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Hindawi Case Reports in Oncological Medicine Volume 2018, Article ID 4714708, 4 pages https://doi.org/10.1155/2018/4714708 Case Report Gastric Linitis Plastica and Peritoneal Carcinomatosis as First Manifestations of Occult Breast Carcinoma: A Case Report and Literature Review 1,2 1 1,2 3 Mara Mantiero , Giovanni Faggioni, Alice Menichetti, Matteo Fassan, 1,2 1,2 Valentina Guarneri, and Pierfranco Conte Medical Oncology Unit 2, Istituto Oncologico Veneto, IRCCS, Padova, Italy Department of Surgical, Oncological and Gastroenterological Sciences, University of Padova, Padova, Italy Department of Medicine, Surgical Pathology & Cytopathology Unit, University of Padova, Padova, Italy Correspondence should be addressed to Mara Mantiero; firstname.lastname@example.org Received 25 February 2018; Revised 14 May 2018; Accepted 11 June 2018; Published 8 July 2018 Academic Editor: Constantine Gennatas Copyright © 2018 Mara Mantiero et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Gastric linitis plastica is a diﬀuse involvement of the stomach walls by neoplastic cells. It represents about 3–19% of primitive gastric adenocarcinomas, but it can also be the manifestation of a metastatic disease. Breast cancer is the most frequent malignancy in women, and the metastatic spread to the stomach occurs in less than 10% of the cases. We present an unusual case of gastric linitis plastica and peritoneal carcinomatosis as manifestations of an occult breast cancer in a 53-year-old woman. Imaging and endoscopic evaluation were not able to discriminate a primary from a secondary gastric lesion. The histological evaluation excluded the diagnosis of a primary gastric neoplasia. The IHC proﬁle was consistent with the diagnosis of metastases from the breast cancer. Due to the hormonal receptors’ positivity, we started therapy with fulvestrant (500 mg, day 0, 14, and 28 and every 28 days thereafter by intramuscular injection). After 20 months, the same therapy is still ongoing and well tolerated, while the patient is in good condition with improvement of the dysphagia. Almost 2 years after the diagnosis of linitis plastica, the primitive breast lesion is still occult. 1. Introduction origin of the lesion was crucial to avoid a potentially useless gastric surgery. Metastatic cancer of unknown primary (CUP site syndrome) is characterized by the presence of the metastatic lesion 2. Case Presentation without the primitive carcinoma. It accounts for 3–5% of all solid malignant tumours, and the prognosis is generally In March 2016, a 53-year-old premenopausal woman was poor . Only microscopic analysis, with histological and admitted to our institute with the diagnosis of gastric linitis immunohistochemical exam, can deﬁne the primary origin plastica and peritoneal carcinomatosis. She presented with of the lesion, and it is fundamental for the clinician to deﬁne upper abdominal pain, dyspepsia, nausea, and daily post- the correct treatment plan. The discussion with the patholo- prandial vomiting with weight loss of approximately 4 kilo- gist is essential. grams in 2 months. The Eastern Cooperative Oncology Metastasis from breast cancer to the gastrointestinal tract Group (ECOG) performance status (PS) was 2. Her medical is rare, less than 10% , and typically occurs many years history was negative for oncologic diseases, and she had no after the diagnosis. relevant comorbidities; no history of Helicobacter pylori- We present an unusual case of gastric linitis plastica and associated gastritis. At clinical examination, she presented peritoneal carcinomatosis as ﬁrst manifestations of an occult with epigastric tenderness and no mass. Blood tests were breast cancer. The correct identiﬁcation of the primary within the normal values, with the exception of CA15.3 2 Case Reports in Oncological Medicine (211 U/ml) and CEA (11.1 ng/ml). Abdominal computed tomography (CT) revealed an increased wall thickness of the pyloric antrum along with mesenteric lymphadenopathy (20 mm) and peritoneal carcinomatosis. No liver metastases were detected. At esophagogastroduodenoscopy (EGDS), a severe pyloric stenosis was reported in the absence of muco- sal lesions. The clinical manifestation was strongly suggestive of linitis plastica. Several gastric biopsies were performed, and histology concluded for a diﬀuse localization of epithelial cancer. Immunohistochemistry excluded gastrointestinal origin. There was a strong immunoreactivity for estrogen and progesterone receptors (ER-PgR: 80%-80%), GATA3 Figure 1: Histology conﬁrmed localization of adenocarcinoma with (3+), and cytokeratin (CK) 7, 8, 18, and 19; the human epi- immunohistochemistry: ER 90%, PgR 35%, CK7 3+, GCDFP-15 3+, and HER2 1+. thelial growth factor receptor 2 (HER2) was negative (1+) and the Ki67 index was <5%. Histological exam concluded for metastatic breast cancer with gastric linitis plastica. primitive lesion prevented any possibility of the histological A complete breast radiological investigation including subdeﬁnition, although the lobular histological subtype is bilateral ultrasound and mammography, and magnetic reso- the most common cause of metastatic gastric linitis plastica caused by breast cancer . nance imaging excluded the presence of breast abnormalities. Multiple bilateral suspicious