- Email: [email protected]
Necrotising sialometaplasia presenting as greater palatine nerve anaesthesia
Necr0tising sial0metaplasia presenting as greater palatine nerve anaesthesia
P.-J. Lamey, M. A. O. Lewis, D. J. Crawford* and D. G. M a c D o n a l d Departments of Oral Medicine and Pathology, and *Oral Surgery, Glasgow Dental Hospital and School, 378 Sauchiehall Street, Glasgow, G2 3JZ, UK
P.-J. Larney, M. A. O. Lewis, D. J. Crawford and D. G. MacDonald." Neerotising sialometaplasia presenting as greater palatine nerve anaesthesia. Int. J. Oral Maxillofac. Surg. 1989; 18: 70-72. Abstract. The spontaneous presentation of oro-faciat paraesthesia or anaesthesia is relatively uncommon. However, clinicians m a y be required to investigate and c o m m e n t on the m a n a g e m e n t of patients with such symptoms. "We report 3 cases of necrotising sialometaplasia which presented with intra-oral anaesthesia.
Necrotising sialometaplasia is an unc o m m o n self-limiting condition, predominantly involving the m i n o r salivary glands first described by ABRAMS et al. 1 in a case affecting the palate. The condition is now known to'affect a number of other sites including the nasopha, rynx 2. The aetiology of the condition is obscure, but it is thought to involve infarction of mucus secreting glands 3. It occurs typically in the adult male, the c o m m o n site involved being the posterior palatal mucosa 4, 7. Occasionally, other sites such' as the nasal mucosa 1°,11,13,17 and the m a j o r salivary glands 6 may be involved. T h e lesion typically presents as swelling or ulceration, with pain being a variable s y m p t o m 3. Accompanying disturbance of sensation has been reported in s o m e cases t3. Clinical and histological diagnosis m a y be difficult, and in the past, s o m e cases have been wrongly considered neoplastic, resulting in inappropriate surgical treatment ~s. A n unusual feature of the cases reported here was anaesthesia of the greater palatine nerve, which preceded the appearance of the typical swelling and ulceration of necrotising sialometaplasia. Case reports Case no. 1
A 35-year-old Caucasian male was referred to the Oral Medicine Unit, Glasgow Dental Hospital, by his general dental practitioner for investigation of a 5-day history of anaesthesia of the left hard palate. Latterly, the right hard palate had also become affected. In addition, the patient complained of nau-
sea, sore throat and a headache affecting both temple regions. Approximately 1 week prior to referral, the patient's medical practitioner had prescribed tetracycline (250 rag, qid) for the sore throat, There were no other relev~mt features in the patient's medical history and he had not recently received any dental treatment. Clinically, there was some tenderness over the distribution of the superficial temporal arteries, but nothing else of note extra-orally. Intra-orally, the oral mueosa appeared moist and healthy, although some erythema of the oro-pharynx was noted. The distribution of anaesthesia involved the area innervated by both greater palatine nerves but spared the soft palate and the area o f the anterior hard palate supplied by the nerve of the incisive canal. Investigations undertaken involved a viral antibody screen, fttll blood count and an erythrocyte sedimentation rate (ESR). The viral screen showed no significant titres to herpes simplex, herpes zoster or cytomegalovirus. Blood film, blood pressure and ESR (Westergren method) were normal. At review after 3 days, the headache, nausea and pain in the temple regions had completely resolved. Sensation was returning to the hard palate, but bilateral swellings had developed in this area at the junction with the soft palate (Fig. 1). These swellings were painless and the patient thought that they began to develop the night of the previous appointment. An incisional biopsy of the right-sided lesion was reported as showing features of necrotising sialometaplasia. Complete resolution occurred within 2 weeks with an accompanying return of normal sensation of palatal tissues. Case no. 2
A 29-year-old Caucasian male was referred to the Department of Oral Surgery by his dental practitioner with a 5-day history of
Key words: necrotising sialometaplasia; salivary glands. Accepted for publication 2 November 1988
palatal ulceration which had been preceded by unilateral anaesthesia over the distribution of the right greater palatine nerve. The patient, a well-controlled insulin-dependent diabetic, had initially sought treatment from his medical practitioner who prescribed amoxycillin (250 mg, qid) for a suspected upper respiratory infection. 24 h after the appearance of the anaesthesia, an intensely painful swelling developed in the palate which ulcerated after a further 24 h. The onset of the ulceration was associated with complete pain relief. Clinical examination revealed an elongated irregular ulcer (2 cm diameter) to the right of the hard palate in the region of the second molar tooth. The mucosa innervated by the right greater palatine nerve was found to be totally unresponsive to the insertion of a straight probe. Radiographs of the area failed to reveal any dental or bony pathology and the teeth in the upper right quadrant gave a vital response to an electric pulp-tester. Histopathological examination of incisional biopsy confirmed the clinical diagnosis of necrotising sialometaplasia (Fig. 2). Little haemorrhage was encountered during the biopsy procedure. Healing occurred within 4 weeks and was associated with return of normal sensation. Case no. 3 A 43-year-old Caucasian female was referred
to the Department of Oral Surgery by her general dental practitioner with a painless palatal ulcer which had been present for 1 week and was associated with bilateral greater and lesser palatine nerve anaesthesia. Past medical history revealed a hysterectomy when aged 27 years and present medication consisted of xipamide and chlordiazepoxide. The patient smoked between 20 and 40 cigarettes per day and was a social drinker of alcoholic beverages. At the time
Necrotising sialometaplasia
71
ited to 0.2% aqueous chlorhexidine gluconate mouthwash. Complete healing occurred in 3 weeks although there was some residual scarring at the ulcer site.
