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CASE REPORT SATOH ET AL THYROID MALT LYMPHOMA PULMONARY METASTASIS

References 1. Kuo T, Shih LY. Histologic types of thymoma associated with pure red cell aplasia: a study of five cases including a composite tumor of organoid thymoma associated with an unusual lipofibroadenoma. Int J Surg Pathol 2001;9: 29–35. 2. Moran CA, Zeren H, Koss MN. Thymofibrolipoma. A histologic variant of thymolipoma. Arch Pathol Lab Med 1994;118:281–2. 3. Qu G, Yu G, Zhang Q, Ma J, Wang X. Lipofibroadenoma of the thymus: a case report. Diagn Pathol 2013;8:117. 4. Aydin Y, Sipal S, Celik M, et al. A rare thymoma type presenting as a giant intrathoracic tumor: lipofibroadenoma. Eurasian J Med 2012;44:176–8.

Rare Pulmonary Metastasis From Thyroid Mucosa-Associated Lymphoid Tissue Lymphoma Kiyoshi Satoh, PhD, Takuya Morita, PhD, Satoshi Fumimoto, MD, Hiroyuki Tsuji, MD, Yoshio Ichihashi, MD, Kaoru Ochi, MD, Nobuharu Hanaoka, PhD, Yoshikatsu Okada, PhD, and Takahiro Katsumata, PhD

FEATURE ARTICLES

Departments of Thoracic Surgery, Respiratory Medicine, and Pathology, Osaka Medical College Hospital, Takatsuki, Japan

Primary pulmonary lymphomas constitute up to 1% of all pulmonary malignancies. Patients with mucosaassociated lymphoid tissue (MALT) lymphoma represent approximately 90% of patients with primary pulmonary lymphoma. Most pulmonary MALT lymphomas are primary tumors. Pulmonary metastasis is extremely rare. A 65-year-old woman was diagnosed with a thyroid MALT lymphoma in 2008 and underwent total thyroidectomy, followed by chemotherapy. After 5 years of follow-up, she referred to our hospital with an abnormal shadow on a chest roentgenogram. She underwent video-assisted thoracoscopic surgery and was diagnosed with metastatic thyroid MALT lymphoma. Postoperatively, she was treated with chemotherapy, including rituximab, and is alive without recurrence. (Ann Thorac Surg 2015;100:700–2) Ó 2015 by The Society of Thoracic Surgeons

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ucosa-associated lymphoid tissue (MALT) lymphoma was first described by Issacson and Wright in 1983 and is now established as a distinct type of nonHodgkin lymphoma. A 65-year-old woman who underwent total thyroidectomy for thyroid MALT lymphoma in 2008 was referred to our hospital for an abnormal shadow on her chest roentgenogram (Fig 1A). Chest computed tomography

Accepted for publication Sept 9, 2014. Address correspondence to Dr Satoh, Department of Thoracic Surgery, Osaka Medical College Hospital, 2-7 Daigaku-cho, Takatsuki 5690-8686 Japan; e-mail: [email protected].

Ó 2015 by The Society of Thoracic Surgeons Published by Elsevier

Ann Thorac Surg 2015;100:700–2

revealed a 25-mm  15-mm mass in the S6 segment of the right lung with bilateral cervical lymphadenopathy (Fig 1B), Imaging with 18F fluoro-deoxyglucose positron emission tomography (FDG-PET) showed high accumulation in the lung lesion, bilateral cervical lymph nodes, and abdominal paraaortic lymph nodes, with maximum standardized uptake values of 5.6, 7.1, and 4.9, respectively (Fig 2). Because a definitive diagnosis was not made by bronchoscopic examination and the cervical lymph nodes did not have palpable masses, we intervened surgically, and the patient underwent a video-assisted thoracoscopic procedure in 2013. Because the lesion was located in the center of the lung, we performed S6 segmentectomy. Frozen sections of the lesion showed atypical lymphoid cells infiltrating the bronchial epithelium; the diagnosis was lymphoma. Postoperative pathologic hematoxylin and eosin findings showed diffuse proliferation of small lymphoid cells that resembled centrocyte-like cells (Fig 3A), and these cells constituted lymphoepithelial lesions (Fig 3B). Immunohistochemical staining was positive for CD79a in all atypical lymphocytes and centrocyte-like cells; staining was negative for CD3, CD5, CD10, cyclin D1, and immunoglobulin M. These findings were consistent with those from the patient’s earlier thyroid specimens, and she was diagnosed with MALT lymphoma. After the operation, she underwent rituximab, etoposide, prednisolone, cytarabine, and cisplatin 6-day-cycle chemotherapy, and all lesions disappeared.

