scd_142.qxd 6/25/10 7:16 AM Page 160 ARTICLE NECROTIZING S I A L O M E TA P L A S I A I N A H I V P O S I T I V E PAT I E N T self-limiting, benign, inflammatory dis- who is HIV positive: a case report ABSTRACT Alessandra Dutra Silva, DDS; * Carolina Amália Barcellos Silva, DDS; Necro mous cell carcinoma. A case report is presented of a patient with NS who was t Cristiane Furuse, DDS, PhD; Rodrigo Calado Nunes e Souza, DDS, MS; Mauro Henrique Melo da Costa, DDS; Vera Cavalcanti de Araújo, DDS, izin PhD g palate. Clinically, the lesion presented Institute and Research Center São Leopoldo Mandic, Campinas, São Paulo, Brazil; Mario Gatti sialometaplasia in a patient Necrotizing sialometaplasia (NS) is a epithelium. The lesion disappeared com- Introduction ease of the minor salivary glands of the hard palate. pletely after 2 weeks. Necrotizing sialometaplasia (NS) was first reported by Abrams et The main al. in 1973, as a self- significance of the NS lesion lies sialometaplasia, HIV event and clinical factors, such as direct local trauma of the in the type produced by intuba- tion, local anesthesia, violent or induced vomiting as in patients with bulimia, use fact that it may of ill-fitting dental prosthesis, use of tobacco or cocaine, infectious processes of the be 1 1 upper respiratory tract, and systemic disease. The ischemic changes could be the mistaken for mucoepidermoid or squa- the glandular tissues and their necrosis. 1 2 2 HIV positive; the lesion was located in 1 the minor salivary glands of the hard 1 2 as a deep ulcer with slightly elevated Municipal Hospital, Campinas, São Paulo, Brazil. irregular borders and a necrotic base in *Corresponding author e-mail: email@example.com the hard palate. Histologically, the tissue was characterized by squamous meta- Spec Care Dentist 30(4): 160-162, 2010 plasia of ducts and acini, lobular coagulation necrosis, and pseudoepithe- liomatous hyperplasia of the overlying 1 limiting, benign, inflammatory disease of the minor salivary glands of the hard palate. KEY WORDS: necrotizin g Although the etiology is not clear, it is consid ered to be associated with an ische m ic 1 2 3 4,5 6 6 7 8 result of physical, chemical, and biological blood-vessel injuries leading to infarction of 2 Clinically, NS may appear as an treatment of systemic manifestations of asymptomatic or painful ulcer or AIDS. The patient had a history of ciga- swelling, resembling a crater with rette smoking and alcohol consumption. indurated edges and well-delimited His past medical history included HIV- shapes, bilateral or unilateral, and com- positive diagnosis and irregular intake of 3
scd_142.qxd 6/25/10 7:16 AM Page 161 N E C R O T I Z I N G S I A L O M E TA P L A S I A I N A H I V P O S I T I V E PAT I E N T vomiting, and several opportunistic infec- tions, such as oral and pharyngeal candidosis, genital herpes, pneu m o nia, and esophageal ulcers, in addition to the lesion of the hard palate. The patient received treatment for the systemic lesions with fluconazole, sulfonamid e, acyclovir, pyrimethamine, om eprazol, m etoclo- pramide, folic acid, and hydrolytic replacem ent. He was referred to the Department of Oral and Maxillofacial Surgery of the Mario Gatti Municipal Hospital for treatment of the oral candido- sis and the lesion of the hard palate. A clinical examination revealed an irregular ulcer with necrotic debris in the central region, an indurated border, m easuring 2.51.0 cm on the hard palate und erneath the ill-fitting denture, with slight symptomatology on palpation present for 1 week (Figure 1A). No cer- vical lymphadenopathy was noted. The initial clinical diagnosis was squa m ou s cell carcinoma. An incisional biopsy was performed and the material was sent to the Pathology Laboratory of S ã o Leopoldo Mandic Institute and Research Center, Cam pinas. The pseu d o m e m branous oral candido- sis was characterized by whitish plaque that could be rubbed off, and was located Figure 1. (A) Irregular ulcer with necrotic debris in central region, an indurated border measuring 2 2.5 1.0 cm. (B) Histopathological examination revealing a fragment of mucosa exhibiting pseu- in the oropharynx and bilateral buccal mucosa (Figure 1 E and 1F). It was associ- UI, 3 or 4 times daily for 15 days), which ated with burning symptom s and did not accomplish the expected results. dysphagia. The patient was prescribed a doepitheliomatous