Discussion
Fig. 1. Bilateral soft tissue swelling in posterior hard palate, case no. 1.
of examination, she complained of an upper respiratory tract infection of several clays duration. Intra-oral examination revealed an irregular area of ulceration (2 cm diameter) in the midline of the junction of the hard and soft palate. In addition to the hard palate, the
area of soft palate accessible to examination was found to be completely anaesthetic to a sharp probe. A provisional diagnosis of necrotising siatometaplasia was made from the clinical history and the appearance of the lesion. The patient declined biopsy and tratment was lim-
These 3 cases are unusual in that loss of normal mucosal sensation was a major presenting feature; indeed, in 2 of these cases, the anaesthesia preceded the onset o f the ulceration. They are also linked by the presence of suspected preexisting respiratory tract infection although it m a y be that the intra-oral discomfort prior to the onset o f the mucosal anaesthesia was mistakenly diagnosed. Each case demonstrates the rapid onset and slow healing of the oral ulceration characteristic o f necrotising sialometaplasia 19. The relationship between anaesthesia and the disease process can only be speculated upon, but it is possible that the process o f infarction of minor salivary glands is associated with reversible nerve damage due to an ischaemic effect in the same area. A n y complaint of altered oral sensation warrants further investigation TM, since it has been reported as a feature of leukaemia 5, 8, systemic malignant diseasel4, l y m p h o m a 5, 16, sickle cell crisis 12, and the myelodysplastic syndrome 9. The cases reported here would support the addition of necrotising sialometaplasia to this list o f conditions.
References
1
Fig. 2. Palatal biopsy of case no. 2 showing ulceration of surface epithelium and underlying salivary gland tissue with obvious squamous metaplasia of ducts (H & E, x 33).
1. Abrams, A. M., Melrose, R. J. & Howell, R. V.: Necrotising sialometaplasia. Cancer 1973: 32: 130-135. 2. Anneroth, G. & Hansen, L. S.: Necrotising sialometaplasia. The relationship of its pathogenesis to its clinical cliaracteristics. Int. J. Oral Surg. 1982: 11: 283-91. 3. Arguelles, M. T., Viloria, J. B., Tallens, M. C. & McCrory, T. R: Necrotising sialometaplasia. Oral Surg. 1978: 42: 86-90. 4. Bannayan, G., Fox, G. & Tilson, H. B.: Necrotising sialometaplasia of the palate. J. Oral Surg. 1976: 34: 727-730. 5. Barrett, A. R: Selective anaesthesias of the inferior aveolar nerve in leukaemia and lymphoma. J. Oral MaxilloJae. Surg. 1985: 43: 992-994. ' 6. Bhargava, S. & Monga, J. N.: Necrotising sialometaplasia of the parotid. Indian J. Cancer 1975: 12: 9%102. 7. Chaudry, A. R, Yamane, G. M., Salman, L., Salman, S., Saxon, M. & Pierri, L. K.: Necrotising sialometaplasia of the palatal minor salivary glands. A report of two cases. J. Oral Med. 1985: 40:2 6. 8. Dawson, D. M., Rosenthal, D. S. & Moloney, W. C.: Neurological complications
72
L a m e y et al.
of acute leukaemia in adults: changing rate. Ann. Intern. Med. 1973: 79: 541-544. 9. Gibson, J., Lamey, E-J., Watson, W. H. & Tansey, E: The myelodysplastic syndrome presenting with oral symptoms. Br. Dent. J. 1987: 163: 234-235. 10. Grillon, G. L. & Lalley, E. T.: Necrotising sialometaplasia: Literature review and presentation of five cases. J. Oral Surg. 1981: 39: 747-753. 1l. Johnston, W. H.: Necrotising sialometaplasia involving the mucous glands of the nasal cavity. Hum Pathol 1977: 8: 589-592. 12. Kirson, L. E. & Tomato, A. J.: Mental nerve paraesthesia secondary to sickle cell crisis. Oral Surg. 1979: 48: 509-512.