Comment Extranodal marginal zone lymphoma or MALT lymphoma accounts for 6% to 8% of all newly diagnosed cases of non-Hodgkin lymphoma and ranks third in frequency after diffuse large B-cell lymphoma and follicular lymphoma. MALT occurs in lymphoid tissues that are integral to the organ, such as Peyer patches of the small intestine, and in lymphoid tissues that are not present physiologically such as in the stomach, lung, thyroid, salivary glands, and ocular adnexa (conjunctiva, lacrimal gland). MALT lymphomas arise from the latter. Most studies report a median age at diagnosis of approximately 60 years and approximately equal numbers of men and women. Primary sites are the stomach (50% of patients), lung (14%), head, neck, and salivary glands (14%), orbit (14%), skin (11%), thyroid (4%), and mammary gland (4%). MALT lymphoma usually arises within tissues affected by chronic inflammatory disorders of an autoimmune or infectious etiology. Examples are MALT lymphoma of the stomach in patients with Helicobacter gastritis and MALT lymphoma of the thyroid in patients with Hashimoto thyroiditis. Because inflammation is present, various inflammatory cells are seen in the background in addition to the plasma cells. Before the malignant nature of the disease was understood, what we now recognize as MALT lymphomas 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2014.09.070

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CASE REPORT SATOH ET AL THYROID MALT LYMPHOMA PULMONARY METASTASIS

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Fig 1. (A) A chest roentgenogram shows a solitary mass in the middle-lung field of the right lung. (B) A computed tomography scan shows the tumor, measuring 25 mm 15 mm, is present in the S6 segment of the right lower lobe.

were considered to be nonneoplastic lesions such as reactive lymphoreticular hyperplasia or pseudolymphoma in the stomach, lung, salivary glands, and thyroid.

FEATURE ARTICLES

Fig 2. A 18F fluoro-deoxyglucose positron emission tomography image shows high accumulation in the lung lesion, bilateral cervical lymph nodes, and abdominal para-aortic lymph nodes (maximum standardized uptake values of 5.6, 7.1, and 4.9, respectively).

The specific etiology differs by organ and region. Involvement of H pylori in the stomach, Campylobacter jejuni in the intestine, Chlamydia psittaci in the ocular adnexal, and Borrelia burgdorferi in the skin has been reported [1]. However, the reported frequency of involvement of the infectious agent has been inconsistent; geographic differences of the ocular adnexal and skin are particularly large. The involvement of H heilmannii, Epstein-Barr virus, and hepatitis C virus (HCV) has been reported in the stomach [2]. About 35% of MALT lymphomas are positive for HCV in several organs other than the stomach [3]. HCV is associated with various low-grade B-cell lymphomas, but whether a relationship exists between HCV and MALT lymphoma is unclear. MALT lymphoma has been reported to recur at a relatively high rate. In a study of 86 cases of MALT lymphoma, Markus and colleagues [4] reported that recurrence occurred in approximately half of MALT lymphomas that developed in organs other than the stomach, but none of the thyroid MALT lymphomas recurred. Because there have been only a few report of metastasis of thyroid MALT lymphoma, it can be said that lung metastasis from thyroid MALT lymphoma is very rare [5]. Pulmonary MALT lymphoma is a diagnostic challenge. Imaging findings of primary pulmonary MALT lymphoma are nonspecific, and nodule, consolidation, and ground-glass opacities are mixed in many cases. The lesions can be solitary, multiple, unilateral, or bilateral, and multiple (70%) and bilateral (43% to 60%) lesions are reported to be more common than solitary and unilateral lesions [6]. Metastatic pulmonary MALT lymphoma is very rare, and imaging features are unknown because few cases have been reported. Therefore, a lung biopsy specimen is necessary for MALT lymphoma diagnosis. Recently, with the development of auxiliary methods, immunophenotyping, cytogenetic and molecular testing, and transbronchial examination have been able to detect MALT lymphoma in some cases, but surgical lung biopsy is still required in many patients. We performed a surgical biopsy because noninvasive procedures were unable to provide a definitive diagnosis.

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CASE REPORT MEHTA ET AL MEDIASTINAL PARAGANGLIOMAS

Ann Thorac Surg 2015;100:702–5

FEATURE ARTICLES

Fig 3. Pathologic findings of the resected specimen show (A) diffuse proliferation of small lymphoid cells (centrocyte-like cells) (hematoxylin and eosin stain, original magnification 120) and (B) lymphoepithelial lesions (hematoxylin and eosin stain, original magnification 40).

