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Breast cancer is the most commonmalignant tumour in women, ac-counting for 30% of newly diag-

nosed cancers.1–3 Breast cancer may recurafter a prolonged disease-free interval inlocal, regional or metastatic form. Metas-tases from breast cancer have been de-scribed to almost every organ; however,bone, lung, liver and brain harbour themajority of distant tumour foci.4 Severalreports cite extrahepatic gastrointestinalmetastases from breast cancer.5–8 Althoughearly diagnosis of the primary malignantlesion is paramount, early detection ofmetastatic disease may also provide somebenefit, as response to hormonal therapyor chemotherapy, or both, has been re-ported in the treatment of disseminatedbreast cancer.5,9 We describe a woman whopresented with linitis plastica of the stom-ach as the first and only indication ofmetastatic breast cancer.

CASE REPORT

A 79-year-old woman presented withearly satiety, progressive dull epigastricpain, and 11.4-kg weight loss over severalmonths. She had undergone a right seg-mental mastectomy with axillary node dis-section 11 years earlier for an invasive lob-ular carcinoma. Pathologically, the marginsof the operative specimen were negative forcancer, and lymph nodes were free of tu-mour. In accordance with National Surgi-

cal Adjuvant Breast Project protocol, thepatient received adjuvant radiotherapy tothe right breast, and she remained diseasefree until the current admission.An extensive work-up of the gastroin-

testinal system was done. An initial gas-troscopy indicated that the gastric wall wasfirm with prominent gastric folds but nomucosal lesion. Multiple random biopsiesof the gastric wall were taken, all of whichwere negative for cancer cells. A subse-quent barium contrast study of the stom-ach demonstrated normal mucosa andnormal transit time. Multiple gastroscopieswith biopsy of gastric mucosa were carriedout because her symptoms persisted.Eventually infiltrating adenocarcinoma,initially thought to be primary in thestomach, was found.Gastric wall thickening was noted on

abdominal CT, but there was no evidenceof intra-abdominal spread. Therefore,elective gastrectomy was scheduled, withthe presumptive diagnosis being primarygastric cancer, linitis plastica type. At la-parotomy, the tumour was found to in-volve the entire stomach and distal esoph-agus; the liver was free of metastases, butan omental implant was detected. Becauseof the extent of the disease, we felt that thepatient would not benefit from an exten-sive resection, so the implant was excisedand a feeding jejunostomy placed. Thepathological features of the omental im-plant raised the suspicion that this tumour

was a metastatic focus of a primary breastcarcinoma. Results of the gastric mucosalbiopsies were consistent with this diagno-sis. In addition, both the omental and gas-tric biopsies were strongly estrogen- andprogesterone-receptor positive, furthersupporting the diagnosis of metastaticbreast cancer.Postoperatively the patient recovered

from the procedure and was dischargedhome on tamoxifen. Unfortunately she didnot respond to the hormonal manipulationand was readmitted shortly thereafter withfeeding intolerance and weight loss. Twocourses of aggressive chemotherapy wereattempted, but she died 14 weeks after theoriginal procedure. The family refused toallow an autopsy.

Pathological findings

The first positive endoscopic biopsy ofthe stomach contained small tumour cells,many of which were signet-ring cells, inthe lamina propria (Fig. 1). They were dis-tributed individually and in loose clusters.The nuclei showed only subtle features ofmalignancy. Intracytoplasmic mucin wasdemonstrated by both periodic acid-Schiffand mucicarmine stains. The histologicpicture was consistent with the clinical di-agnosis of primary gastric cancer, linitisplastica type. The initial negative endo-scopic biopsies were carefully reviewed andcontained no tumour cells.

Brief CommunicationCommunication abrégée

LINITIS PLASTICA AS THE FIRST INDICATIONOF METASTATIC LOBULAR CARCINOMA OF THE BREAST:CASE REPORT AND LITERATURE REVIEW

Lorenzo E. Ferri, MD;* Ron Onerheim, MD;† Carl Emond, MD‡

From the Department of Surgery, Faculty of Medicine, McGill University, Montreal, Que.

*Resident in General Surgery, Surgical Scientist Program, McGill University

†Associate Professor of Pathology and ‡Assistant Professor of Surgery, McGill University and Saint Mary’s Hospital, Montreal, Que.

Accepted for publication Feb. 9, 1999.

Correspondence to: Dr. Lorenzo Ferri, Surgical Research Laboratories, Rm. H6.40, Royal Victoria Hospital, 687 Pine Ave. W, Montreal QC H3A 1A1; fax 514 843-1411

© 1999 Canadian Medical Association

A biopsy of an omental metastasis wasobtained during exploratory laparotomy.A quick- frozen section revealed a metasta-tic deposit of small signet-ring cells in aninflammatory and fibrotic background,consistent with metastatic carcinoma froma gastric linitis plastica. Permanent sections revealed a metasta-

tic deposit of small cells with a prominentsignet-ring cell population. The cellstended to arrange themselves in Indian-file trabeculae and to form target-like le-sions (Fig. 2). At high power, the cellsagain showed only subtle malignant fea-tures, nuclear molding of adjacent cellsand occasional cells with small intracyto-plasmic lumina in addition to the moreprominent signet–ring cells (Fig. 3). Thesearchitectural and cytologic features werereminiscent of a lobular carcinoma of thebreast with prominent signet-ring cells,features well described in the literature.10–12