axillary lymph nodes (maxi- mum diameter of approximately 10 mm) were identiﬁed at ultrasonography and MRI. A ﬁne-needle aspiration of a right 3. Discussion axillary lymph node was performed, and cytology was posi- tive for epithelial malignant cells. Breast cancer is the most common malignancy in women, To deﬁnitively exclude a gastrointestinal origin of the accounting for about 30% of new diagnosis. Approximately neoplasm, the patient also underwent laparoscopic perito- 6–10% of new breast cancer cases are initially metastatic, neal biopsy. Histological and immunohistochemical studies and the most common sites of metastatization are the liver, conﬁrmed breast origin. After the multidisciplinary discus- lung, brain, and bone . Metastases from breast cancer to sion, a surgical approach was excluded. A Witzel feeding jeju- the gastrointestinal tract are rare. Harris et al. published in nostomy was created. 1984 the data about an autopsy series of 109 patients who All international breast cancer guidelines recommend died from breast cancer: 84% of them were metastatic and endocrine therapy in luminal metastatic breast cancer with- only 8.8% had gastric involvement . out visceral crisis. Our patient, after jejunostomy creation Typically, metastatic spread to the gastrointestinal tract and starting of enteral nutrition, was asymptomatic, and so, occurs many years after the diagnosis of breast cancer. In in April 2016, hormone therapy with fulvestrant was started our case, it was at the onset of the disease. Gastric metastati- (500 mg, day 0, 14, and 28 and every 28 days thereafter by zation can have two diﬀerent patterns of manifestation: nod- intramuscular injection). We decided on intramuscular ther- ular pattern with ulcerative masses, typical of invasive ductal apy to overcome the patient’s dysphagia. carcinoma (IDC), or a diﬀuse mural involvement, typical of After four months of hormone therapy, CT scan was per- invasive lobular carcinoma (ILC). In the latter case, multiple formed and reported stable disease. The patient also experi- and deep biopsies are recommended for the diagnosis enced clinical improvement with weight increase (1 kg) and because sometimes the scirrhous and ﬁbrotic reaction can palliation of dysphagia. Sporadic postprandial vomiting was invade the gastric wall without mucosal involvement. still present. Although the cases described are not many, the lobular In January 2017, CA15.3 was normalized (3.8 U/ml) and histological subtype is the most common cause of metastatic a new EGDS with biopsies was performed. Histology con- gastric linitis plastica caused by breast cancer . Taal et al. ﬁrmed localization of adenocarcinoma with immunohisto- performed a retrospective analysis in a 15-year period show- chemistry ER 90%, PgR 35%, CK7 3+, gross cystic disease ing that 83% of patients with breast cancer and gastric metas- ﬂuid protein 15 (GCDFP-15) 3+, and HER2 1+ (Figure 1). tasis have lobular histological subtype . Rare cases of linitis The patient is still in a good clinical condition with plastica of the rectum as a possible clinical presentation of ECOG PS 1 up to this day. Supportive enteral nutrition is still lobular breast carcinoma are also described [7–10]. However, ongoing, but dysphagia has signiﬁcantly improved. Hormone the biological mechanism underlying this unusual correla- therapy with fulvestrant is still ongoing and well tolerated. tion is not yet clear. The last radiological evaluation was performed in February The presence of the metastatic lesion without primitive 2018, and it showed a stable disease. carcinoma represents a heterogeneous group deﬁned as “car- Additionally, because of a potential genetic correlation cinoma of unknown primary” (CUP). They account for 3– between diﬀuse gastric carcinoma and early-onset lobular 5% of all tumors, and the prognosis is poor . Probably, breast carcinoma , we also performed a genetic evaluation these tumors acquire the capacity to metastasize before the and searched for CDH1 germline mutations, but no genetic development of a clinically evident primary lesion . A abnormalities were identiﬁed. In our case, the absence of historical autopsy study showed that the breast was the Case Reports in Oncological Medicine 3 primary tumor site in CUP syndrome in only 2% of the cases Conflicts of Interest [12, 13]. The authors declare that there is no conﬂict of interest Immunohistochemistry is fundamental to correctly iden- regarding the publication of this article. tify the primary site and, in our case, was essential to decide the therapeutic strategy. Since about 80% of human breast cancer cells express hormone receptors, ER and PR statuses References are usually used as reliable markers for breast origin . However, the primary gastric carcinomas can also express  K. Fizazi, F. A. Greco, N. Pavlidis, G. Daugaard, K. Oien, and sex hormone receptors. According to Tokunaga and col- G. Pentheroudakis, “Cancers of unknown primary site: ESMO leagues, the rates of positivity are about 26.6% for ER and Clinical Practice Guidelines for diagnosis, treatment and 20.6% for PR . In a more recent analysis by Matsui follow-up,” Annals of Oncology, vol. 26, Supplement 5, et al., the positivity is about 32% and 12% for ER and PR, pp. v133–v138, 2015. respectively . 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