Segmentectomy was unavoidable because the lesion was located in the central lower lobe. Video-assisted thoracoscopic segmentectomy was a reasonable approach for our patient with metastatic pulmonary MALT lymphoma because it is minimally invasive. In general, the therapy for MALT lymphoma varies, depending on the specific site. In thyroid MALT lymphoma, the usual therapy is irradiation for localized disease and chemotherapy for disseminated disease, and recurrence is treated with irradiation or a combination of irradiation and chemotherapy. Treatment with rituximab, with or without chemotherapy, has been reported recently, and the prognosis is improving [7, 8]. After the surgical procedure, our patient received rituximab, etoposide, prednisolone, cytarabine, and cisplatin 6-day-cycle chemotherapy. All lesions disappeared, and she is alive without recurrence.

References 1. Guidoboni M, Ferreri AJ, Ponzoni M, et al. Infectious agents in mucosa-associated lymphoid tissue-type lymphomas: pathogenic role and therapeutic perspectives. Clin Lymphoma Myeloma 2006;6:289–300. 2. Montalban C, Norman F. Treatment of gastric mucosaassociated lymphoid tissue lymphoma: Helicobacter pylori eradication and beyond. Expert Rev Anticancer Ther 2006;6: 361–71. 3. Acraini L, Burcheri S, Rossi A, et al. Prevalence of HCV infection in nongastric marginal zone B-cell lymphoma of MALT. Ann Oncol 2007;18:346–50. 4. Markus R, Berthold S, Stefan W, et al. High relapse rate in patients with MALT lymphoma warrants lifelong follow-up. Clin Cancer Res 2005;11:3349–52. 5. Pier LZ, Massimo M, Piero G, et al. Nongastrointestinal lowgrade mucosa-associated lymphoid tissue lymphoma: analysis of 75 patients. J Clin Oncol 1999;17:1254–8. 6. Bae YA, Lee KS, Han J, et al. Marginal zone B-cell lymphoma of bronchus-associated lymphoid tissue: imaging findings in 21 patients. Chest 2008;133:433–40. 7. Kiesewatter B, Mayerrhoefer ME, Lukas J, et al. Rituximab plus bendamustine is active in pretreated patients with extragastric marginal zone B cell lymphoma of the mucosaassociated lymphoid tissue (MALT lymphoma). Ann Hematol 2014;93:249–53. 8. Ennishi D, Yokoyama M, Mishima Y, et al. Rituximab plus CHOP as an initial chemotherapy for patients with disseminated MALT lymphoma. Leuk Lymphoma 2007;48: 2241–3. Ó 2015 by The Society of Thoracic Surgeons Published by Elsevier

Rare Middle Mediastinal Paraganglioma Mimicking Metastatic Neuroendocrine Tumor Christopher K. Mehta, MD, Colin T. Gillespie, MD, Xiaoqi Lin, MD, PhD, Anjana Yeldandi, MD, Malcolm DeCamp, MD, and Ankit Bharat, MD Department of Surgery, Division of Thoracic Surgery; Department of Medicine, Division of Pulmonary & Critical Care Medicine; and Department of Pathology, Northwestern Memorial Hospital, Chicago, Illinois

Mediastinal paragangliomas are rare neural crest derived tumors that may produce symptoms of excess catecholamine production or mass effect. Paragangliomas can histologically mimic neuroendocrine tumors. Further, both can arise in similar locations. We report a patient who presented with a right upper lobe as well as middle mediastinal lesion. Preoperative biopsy as well as intraoperative frozen section of these lesions failed to distinguish between paraganlioma or neuroendocrine tumor, necessitating a right upper lobectomy and complete mediastinal lymphadenectomy. Final pathology revealed carcinoid tumorlets in the right upper lobe and a middle mediastinal paraganglioma. (Ann Thorac Surg 2015;100:702–5) Ó 2015 by The Society of Thoracic Surgeons

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aragangliomas are rare tumors of the mediastinum and are usually located in the posterior mediastinum. They arise from chromaffin cells of the autonomic nervous system but can be difficult to differentiate from neuroendocrine tumors. Here we present a case of an extremely rare middle mediastinal paraganglioma mimicking a metastatic carcinoid and discuss the important clinical and pathologic findings that differentiate these tumors. A 66-year-old female presented with episodic hot flashes and diaphoresis for over 6 months. Her medical history Accepted for publication Sept 9, 2014. Address correspondence to Dr Bharat, 676 N Saint Clair St, Ste 650, Chicago, IL 60611; e-mail: [email protected].

0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2014.09.068

Rare Pulmonary Metastasis From Thyroid Mucosa-Associated Lymphoid Tissue Lymphoma.

Primary pulmonary lymphomas constitute up to 1% of all pulmonary malignancies. Patients with mucosa-associated lymphoid tissue (MALT) lymphoma represe...
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