13. Maisel, R. H., Johnston, W. H., Anderson, H. A. & Cantrell, R. W.: Necrotising sialometaplasia involving the nasal cavity. Report of two cases. Laryngoscope 1977: 87: 429-434. 14. Massey, E. W., Moore, J. & Schoid, S. C.: Mental neuropathy from systemic cancer. Neurology (NY) 1981: 31: 1277-1281. 15. Mesa, M. L., Gertler, R. S. & Schneider, L. C.: Necrotising sialometaplasia: frequency of histologic misdiagnosis. Oral Surg. 1984: 75: 71-73. 16. Nubter, M. E: Mental nerve palsy in malignant lymphoma. Cancer 1969: 24: 122-127. 17. Papanaytou, E M., Kayavis, J. G., Epivationos, A. A. & Trigonidis, G.: Necroti-
sing sialometaplasia of the cheek: report of a case and review of the literature. J. Oral Surg. 1980: 38: 538-540. 18. Ruistacher, S. L.: Numbness - a significant finding. Oral Surg. 1973: 36: 22-27. 19. Suckiel, E H., Davis, W. H., Patakas, B. M. & Kaminishi, R. M.: Early and late manifestations of necrotising sialometaplasia. J. Oral Surg. 1978: 36: 902-905. Address: P.-J. Lamey Senior Lecturer in Oral Medicine Glasgow Dental Hospital and School 378 Sauchiehall Street Glasgow, G2 3JZ Scotland UK
P.-J. Lamey, M. A. O. Lewis, D. J. Crawford* and D. G. M a c D o n a l d Departments of Oral Medicine and Pathology, and *Oral Surgery, Glasgow Dental Hospital and School, 378 Sauchiehall Street, Glasgow, G2 3JZ, UK
P.-J. Larney, M. A. O. Lewis, D. J. Crawford and D. G. MacDonald." Neerotising sialometaplasia presenting as greater palatine nerve anaesthesia. Int. J. Oral Maxillofac. Surg. 1989; 18: 70-72. Abstract. The spontaneous presentation of oro-faciat paraesthesia or anaesthesia is relatively uncommon. However, clinicians m a y be required to investigate and c o m m e n t on the m a n a g e m e n t of patients with such symptoms. "We report 3 cases of necrotising sialometaplasia which presented with intra-oral anaesthesia.
Necrotising sialometaplasia is an unc o m m o n self-limiting condition, predominantly involving the m i n o r salivary glands first described by ABRAMS et al. 1 in a case affecting the palate. The condition is now known to'affect a number of other sites including the nasopha, rynx 2. The aetiology of the condition is obscure, but it is thought to involve infarction of mucus secreting glands 3. It occurs typically in the adult male, the c o m m o n site involved being the posterior palatal mucosa 4, 7. Occasionally, other sites such' as the nasal mucosa 1°,11,13,17 and the m a j o r salivary glands 6 may be involved. T h e lesion typically presents as swelling or ulceration, with pain being a variable s y m p t o m 3. Accompanying disturbance of sensation has been reported in s o m e cases t3. Clinical and histological diagnosis m a y be difficult, and in the past, s o m e cases have been wrongly considered neoplastic, resulting in inappropriate surgical treatment ~s. A n unusual feature of the cases reported here was anaesthesia of the greater palatine nerve, which preceded the appearance of the typical swelling and ulceration of necrotising sialometaplasia. Case reports Case no. 1
A 35-year-old Caucasian male was referred to the Oral Medicine Unit, Glasgow Dental Hospital, by his general dental practitioner for investigation of a 5-day history of anaesthesia of the left hard palate. Latterly, the right hard palate had also become affected. In addition, the patient complained of nau-
sea, sore throat and a headache affecting both temple regions. Approximately 1 week prior to referral, the patient's medical practitioner had prescribed tetracycline (250 rag, qid) for the sore throat, There were no other relev~mt features in the patient's medical history and he had not recently received any dental treatment. Clinically, there was some tenderness over the distribution of the superficial temporal arteries, but nothing else of note extra-orally. Intra-orally, the oral mueosa appeared moist and healthy, although some erythema of the oro-pharynx was noted. The distribution of anaesthesia involved the area innervated by both greater palatine nerves but spared the soft palate and the area o f the anterior hard palate supplied by the nerve of the incisive canal. Investigations undertaken involved a viral antibody screen, fttll blood count and an erythrocyte sedimentation rate (ESR). The viral screen showed no significant titres to herpes simplex, herpes zoster or cytomegalovirus. Blood film, blood pressure and ESR (Westergren method) were normal. At review after 3 days, the headache, nausea and pain in the temple regions had completely resolved. Sensation was returning to the hard palate, but bilateral swellings had developed in this area at the junction with the soft palate (Fig. 1). These swellings were painless and the patient thought that they began to develop the night of the previous appointment. An incisional biopsy of the right-sided lesion was reported as showing features of necrotising sialometaplasia. Complete resolution occurred within 2 weeks with an accompanying return of normal sensation of palatal tissues. Case no. 2
A 29-year-old Caucasian male was referred to the Department of Oral Surgery by his dental practitioner with a 5-day history of
Key words: necrotising sialometaplasia; salivary glands. Accepted for publication 2 November 1988
palatal ulceration which had been preceded by unilateral anaesthesia over the distribution of the right greater palatine nerve. The patient, a well-controlled insulin-dependent diabetic, had initially sought treatment from his medical practitioner who prescribed amoxycillin (250 mg, qid) for a suspected upper respiratory infection. 24 h after the appearance of the anaesthesia, an intensely painful swelling developed in the palate which ulcerated after a further 24 h. The onset of the ulceration was associated with complete pain relief. Clinical examination revealed an elongated irregular ulcer (2 cm diameter) to the right of the hard palate in the region of the second molar tooth. The mucosa innervated by the right greater palatine nerve was found to be totally unresponsive to the insertion of a straight probe. Radiographs of the area failed to reveal any dental or bony pathology and the teeth in the upper right quadrant gave a vital response to an electric pulp-tester. Histopathological examination of incisional biopsy confirmed the clinical diagnosis of necrotising sialometaplasia (Fig. 2). Little haemorrhage was encountered during the biopsy procedure. Healing occurred within 4 weeks and was associated with return of normal sensation. Case no. 3 A 43-year-old Caucasian female was referred
to the Department of Oral Surgery by her general dental practitioner with a painless palatal ulcer which had been present for 1 week and was associated with bilateral greater and lesser palatine nerve anaesthesia. Past medical history revealed a hysterectomy when aged 27 years and present medication consisted of xipamide and chlordiazepoxide. The patient smoked between 20 and 40 cigarettes per day and was a social drinker of alcoholic beverages. At the time
Necrotising sialometaplasia
71
ited to 0.2% aqueous chlorhexidine gluconate mouthwash. Complete healing occurred in 3 weeks although there was some residual scarring at the ulcer site.
Discussion
Fig. 1. Bilateral soft tissue swelling in posterior hard palate, case no. 1.
of examination, she complained of an upper respiratory tract infection of several clays duration. Intra-oral examination revealed an irregular area of ulceration (2 cm diameter) in the midline of the junction of the hard and soft palate. In addition to the hard palate, the
area of soft palate accessible to examination was found to be completely anaesthetic to a sharp probe. A provisional diagnosis of necrotising siatometaplasia was made from the clinical history and the appearance of the lesion. The patient declined biopsy and tratment was lim-
These 3 cases are unusual in that loss of normal mucosal sensation was a major presenting feature; indeed, in 2 of these cases, the anaesthesia preceded the onset o f the ulceration. They are also linked by the presence of suspected preexisting respiratory tract infection although it m a y be that the intra-oral discomfort prior to the onset o f the mucosal anaesthesia was mistakenly diagnosed. Each case demonstrates the rapid onset and slow healing of the oral ulceration characteristic o f necrotising sialometaplasia 19. The relationship between anaesthesia and the disease process can only be speculated upon, but it is possible that the process o f infarction of minor salivary glands is associated with reversible nerve damage due to an ischaemic effect in the same area. A n y complaint of altered oral sensation warrants further investigation TM, since it has been reported as a feature of leukaemia 5, 8, systemic malignant diseasel4, l y m p h o m a 5, 16, sickle cell crisis 12, and the myelodysplastic syndrome 9. The cases reported here would support the addition of necrotising sialometaplasia to this list o f conditions.
References
1
Fig. 2. Palatal biopsy of case no. 2 showing ulceration of surface epithelium and underlying salivary gland tissue with obvious squamous metaplasia of ducts (H & E, x 33).
1. Abrams, A. M., Melrose, R. J. & Howell, R. V.: Necrotising sialometaplasia. Cancer 1973: 32: 130-135. 2. Anneroth, G. & Hansen, L. S.: Necrotising sialometaplasia. The relationship of its pathogenesis to its clinical cliaracteristics. Int. J. Oral Surg. 1982: 11: 283-91. 3. Arguelles, M. T., Viloria, J. B., Tallens, M. C. & McCrory, T. R: Necrotising sialometaplasia. Oral Surg. 1978: 42: 86-90. 4. Bannayan, G., Fox, G. & Tilson, H. B.: Necrotising sialometaplasia of the palate. J. