RETROSPECTIVE STUDY REGARDING LYMPHOMAS OF THE PAROTID GLAND

Transcription

1 RETROSPECTIVE STUDY REGARDING LYMPHOMAS OF THE PAROTID GLAND Andrei Nicolau*, Constantin Mihai, Otilia Boișteanu, Doriana Forna, Eugenia Popescu Grigore T. Popa" University of Medicine and Pharmacy - Iași, Romania, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery *Corresponding author: Andrei Nicolau, OMS trainee, MD, DMD, PhD student Gr. T. Popa University of Medicine and Pharmacy, - Iași, Romania ; nicolauandrei@yahoo.com ABSTRACT Aim of the study Carrying out a clinical statistical study of patients diagnosed with parotid lymphomas in the Oral and Maxillofacial Surgery Clinic of Iasi with a view to improving the methods of early diagnosis and establishing the best therapeutic indication. Material and methods The study was carried out on a number of 56 patients diagnosed with parotid lymphomas and surgically treated in the period The study relied on all the data registered for each patient in the observation charts and in the anatomo-pathological bulletins, focusing on the information about the histopathological form of the lymphoma, its location, the surgical intervention applied and the post-operative sequelae. Results Parotid gland lymphomas represent about 5% of all parotid tumors and 13% of the malignant ones. 80% were non-hodgkin lymphomas. The highest incidence was found in the age decades and 41-50, predominantly in males. 80% of the parotid lymphomas were on the superficial lobe. In most cases, the facial nerve was preserved. Conclusions The anatomical particularities and the increased quantity of tumoral tissue necessary for the anatomo-pathological examination require the removal of the tumor by parotidectomy in the cases when there was only one tumoral location at the level of the parotid gland. Keywords: parotid gland, lymphomas, Hodgkin INTRODUCTION Lymphomas represent 3% of all malignant diseases and 5% of all the head and neck neoplasias, being the most frequent malignant hematopathia [1,2]. At the level of the salivary glands, lymphomas are relatively rare, constituting 2-5% of the malignant diseases thus localized [3]. Salivary gland lymphomas involve the major salivary glands most frequently, the parotid gland being the most affected, in over 70% of the cases [4,5]. Parotid gland lymphomas can have their origin both in the intraparotid lymph nodes and in the glandular tissue, so that a differential diagnosis between a nodal lymphoma and an extranodal one becomes difficult to give, particularly in the case of nodal parotid lymphomas extending into glandular parenchyma [3]. Both types of lymphomas can be formed at the level of the parotid glands: Hodgkin and non-hodgkin. Updated WHO classification of lymphomas use as main element the involved cellular line, presenting a large variety of tumoral forms. The parotid Hodgkin lymphomas are very 57

2 rare [6]. Both the classical Hodgkin lymphoma and the nodular lymphocytic predominant Hodgkin lymphoma were reported, affecting the parotid gland exclusively [7]. Rarely, Hodgkin parotid lymphomas can have their origin in the lymphomatose component of the Warthin tumor [8,9]. Non-Hodgkin parotid lymphomas are predominantly B-cell lymphomas, represented by the MALT lymphoma, follicular lymphoma and the diffuse large B- cell lymphoma [3]. The MALT lymphoma is frequently found at the level of gastrointenstinal extranodal sites, of the thyroid and the salivary glands [10]. The extranodal marginal zone B-cell lymphoma of MALT-type is the most frequent lymphoma originating in the salivary glands [3]. The lymphoepitelial sialadenitis associated to the Sjogren syndrome represents a forerunning lesion of the extranodal marginal zone B-cell lymphoma of MALTtype [11]. The parotid follicular lymphoma in most cases has its origin in the intraparotid lymph nodes, afterwards infiltrating the glandular parenchyma or it may be the extension of a extranodal marginal zone B-cell lymphoma of MALT-type, with multiple areas of follicular colonization [5]. Apart from the intraparotid lymph nodes implication, the follicular lymphoma can primarily affect the glandular parenchyma. Other extranodal locations inside the head and neck are the orbit and the thyroid gland [12]. The diffuse large B-cell lymphoma is an infiltration tumor frequently associating the destruction of the glandular parenchyma with the interstitial inflammation of the remaining salivary acins. The tumor contains lymphoid cells similar to centroblasts and immunoblasts, these in some cases resulting from the transformation of a extranodal marginal zone B-cell lymphoma of MALTtype [4,11]. The salivary gland lymphomas with T- cells are very rare, the most frequent being the anaplastic large cell lymphoma [14] Similarly, the extranodal NK/T-cell lymphoma of nasal-type can affect the salivary glands in few cases [15]. MATERIAL AND METHODS The retrospective clinic-statistically study of parotid gland lymphomas was carried out at the Oral and Maxillofacial Surgery Clinic of the St. Spiridon Hospital in Iasi, over a period of 63 years ( ), analyzing the data in the observation charts and the anatomopathological bulletins of the patients admitted here and diagnosed after the surgery. The following data was recorded for each patient: age, sex, the location of the tumor in the architecture of the parotid gland, the type of surgery treatment applied, the anatomopathological type of the lymphoma and the post-operative complications in connection with the facial nerve. The period included in the study was divided into decades. For the same period of time the total number of parotid tumors, both malignant and benignant, was also assessed. The objectives of the study were multiple. One was relating the parotid lymphomas to the total number of parotid tumors, especially malignant. There was also the assessment of the incidence of parotid lymphomas on age groups and depending on the patient s sex. All parotid lymphomas presented, clinically and imagistically, only one tumoural location without other locations accessible for the sampling of tissular material for the anatomo-pathological examination, being considered Ann-Arbor stage I. All cases benefited from surgical treatment. The study had in view the type of surgical treatment applied, correlated both to the 58

3 tumoral location in the structure of the gland and to the post-operative disorders od the facial nerve. RESULTS In the period between , 1153 patients with parotid tumors were admitted to the Oral and Maxillofacial Surgery Clinic in Iasi, of which 1103 were operated, 425 parotid tumors were malignant and 728 were benignant. 56 parotid lymphomas were diagnosed, representing 4,85% of the admitted parotid tumors and 13,17% of the parotid gland malignant tumors (Figure 1). The Presence of Parotid Gland lymphoma in Hospitalized Parotid Gland Tumors 95.15% Hospitalized Tumors of The Parotid Gland Lymphomas of the Parotid Gland Figure 1. Histopathologically, the 56 parotid lymphomas were: 10 Hodgkin lymphomas and 46 non-hodgkin, of which 19 MALT type lymphomas, 16 diffuse large B-cell lymphomas and 11 lymphomas that could not be attributed to any subcategory of non- Hodgkin lymphomas (Figure 2, Figure 3). Lymphomas of the parotid gland 79.17% Figure % 4.85% Hodgkin's Lymphomas Non-Hodgkin's Lymphomas 16 Figure 3. Lymphomas were constantly present in the tumoral pathology of the parotid gland in the period of time assessed. The non-hodgkin lymphomas showed an increase of incidence in the decade in the period between The Hodgkin lymphomas registered the highest values of incidence in the period and, respectively, (Figure 4) Non-Hodgkin's Lymphomas of the Protid Gland 19 MALT Lymphoma Diffuse Large B Cell Lymphoma Figure Uncategorized Lymphoma Incidence of Parotid Gland Lymphomas in Decades The distribution of parotid lymphomas on the patients age decades showed two peaks of maximum incidence in the age decade, and years. Of the 56 cases of parotid lymphomas, 37 were in men and 19 in women. The men/ women ratio was 1,94, showing an almost double incidence in men Hodgkin's Lymphoma Non- Hodgkin's Lymphoma 59

4 (Figure 5). Lymphomas of the Parotid Gland Patients according to sex (%) 66.07% Figure 5. Most tumors were situated on the superficial lobe of the parotid gland, representing 46 cases (82,14) (Figure 6). Superficial lobe of the Parotid Gland Deep Lobe of the Parotid Gland 82.14% Figure 6. The methods of surgical treatment employed were represented by parotidectomies- superficial, total or extended, with or without the partial or total sacrifice of the facial nerve. The surgical procedure applied in most cases (28 cases) was superficial parotidectomy. The total preservation of the facial nerve during surgical treatment was achieved in 41 cases. In 64% of the patients, there were no neurological complications after the surgery. The total sacrifice of the facial nerve was necessary in 5 cases (Figure7) % 8.34% 75% Figure 7. Men Women 33.93% 17.86% Preservation of the Facial Nerve Partial Facial Nerve Sacrifice Complete Facial Nerve DISCUSSIONS Parotid lymphomas are rare tumors representing 0,3% of all neoplasias and 5% of all extranodal lymphomas [16]. Feinstein et al. reported that Hodgkin lymphoma, represents 3,5% of parotid lymphomas (27,9), the follicular lymphoma (25,8%) and the diffuse large B-cell lymphoma (23,7%) [17]. Agora et al. assessed that most parotid lymphomas are MALT type, having their origin in sialadenitis associated with selfimmune disease like the Sjogren syndrome [8]. MALT lymphomas constitute a group of low grade extranodal B-cell neoplasias, that has the same clinical, pathological, immunological and molecular characteristics that appear in the context of long term lymphoid proliferation [19]. These clinical antities were the most frequent form of parotid lymphomas in the present study. The follicular lymphoma was reported to have an incidence of about 30% of the salivary gland lymphomas [12]. In the mucosa associated lymphoid tissue, follicular lymphomas appear frequently at the level of the salivary glands, the skin and the duodenum. These look like the MALT-type lymphoma, beinglow grade tumors, having a mioepitelial sialadenitis and a rare BLC-2 gene rearrangement as a substratum [12]. In its evolution, it can change into a diffuse lymphoma with large B-cells. In the present study, no follicular lymphoma was recorded. Biopsy is the essential factor in establishing the lymphoma diagnosis. This must ensure sufficient tissue material for the anatomo-pathological examination and the immunohiptochemical tests. Fine needle aspiration biopsy is not appropriate for the histological diagnosis of lymphomas [21]. The fine needle aspiration biopsy is associated with an increased number of inadequate or falsely-negative diagnoses [22]. 60

5 Chow et al. describe a rate of non diagnoses of 12,2% in the use of needle biopsy [22]. The incision biopsy is unadvisable in parotid lymphomas with only one tumoural location and without other accessible sites due to the presence of the facial nerve in this area and to the possible salivary complications (cutaneous salivary fistulas) of this procedure. In this situation, the tumour removal by parotidectomy will ensure the tissue material necessary for the histo-pathological examination. It is compulsory to add an extempore examination in order to established the indication of plasty for the postopperative defect. Extended parotidectomy was most frequently used in the case of diffuse large B- cells lymphomas, which can be explained through the high grade of these lymphomas. The existence of a group of non-hodgkin lymphomas whose subtype could not be established can be explained through the insufficiency of anatomopathological diagnosis means in the first period of time included in the study. Feinstein et al. report, in a study carried out on 2014 patients with parotid lymphomas, that only 72% had the advantage of surgical treatment. Survival decreased for the patient over 50 years old, the stage of the lymphoma being correlated to its histopathological type, as well [17]. CONCLUSIONS Parotid lymphomas are rare tumors of the salivary glands, representing 4,85% of all parotid tumors diagnosed over a period of 63 years. Of the any histopathological types of lymphomas, only three types were found affecting the parotid gland for the cases under study: Hodgkin lymphomas, MALT lymphomas and diffuse large B-cell lymphomas. MALT lymphomas were the most frequent. The treatment was first of all surgical (due to the impossibility of a biopsy requested by unique anatomical features and tumor location), involving the removal of the tumor through parotidectomy, sacrificing or preserving the facial nerve, this permitting a certainty of histopathological diagnosis. In most cases, the lymphomas were situated on the superficial lobe of the parotid gland, superficial parotidectomy being the most frequently used surgical procedure. In most cases, it was possible to preserve the facial nerve, with restitutio ad integrum of the neurological function. The postoperative prognostic of the patients was favourable. REFERENCES 1 Marcus R., Sweetenham J.W, Williams M.E Lymphoma Pathology, Diagnosis and Treatment Cambridge University Press, New York, Pamajer F.A, Haas R.L.M Neck Lymphoma in Hermans R (ed): Head and Neck Cancer Imaging, Springer, Berlin, Barnes L., Everson J.W., Reichart P., Sidransky D. Pathology & Gentics of Head and Neck Tumours IARC Press, Lyon Cheuk W, Chan JKC- Salivary glands tumors in Fletcher CDM (ed): Diagnositic histopatology of tumors vol I, 3ed Edition, Churchil Livingstone, 2007, 7: Harris NL Lymphoid proliferations of the salivary glands American Journal of Clinical Pathology 1999, 111: S Gleeson MJ, Bennet MH, Cawson RA- Lymphomas of the salivary glands- Cancer:1986; 58: Chang KL, Kamel OW, Arber DA, Horyd ID, Weiss LM Pathologyc features of nodular lymphocite predominance Hodgkin s disease in extranodal sites The American Journal of Surgical 61

6 Pathology 1995 Nov, 19 (11): Melato M, Falconieri G, Fanin R, Baccaroni M Hodgkin s disease occuring in a Warthin s tumor: first case report Pathology Research and Practice 1986 oct; 181(5): Pescarmona E, Perez M, Faraggiano T, Granati L, Baroni CD Nodal peripheral T-cell lymphoma associated with Warthin s tumor Histopatology (2): Borovecki A, Korac P, Ventura RA, Perisa MM, Bonham AH, Dominis M MALT1, BCL 10 and FOXP 1 in salivary gland mucosa-associated lymphoid tissue lymphomas- Pathology International (1): De Vita S, De Marchi G, Sacco S, Gremese E, Fabris M, Ferraccioli G Preliminary classification of nonmalignant B cell proliferation in Sjogren syndrome: perspectives on pathology and treatment based on an integrated clinico-pathologic and molecular study approach- Blood cells, molecules and diseases 2001, 27 (4): Kojima M, Nakamura S, Ichimura K, Shimuzu K, Itoh H, Masawa N Follicular lymphoma of the salivary gland: a clinicopathological and molecular study of six cases- International Journal of surgical pathology, 2001, 9(4): Quintana PG, Kapadia SB, Bahler DW, Johnson JT, Swardlow SH - Salivary gland lymphoid infiltrates associated with lymphoepitelial lesions: a clinicopathological immunofenotypic and genotypic study Human Pathology : Yamamoto H, Shirasuchi H, Yao T, Uruyn H, Oday Y, TamiyaS, Tsuneyoshi M ALK-positive anaplastic large cell lymphoma of the parotid gland- Histopatology, 2003, 43(4): Chan JK, Tsang WY, Hui PK, Sin VC, Khan SM, Sin LL T and T/Natural killer-cell lymphomas of the salivary gland: a clinicopathologic, immunohistochemical and molecular study of six cases- Human Pathology : Dispenza F, Cicero G, Mortellaro G, Marchese D, KurlamarvaG, Dispenza C Primary Non- Hodgkin lymphoma of the parotid gland Brazilian Journal of Otorhinolaryngology 2011 vol77 no5 Sao Paolo Sept/Oct Feinstein AJ, Ciarleglio M, Long X, Otremba MD, Judson BL Parotid gland lymphoma:prognostic analysis of 2014 patiens- The Laryngoscope 2013 may 123(5): Arora I, Krishnappa R, Metikurke S, Romachar S Primary malignant lymphoma of the parotid gland- Journal of Cancer Research and Therapeutics vol 8, no4, oct-dec 2012, Cohen SM, Petryk M, Varma M, Kozuch PS, Ames ED, Grossband ML Non Hodgkin s lymphoma of mucossa associated lymphoid tissue The Oncoloist, 2006, nov-dec:11(10): Davies Aj, Rosenwald A, Wright G, Lee A, Last K Transformation of follicular lymphoma to diffuse large B-cell lymphoma proceeds by distinc oncogenic mecanism- British Journal of Haematology Jan 2007:136 (2): Miron L, Miron I, Danaila C, Marinca M, Dascalescu A Terapia oncologica un abord bazat pe dovezi Ed Institutului European, Iasi, Cho HW, Kim J, Choi J- sonographically guided fine needle aspiration biopsy of major salivary gland masses: a review of 245 cases, AJR 2011;196: