293

25. Tenzer JA, Rypins RD, Jakowatz JG. Malignant cystosarcoma phyl-lodes metastatic to the maxilla. J Oral Maxillofac Surg 1988;46(1):802.

26. Yoshimura Y, Inoue Y, Mihara Y, et al. Metastatic malignant cystosarcoma-phyllodes report of a case presenting with an oral tumor and review of theliterature. J Craniomaxillofac Surg 1991;19(5):22731.

27. Abemayor E, Nast CC, Kessler DJ. Cystosarcoma phyllodes metastatic to themandible. J Surg Oncol 1988;39(4):23540.

28. Masmoudi A, Ayadi L, Bouassida S, et al. Gingival metastasis in breast phyllodesarcoma. Ann Dermatol Venereol 2006;133(5):44951 (in French with Englishabstract).

29. Deeming G, Divakaran R, Butterworth D, et al. Temporomandibular regionmetastasis from cystosarcoma phyllodes: a case report and review of the lit-erature. J Craniomaxillofac Surg 2003;31(5):3258.

30. Zhang JZ, Gu M. Malignant phyllodes tumor of the breast metastatic to theparotid gland diagnosed by ne needle aspiration biopsy a case report. ActaOtolaryngol 2003;47(2):2538.

31. Giorgadze T, Ward RM, Baloch ZW, et al. Phyllodes tumor metastatic to thyroidHurthle cell adenoma an unusual tumor-to-tumor metastasis. Arch Pathol LabMed 2002;126(10):12336.

32. Geisler DP, Boyle MJ, Malnar KF, et al. Phyllodes tumors of the breast: a reviewof 32 cases. Am Surg 2000;66(4):3606.

Open AccesThis article undepermits un any mcredited.s is published Open Access at sciencedirect.com. It is distributed restricted non commercial use, distribution, and reproduction in r the IJSCR Supplemental terms and conditions, whichedium, provided the original authors and source are

Phyllodes tumor metastasis to the tonsil with synchronous undifferentiated carcinoma1 Introduction2 Presentation of case3 Discussion4 ConclusionConflict of interest statementFundingEthical approvalReferences