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□ CASE REPORT □

Mucosa-associated Lymphoid Tissue Lymphomaof the Trachea in a Patient with Breast Cancer

Hitomi Yamashita 1, Yasuto Ueda 1, Katsuyuki Tomita 1, Tsuyoshi Kitaura 1,

Tomoaki Koshobu 1, Yoshimasa Suzuki 2 and Eiji Shimizu 3

Abstract

We herein report the case of a 93-year-old woman with breast cancer on the left side. Preoperative com-

puted tomography of the chest showed irregularities and narrowing of the mid-trachea. Bronchoscopy was

performed, and the results of a biopsy supported a diagnosis of mucosa-associated lymphoid tissue (MALT)

lymphoma. The patient responded to treatment with prednisone alone, with a reduction in the size of the le-

sion. MALT lymphoma of the trachea is extremely rare, and there are only a few case reports of double can-

cer, i.e., MALT lymphoma of the trachea and breast cancer.

Key words: breast cancer, MALT lymphoma, prednisone, trachea

(Intern Med 54: 2041-2044, 2015)(DOI: 10.2169/internalmedicine.54.3925)

Introduction

Mucosa-associated lymphoid tissue (MALT) lymphoma is

most commonly found in the stomach, lungs, orbital soft tis-

sue, salivary glands and thyroid; involvement of the trachea

is extremely rare (1, 2). There are no clear guidelines for the

treatment of MALT lymphoma, and, with respect to lesions

located in the trachea, a broad range of treatments have

been shown to be effective, including surgical resection, ra-

diotherapy, bronchoscopic therapy, chemotherapy, immuno-

therapy (rituximab) and immunochemotherapy.

This report describes a rare case of MALT lymphoma of

the trachea in a 93-year-old woman with breast cancer. The

administration of prednisone alleviated her symptoms and

reduced the size of the MALT lymphoma in the trachea.

Case Report

The patient was a 93-year-old woman who presented to

the thoracic surgery department of Yonago Medical Center

complaining of a self-destructed left breast tumor. In the fol-

lowing month, the patient complained of wheezing during

movement. She had a previous medical history of hyperten-

sion and Alzheimer’s disease, which had been treated with

enalapril (5 mg daily) and donepezil (5 mg daily), respec-

tively. Her medical history did not include chronic autoim-

mune diseases, such as Sjögren syndrome and Hashimoto’s

thyroiditis, or chronic infections, such as that with Helico-bacter pylori.

On a physical examination, the patient presented with a

left breast mass and chest wheezing during inspiration, with

no other remarkable findings. Renal and liver function tests,

hemogram findings and the lactic dehydrogenase level were

normal.

A chest X-ray showed tracheal deviation to the left side

(Fig. 1). Computed tomography (CT) of the chest demon-

strated irregularities of the tracheal wall and narrowing of

the mid-trachea (Fig. 2A).

Flexible bronchoscopy was performed during intravenous

anesthesia for mastectomy, the results of which revealed a

protuberant lesion involving all the walls of the trachea, ex-

tending from the trachea to the left main bronchus (Fig. 3).

A biopsy showed aggregation of small lymphocytes, and the

morphological and immunohistochemical features confirmed

the presence of low-grade non-Hodgkin’s B-cell lymphoma

１Department of Respiratory Medicine, Yonago Medical Center, Japan, ２Department of Thoracic Surgery, Yonago Medical Center, Japan and ３Di-

vision of Medical Oncology and Molecular Respirology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori Uni-

versity, Japan

Received for publication August 18, 2014; Accepted for publication January 6, 2015

Correspondence to Dr. Katsuyuki Tomita, tomita-katsuyuki@nho-yonago.jp

Intern Med 54: 2041-2044, 2015 DOI: 10.2169/internalmedicine.54.3925

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Figure　1.　Chest X-ray showing a mass protruding into the tracheal lumen.

Figure　2.　Chest computed tomography (CT) demonstrating irregularities of the tracheal wall and narrowing of the mid-trachea before (A) and four weeks after treatment with prednisone (B).

with plasmacytic differentiation, suggestive of MALT lym-

phoma. Hematoxylin and Eosin (H&E) staining disclosed

characteristic infiltration of centrocyte-like cells surrounding

the reactive follicles and forming lymphoepithelial lesions

(Fig. 4A). An immunohistochemical panel revealed that the

lymphoma cells were positive for CD20 and bcl2 and nega-

tive for CD5 and CD10 (Fig. 4B-E). The pathology of the

breast cancer was pT4bN0M0, stage III, solid-tubular carci-

noma, with positive estrogen- and progesterone-receptor im-

munostaining, a proliferative index as high as 5.8% and a

negative human epidermal growth factor receptor type 2

(HER2) status.

