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
z A 24-year-old man was suffered from a rapidly growing palatal lesion discovered 2 months earlier.
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
the lesion was interpreted as a nonspecific inflammatory lesion o
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.
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.
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.
Angiocentric T-cell lymphoma
z She received radiotherapy and 4 cycles of chemotherapy,but lost to follow-up at 7 months of treatment
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
z Results for other markers including C20, CD56, and CD34 were negative.
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
atherosclerosis, local surgical trauma, allergy, thromboangiitis, obliterans (Buerger disease), dental injections, dental prosthesis, sickle cell anemia , heavy alcohol and tobacco consumption.
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
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.
z The author postulate that vascular occlusion by the neoplastic lymphoid cells produced ischemia and contributed to the development of the salivary gland lesion.
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
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