In addition, the sections of both theomental metastases and the gastric biop-sies were stained immunohistochemicallyfor estrogen and progesterone receptors,and both were strongly positive.Preoperatively, the nature of the pa-

tient’s original breast surgery was notknown. As a result of the histopathologicfindings mentioned above and the possi-bility of breast cancer metastases to thestomach, we recalled the patient’s filesfrom the institution where the mastec-tomy had taken place. The records con-firmed that the patient had undergone asegmental mastectomy for lobular carci-noma of the breast 11 years before. We re-trieved the slides from this specimen andcompared the two tumours. They werehistologically identical. In addition, blocksfrom the original specimen were provided,and further histochemical tests for mucin,and immunohistochemical tests for estro-gen and progesterone receptors showedappearances identical to the omentalmetastases. We concluded that the clinicalpicture was related to lobular carcinomaof the breast metastatic to the stomach andomentum.

DISCUSSION

Metastases to the lung, liver, brain andbone account for the majority of breastcancer dissemination; however, extrahep-atic gastrointestinal metastases have beenwell documented.4 Several authors have

commented on gastric involvement ofmetastatic breast cancer.5–8 From an au-topsy series, Abrams and associates13 re-ported a rate of gastric metastases of 15%.Most researchers, however, have reporteda lower rate.14–16 Gastric metastases frominvasive breast cancer may occur as dis-

crete nodules or as linitis plastica with ex-tensive diffuse infiltration of the submu-cosa and muscularis propria. In the case oflinitis plastica the mode of disseminationis usually hematogenous, first affecting thesubmucosa and muscularis propria. Thismay hinder tissue diagnosis by endoscopic

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CJS, Vol. 42, No. 6, December 1999 467

FIG. 1. Small cancer cells with several signet-ring cells (arrowhead) in the lamina propria of a gastricmucosal biopsy (hematoxylin–eosin, original magnification × 400).

FIG. 2. Metastatic carcinoma in omentum, with targetoid and Indian-file patterns (hematoxylin–eosin,original magnification × 100).

biopsy because superficial biopsies maygive false-negative results.17 Therefore,when linitis plastica is suspected, deeperbiopsies are warranted.Cormier and associates5 reported 6 cases

of linitis plastica of the stomach due tometastatic breast cancer and reviewed anadditional 25 patients from the Mayo Clinicregistry over 20 years. Although the major-ity of these patients presented with synchro-nous or early (within 5 years) gastric metas-tases, there may be an extended disease-freeinterval before symptoms develop. Lessthan 10% of patients in the Mayo Clinic se-ries presented with symptoms related togastric involvement more than 10 years af-ter diagnosis of their primary disease. How-ever, Benfiguig and associates8 described apatient with linitis plastica metastatic frombreast cancer arising 30 years after mastec-tomy. Symptoms encountered most oftenare weight loss, nausea and vomiting, epi-gastric pain and early satiety.5

The radiologic appearance of linitisplastica from breast cancer metastases issimilar to that of primary gastric cancer.Barium swallow usually demonstratesmural rigidity, with thickening of the gas-tric wall.18,19 CT findings of gastric metas-tases of breast cancer usually present as dif-fuse gastric wall thickening. Elliott andcolleagues20 suggested that CT criteria forlinitis plastica type of gastric metastasesfrom breast cancer include a gastric walldiffusely thickened by more than 1 cm inan adequately distended stomach.The histologic subtype of invasive breast

cancer appears to influence the site ofmetastasis.21 All reported cases of linitis plas-tica of gastric metastases from breast cancerhave arisen in patients with documented in-vasive lobular carcinoma. Lobular carci-noma of the breast can contain a high per-centage of signet-ring cells,11 which, whencombined with a diffuse pattern of spread,can render the metastasis to the stomach al-most indistinguishable from primary gastriclinitis plastica.6 Ductal carcinoma has beennoted to metastasize to the stomach, but ina discrete nodular pattern.5

Hormone-receptor status of the pri-mary tumour is of vital importance whenconsidering the therapeutic options for pa-tients with advanced breast cancer. Re-sponse to hormonal manipulation is welldescribed in patients with metastatic estro-gen- and progesterone-positive breast can-cer.9 With regard to gastric metastases of

breast cancer in particular, survival of upto 4 years was reported in patients treatedwith various hormonal or chemotherapeu-tic regimens.5 This is in contrast to the dis-mal prognosis and usually ineffective non-surgical management options facing thepatient with linitis plastica due to primarygastric cancer.Our objective in reporting this case was

to increase the awareness of surgeons togastric metastases from primary breast can-cer. Although not common, this is welldocumented in the literature. The onsetof gastrointestinal symptoms in a patientwith a history of invasive lobular carci-noma should prompt the treating surgeonto entertain the possibility of gastricmetastases and organize the appropriateinvestigations. Gastric metastases and lini-tis plastica may arise much later than theprimary disease, thus a prolonged disease-free period should not discount the possi-bility that the gastrointestinal symptomsmay be due to breast cancer dissemination.We recommend that investigation of earlysatiety, weight loss and epigastric pain inpatients previously treated for invasive lob-ular carcinoma should include gastroscopyand biopsy, with the understanding that anegative biopsy may represent an insuffi-ciently deep sampling of the gastric walland multiple bites of tissue may be re-quired. Although the prognosis is still un-

certain, patients with linitis plastica frombreast cancer metastases have been notedto respond to hormonal therapy orchemotherapy, or both, particularly if themetastases are positive for estrogen orprogesterone receptors. As more of thesepatients are identified and followed up, thetreatment modalities and response may bebetter understood and defined.

References

1. Landis SH, Murray T, Bolden S, WingoPA. Cancer statistics 1998. CA Cancer JClin 1998;48(1):6-29.

2. Breast cancer incidence and mortality —United States 1992. MMWR 1996;45 (39):833-7.

3. Hortobagyi GN. Multidisciplinary manage-ment of advanced primary and metastaticbreast cancer [review]. Cancer 1994;74(1 Suppl): 416-23.

4. Lee YT. Breast carcinoma: pattern of recur-rence and metastases after mastectomy. AmJ Clin Oncol 1984;7(5):443-9.

5. Cormier WJ, Gaffey TA, Welch JS, Edmon-son JH. Linitis plastica caused by metastaticlobular carcinoma of the breast. MayoClinic Proc 1980;55(12):747-53.

FERRI ET AL

468 JCC, Vol. 42, No 6, décembre 1999

FIG. 3. Small cancer cells in omentum with prominent signet-ring cells (arrowhead) in Indian file(hematoxylin–eosin, original magnification × 400).

6. Clavien PA, Laffer U, Torhost J, Harder F.Gastro-intestinal metastases as first clinicalmanifestation of the dissemination of abreast cancer. Eur J Surg Oncol 1990;16 (2):121-6.

7. Walker Q, Bilous M, Tiver KW, LanglandsAO. Breast cancer metastases masqueradingas primary gastric carcinoma. Aust N Z JSurg 1986;56(5):395-8.

8. Benfiguig K, Anciaux M, Eugene C, Benke-moun G, Etienne JC. [Gastric metastases ofbreast cancer occurring after a cancer-freeinterval of 30 years]. Ann Gastroenterol He-patol (Paris) 1992;28(4):175-7.

9. Dao TL, Nemoto T. Steroid receptors andresponse to endocrine ablations in womenwith metastatic cancer of the breast. Cancer1980;46(12 Suppl):2779-82.

10. DiCostanzo D, Rosen PP, Gareen I,Franklin S, Lesser M. Prognosis in infiltrat-ing lobular carcinoma. An analysis of “clas-sical” and variant tumors. Am J Surg Pathol1993;14(1):12-23.

11. Eltorky M, Hall JC, Osborne PT, el ZekyF. Signet-ring cell variant of invasive lobularcarcinoma of the breast. A clinicopathologicstudy of 11 cases. Arch Pathol Lab Med1994;118(3):245-8.

12. Martinez V, Azzopardi JG. Invasive lobularcarcinoma of the breast: incidence and vari-ants. Histopathology 1979;3(6):467-88.

13. Abrams H, Spiro R, Goldstein N. Metas-tases in carcinoma: analysis of 1000 autop-sied cases. Cancer 1950;3:74-85.

14. Asch MJ, Wiedel PD, Habif DV. Gastroin-testinal metastases from carcinoma of thebreast. Autopsy study and 18 cases requir-ing operative intervention. Arch Surg 1968;96 (5):840-3.

15. Choi SH, Sheehan FR, Pickren JW.Metastatic involvement of the stomach bybreast cancer. Cancer 1964;17:791-9.

16. Caskey CI, Scatarige JC, Fishman EK. Dis-tribution of metastases in breast carcinoma:CT evaluation of the abdomen. Clin Imag-ing 1991;15(3):166-71.

17. Taal BG, den Hartag Jager FC, SteinmetzR, Peterse H. The spectrum of gastroin-testinal metastases of breast carcinoma: I.Stomach. Gastrointest Endosc 1992;38 (2):130-5.

18. Joffe N. Metastatic involvement of thestomach secondary to breast carcinoma. AmJ Roentgenol Radium Ther Nucl Med 1975;123(3):512-21.

19. Meyers MA, McSweeney J. Secondary neo-plasms of the bowel. Radiology 1972;105(1):1-11.

20. Elliott LA, Hall GD, Perren TJ, Spencer JA.Metastatic breast carcinoma involving thegastric antrum and duodenum: computedtomography appearances. Br J Radiol 1995;68(813):970-2.

21. Harris M, Howell A, Chrissohou M,Swindell RI, Hudson M, Sellwood RA. Acomparison of the metastatic pattern of in-filtrating lobular carcinoma an infiltratingduct carcinoma of the breast. Br J Cancer1984;50(1):23-30.

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