hyperplasia (*), squamou s metapla of salivary gland ducts (arrow), and Then it was necessary to use a systemic topical antifungal agent (Nystatin 100,00 0 coagulation necrosis of the acinis in the lower portion of the speci m e n (HE 40X). (C) Lobular coag- ulation necrosis of the acinis showing architectural preservation (HE 400X). (D) Squam ou s m etaplasia of the ducts (HE 400X). (E and F) Bilateral pseu d o m e m branous oral candidosis in the buccal muco sa, these white plaques could be rubbed off. therapy with intravenous fluconazole; plasia, lobular coagulation necrosis with Discussion 2.5 after 3 weeks, the pseud o m e m branou s palatal cm. In the literature, there are mucosa. Two-months after the the lesion was located in the minor sali- candidiasis had disappeared . treatment, the patient died due to com - vary glands of the hard palate. The biopsy speci m e n s of thed har plications from AIDS . The lesion was clinically character- palate revealed a fragment of mucos a ized as an irregular ulcer with necrotic exhibiting pseu d o e pitheliomatous hyper - debris in the central region, m easuring 2,5,8,17 preservation of the lobular architecture, NS is a rare benign, self-healing, reactive reports of patients who present swelling squamous metaplasia of ducts, and inflammatory process that most fre- without ulcerations, but the most typical chronic inflammatory infiltrate in or quently affects the minor salivary glands presentation is a deep ulcer, as we found. 1,5,7
dence suppor t ed a diagnosis S N of har d and soft pal at e is t he second mos t (1 men: 1 woman), whit es (4.9 whit es: .9(Figur e 1B, 1C, and .1D) common sit e of t his l esion, but ot her lor a 1 bl ack), and middl e- aged adul t s, as No f urt her sur ger y was perf or med . sit es may be invol ved, incl uding t h e ment ioned by sever al pr evious 1,6-8,12,17 3-5,9-11Silva et al. Spec Care Dentist 30(4) 2010 161around the glands. The microscopic evi- of the hard palate. The junction of the NS is more frequentlyfound in menThe lesion disappeared completely after lower lip, retromolar area, tongue, buccal reports. Thepatient reported2 weeks, with complete repair of the mucosa, and others. In our patient, here was a 50-year-old white man.
scd_142.qxd 6/25/10 7:16 AM Page 162 N E C R O T I Z I N G S I A L O M E TA P L A S I A I N A H I V P O S I T I V E PAT I E N T It has been suggested that the patho- early NS, suggesting a possible relationshi p reports. J Am Dent Asso 1996;127: 1087- c genesis of the lesion is a reactive proces s between the two conditions . 92. necrosis of the glands. This is the most healing time is from 3 to 6 weeks. In 8. Brannon RB, Fowler CB, Hartmanaccepted theory. widely KS. NS has been our patient, the lesion disappeared com- Necrotizing sialometaplasia: a clinicopatho- described in association with sev- pletely after 2 weeks and follow-up was logic study of sixty-nine cases and review of possibly related to trauma. The mai n Once the diagnosis of NS hasn bee 7. Grillon GL, Lally ET. Necrotizing sialometa- cause of the lesion is the loss ofe th mad e, close follow-up is recom m edn d e plasia: literature review and presentation the blood suppl leading to infarction an y, d until healing Malagon et al. demonstrated is complete. The an unusual association of NS with T-cell Conclusion 10. Chen average cavity. Hum Pathol 1977;8:589-92. f ive cases. J Oral KT. Necrotizing sialometaplasia of the Surg 1981;39:747-53 13,14 1,2,16 1,2,6,13-15 eral non-neoplastic conditions, such as the procedure of choice. There was no the literature. Oral Surg Oral Med Oral local trauma, upper respiratory tract recurrence before his death, which Pathol 1991;72:317-25. infection, induced vomiting in bulimia, occurred 2 months after treatment. 