Treatment with prednisone (30 mg/day) was effective in

improving the patient’s symptoms. Chest CT performed af-

ter the four weeks of therapy showed a reduction in the size

of the lesion in the trachea (Fig. 2B), and the dose of pred-

nisone was tapered four weeks after the first dose. Although

the patient had exhibited good disease control, she died a

natural death while undergoing treatment with 10 mg/day of

prednisone in a nursing home, five months after the opera-

tion.

Discussion

MALT lymphoma was first described by Issacson and

Wright in 1983 in a small series of patients with low-grade

B cell gastrointestinal lymphoma (3). MALT lymphomas are

characterized by the presence of neoplastic marginal cells

that exhibit a variable combination of colonization of reac-

Intern Med 54: 2041-2044, 2015 DOI: 10.2169/internalmedicine.54.3925

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Figure　3.　Bronchoscopic view showing an intratracheal pol-ypoid lesion.

Figure　4.　Histological examination demonstrating centro-cyte-like cells infiltrating the mucosa (A, Hematoxylin and Eo-sin staining, ×100). Many of the cells exhibited positive staining for CD20 (B, ×200) and bcl2 (C, ×200) and negative staining for CD5 (D, ×200) and CD10 (E, ×200).

tive germinal centers, plasma differentiation and destructive

epithelial infiltration forming lymphoepithelial lesions. Im-

munohistochemical examinations often show characteristic B

cell lymphoma with monoclonal expansion by monotypic

cytoplasmic immunoglobulin. Although MALT lymphomas

occur most frequently in the gastrointestinal tract, they can

also arise in a number of non-gastrointestinal sites, such as

the lungs, orbital soft tissue, salivary glands and thyroid (1).

Among nongastrointestinal MALT lymphomas, pulmonary

lymphomas are the most frequent, representing up to 19%

of MALT lymphomas (3). As primary tracheal MALT lym-

phomas are extremely rare, most reports in the literature are

case reports (4-6).

The current case involved double cancer, breast cancer

and MALT lymphoma of the trachea. A cancer prone pheno-

type may exist in MALT lymphoma patients. For example,

Zucca et al. observed a high incidence of other neoplasms

(20%) in patients with low-grade gastric MALT lym-

phoma (7). However, Au et al. reported that, in their study,

MALT lymphoma patients did not appear to have a signifi-

cantly increased rate of cancer compared with an age-

matched population followed for the same period of

time (8).

Previous reports have shown the effectiveness of a range

of treatments, including surgical resection, radiotherapy,

bronchoscopic therapy, chemotherapy, immunotherapy and

immunochemotherapy, i.e., rituximab, cyclophosphamide,

adriamycin, oncovin and prednisone (R-CHOP) (4-6). How-

ever, combination regimens have not been proven to be any

more effective than single chemotherapy regimens (9). In

the present case, despite the positive test findings for CD20,

rituximab was not selected for treatment due to the age of

the patient. In this case, treatment with prednisone alone im-

proved the patient’s symptoms and reduced the size of the

mass. Prednisone monotherapy may a less intense che-

motherapeutic regimen for older patients, and the effective-

ness of antibiotic therapy has also been reported in some

cases of pulmonary MALT lymphoma (10).

As our patient presented with symptomatic stenosis of the

airway, we selected treatment with prednisone, taking into

consideration her advanced age. Importantly, prednisone was

effective in reducing her symptoms and the size of the le-

sion.

This is the first case of MALT lymphoma of the trachea

in a patient with breast cancer treated with prednisone alone.

The details of this case suggest that prednisone monotherapy

may be an effective strategy for treating MALT lymphoma.

MALT lymphoma of the trachea is an extremely rare and

indolent disease and must be considered in the differential

diagnosis of airway lesions. Tracheal tumors may mimic the

features of asthma and chronic obstructive pulmonary dis-

ease (COPD) and should be kept in mind as a rare cause of

asthma and COPD-like symptoms. In the current case, pred-

nisone monotherapy was beneficial in managing the tracheal

lymphoma in our patient.

The authors state that they have no Conflict of Interest (COI).

Intern Med 54: 2041-2044, 2015 DOI: 10.2169/internalmedicine.54.3925

2044

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