Presentation of the cases

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內文:

Introduction

z Necrotizing sialometaplasia (NS) is a benign inflammatory condition that may occur in any site containing salivary gland elements,

z It most frequently involves the minor salivary glands of the hard palate.

z NS is characterized by

¾ lobular necrosis with granulation tissue and inflammation.

¾ pseudoepitheliomatous hyperplasia of the surface epithelium

¾ squamous metaplasia of ducts and acini with preservation of the lobular architecture.

z The cause of this phenomenon is uncertain, mostly related to local trauma and injury:

¾ dental injections,

¾ alcohol abuse, smoking, cocaine use,

¾ upper respiratory infections, prior surgery, and adjacent tumor growths .

z Most authors believe NS is the result of ischemia that leads to local infarction.

z Association of NS with neoplastic conditions is a relatively rare event

Materials and methods

z Histologic analysis :

paraffin-embedded tissue and stained by routine hematoxylin-eosin method

z Immunohistochemical studies

using primary antibodies to CD45 (LCA), CD43 (MTI), CD45RO

(UCHL-1), CD15 (LeuM-1), CD30 (BerH2), CD4, CD8(Dako, Carpinteria, CA), CD57 (Biogenex, San Ramon, CA), Epstein-Barr latent membrane Protein (LMP-1, Dako), CD20(L26), CD3, CD56, granzyme, and TIA-1.

z In situ hybridization for Epstein-Barr virus (EBV)–encoded RNA (EBER)

Presentation of the cases

Case1

z A 24-year-old man was suffered from a rapidly growing palatal lesion discovered 2 months earlier.

o

Physical examination :

a 3 × 2-cm ulcerated and bleeding lesion extending from the hard palate to the oropharynx.

z Lymphadenopathy(-) z Needle biopsy :

原文題目(出處): Necrotizing sialometaplasia of the palate associated with angiocentric T-cell lymphoma. Annals of Diagnostic Pathology 2009;13:60-4

原文作者姓名: Hugo Dominguez-Malagon, Adalberto Mosqueda-Taylor, Ana Maria Cano-Valdez

通訊作者學校: Pathology Department, Instituto Nacional de Cancerología, Mexico City 14080, Mexico

Health Care Department, Universidad Autonoma

Metropolitana Xochimilco, Mexico City 04960, Mexico 報告者姓名(組別): Intern J組 吳庭祐

報告日期: 98.04.07

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the lesion was interpreted as a nonspecific inflammatory lesion o

Diagnosis :

NS associated with angiocentric T-cell lymphoma

z Bone marrow biopsy :

failed to identify any involvement by lymphoma

z He received 5 cycles of chemotherapy, but the response was poor and the lesion extended into the nasal mucosa, producing obstruction of the left side.

z The patient had intermittent fever but results of repeated cultures of the ulcer were negative for bacteria.

z He developed pancytopenia and persistent fever suggestive of virus-associated hemophagocytic syndrome.

z Finally he died of acute hepatic failure 5 months after admission.

Histologic findings

z an ulcerated lesion characterized by

abundant granulation tissue associated with

„ pseudoepitheliomatous hyperplasia of the covering epithelium

„ acinar necrosis of the adjacent salivary glands

„ squamous metaplasia of ducts and acini with preservation of the lobular architecture

z A profuse lymphocytic infiltrate with a population of atypical cells surrounded the epithelial structures and extended downward to infiltrate adjacent glandular tissue

z The cell population showed a diffuse growth pattern with some patchy necrotic areas, and it was prominent around and within blood vessels.

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Immunohistochemical findings

z Results for other markers including CD20, CD4, and CD8 were negative Case 2

z A 39-year-old woman was suffered from rhinorrhea, otalgia, and nasal obstruction accompanied by fever and weight loss of 20 kg.

z Computerized axial tomography scan

a tumor involving all paranasal sinuses with a zone of osteolysis of the medial wall of the maxillary sinus.

z Biopsy

Angiocentric T-cell lymphoma

z She received radiotherapy and 4 cycles of chemotherapy,but lost to follow-up at 7 months of treatment

Histologic findings

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z Respiratory epithelium with extensive areas of coagulative necrosis

z The accessory mucous glands featured lobular necrosis and squamous metaplasia involving ducts and acini with preservation of the lobular architecture

z A diffuse infiltrate with a population of large atypical lymphoid cells surrounded the epithelial structures and invaded the walls of some blood vessels

Immunohistochemical findings

z Results for other markers including C20, CD56, and CD34 were negative.

Discussion

z More than 75% of the reported cases of NS occurred in the salivary glands of the palate.

z It usually starts as a nonulcerated swelling, often associated with pain or paresthesia, which eventually breaks to produce a deep and well-demarcated ulcer that ranges from less than 1 to more than 5 cm in diameter and heals spontaneously after 4 to 6 weeks.

z Necrotizing sialometaplasia is believed to be a lesion that results from local ischemia, secondary to blockage of blood supply to the salivary gland tissue which is based on histology.

z Because of its clinical and microscopic features, it has been confused with squamous carcinoma and mucoepidermoid carcinoma.

z Morphologic evidence is provided by the presence of lobular infarction in the early phases of the disease.

z NS may be in association with other conditions that may produce local vascular obliteration

Nonneoplastic conditions:

atherosclerosis, local surgical trauma, allergy, thromboangiitis, obliterans (Buerger disease), dental injections, dental prosthesis, sickle cell anemia , heavy alcohol and tobacco consumption.

Neoplasm including:

Mixed tumor, monomorphic adenoma, Warthin tumor, and rhabdomyosarcoma

Angiocentric T/NK cell lymphomas (Lethal midline granuloma syndrome)

z The lesions that originate in the nasal cavity, paranasal sinuses, and hard palate constitute a diagnostically challenging group of diseases

z Most of these lesions have proved to be malignant lymphomas, with a predominance of T/NK-cell lineage in Asian and some Latin-American populations.

z It produce widespread necrosis, which is the result of vascular angioinvasion and angiodestruction by the cytotoxic cells and the surrounding inflammatory cell infiltrate.

z The 2 cases presented here could be due to local ischemia of palatine salivary

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glands, possibly as a consequence of angioinvasion and angiodestruction by the neoplastic lymphocytes.

z Most authors agree that most angiocentric lymphomas are derived from Epstein-Barr virus–infected cytotoxic lymphocytes of both NK- and T cell lineages

z Case 1 was characterized as CD56−, CD57+. CD3+, TIA-1+, CD4−, CD8+, and EBER+

z case 2 as CD3+, CD56−, and granzyme+. They are not considered of NK-cell lineage but better classifiable as cytotoxic T-cell lymphomas .

z EBV is a significant and consistent finding among NK- and T-cell malignant lymphomas and, regardless of geographic origin, this infectious agent has been demonstrated in more than95% of cases .

z TIA-1 and granzyme are not limited to NK-cell lymphomas but are also seen in cytotoxic lymphomas of true T-cell lineage

z The question of how to classify EBER+, CD56−,and TIA-1+ angiocentric lymphomas of T-cell lineage occurring in similar sites with similar morphology still remains to be resolved.

Conclusions

z The author postulate that vascular occlusion by the neoplastic lymphoid cells produced ischemia and contributed to the development of the salivary gland lesion.

Questions

題號 題目

1 Which disease is often cause destructive inflammatory condition in the palatal salivary gland and easily misdiagnoses as squamous cell carcinoma or

mucoepidermoid carcinoma ? (A) Basal cell adenoma

(B) Necrotizing sialometaplasia (C) Warthin tumor

(D) Acinic cell adenocarcinoma

答案(B) 出處:Oral & Maxillofacial Pathology,second edition,SAUNDERS p.405-406

題號 題目

2 This 62-year-old man has a lesion over palatal side,the surface is ulcerative.

.

In histology, it shows atypical lymphoid cells infiltrating the wall and filling the lumen of a blood vessel? Which disease is the most likely?

(A) Multiple myeloma

(B) Angiocentric T-cell lymphoma (C) Burkitt’s lymphoma (D) Plasmacytoma

答案(B) 出處:Oral & Maxillofacial Pathology,second edition,SAUNDERS p.524-526

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