9. Johnston WH. Necrotizing sialometaplasia cocaine use, and radiation. Dominguez- involving the mucous glands of the nasal 18 lymphoma and reported that vascular It is important for both clinicians and nasal cavity. Am J Otolaryngol 1982;3:444-6. Scully and Eveson and Solomon who are HIV positive and occlusion by the neoplastic lymphoid immunocom- 1992;21:280-2. et al. have suggested that the pathogene- promised would be desirable in order to 14. Walker GK, Fechner RE, Johns ME, Teja K. cell pathologists to be aware of the pathogen- 11. Pulse CL, Lebovics RS, Zegarelli DJ. produces ischemia and chronic mechanical injury of contributes to the esis of this palatine References 15. Prabhakaran VC, Flora RS, Kendall disease, as well as its clinical Necro C. tizing sialometaplasia: report of a case development of this salivary gland lesion. and histopathological behavior during after lower lip mucocele excision. J Oral In our patient, the lesion could be related different stages of development. Maxillofac Surg 2000;58:1419-21. to local trauma of the hard palate caused Recognition of the histological spectrum 12. Forney SK, Foley JM, Sugg WE Jr, Oatis GW by the ill-fitting denture, or persistent and the varied clinical findings, in which Jr. Necrotizing sialometaplasia of the vomiting. It is interesting to speculate NS can be found, is essential to avoid mandible. Oral Surg Oral Med Oral Pathol that undue denture pressure might com- misinterpretation and inappropriate 1977;43:720-6. promise the palatal blood supply treatment for this benign lesion. 13. Romagosa V, Bella MR, Truchero C, Moya J. sufficiently to result in infarction of Studies on specific therapeutic regi- Necrotizing sialometaplasia (adenometapla- minor salivary glands. mens for the treatment of NS in patients sia) of the trachea. Histophatology 5 4 sis of NS is widely believed to be related establish a standard protocol. Necrotizing sialometaplasia of the larynx to ischemic changes because of vomiting secondary to atheromatous embolization. induced by bulimia, which could lead to Am J Clin Pathol 1982;77:221-3. mucosa. This theory can explain the 1. Abrams AM, Melrose RJ, Howell FV. Pressure-induced necrotizing sialometaplasia association of the lesion with frequent Necrotizing sialometaplasia. A disease simu- of the parotid gland. Histopathology vomiting episodes, as reported by our lating malignancy. Cancer 1973;32:130-5. 2006;48:464-5. patient. 2. Anneroth G, Hansen LS. Necrotizing 16. Rizkalla H, Toner M. Necrotizing sialometa- The diagnosis of NS can only be made sialometaplasia. The relationship of its plasia versus invasive carcinoma of the head after the biopsy and microscopic examina- pathogenesis to its clinical characteristics. and neck: the use of mioepithelial markers tion. Histologically, the biopsy from our Int J Oral Surg 1982;11:283-91. and keratin subtypes as an adjunct to diag- patient had all the features described in 3. Keogh PV, O’Regan E, Toner M, Flint S. nosis. Histopathology 2007;51:184-9. 1
The impor t ance of descr ibingst hi er al pr esent at ion associat ed wit h ant eceden t sial omet apl asia: r eport of five cases. Or all esion is t he fact t hat it coul d be mist aken anaest hesia and l ack of r esponse t o intral e - Sur g Or al Med Or al Pat ho 1974;37:722- 7. lf or mucoepider moid car cinoma or squa - sional st er oids. Case r eport and r eview of t he 18. Dominguez- Mal agon H, Mosqueda- Tayl or A,mous cel l car cinoma, because of t h e l it erat ur e. Br Dent J 2004;1 96:79-81 . Cano- Val dez AM. Necr ot izing sial omet apl a-cl inical simil arit ies of t hese l esions. e Th 4. Sol omon W, Mer zianu M, Sul l ivan M L , sia of t he pal at e associat ed wit h angiocent r icdist inct ion can onl y be made hist ol ogical l y, Rigual NR. Necr ot izing sial omet apl asia asso - T-cel l l ymphoma. Ann Diagn Pat holwit h empha sis on t he l obul ar mor phol og y, ciat ed wit h bul im case r eport d ia: an 2009;1 3:60-4.t he bl and appear ance of t he squa mou s l it erat ur e r eview. Or al Sur g Or al Med Or a l 19. Cohen D, Bhattachar yya I. Case of t heisl ands, and evidence of r esidual duct a l Pat hol Or al Radiol Endo 2007;1 d 03:e39-e42 . mont h. Necr ot izing sial omet apl asia. Todays 16,19 20ferential diagnosis of NS is subacute J Oral Maxillofac Surg 2004;33:808-10. sialadenitis: a clinicopathological study. Oralnecrotizing sialadenitis. This condition 6. Imbery TA, Edwards PA. Necrotizing Surg Oral Med Oral Pathol Oral Radiol Endodshares some of the histological features of sialometaplasia: literature review and case 2007;104:385-90.162 Spec Care Dentist 30(4) 2010 Ne crot i zi ng s ia l om e ta p la si a i n a HI V p osi t iv e p a ti e ntthe literature by Abrams et al. Necrotizing sialometaplasia: an unusual bilat- 17. DunlapCL, Barker BF. Necrotizingluminal in these islands. According to 5. Scully C, Eveson J. Sialosis andnecrotising FDA 2008;20:21-3.Suresh and Aguirre, another possible dif- sialometaplasia in bulimia; a case report. Int 20. Suresh L, Aguirre A. Subacutenecrotizing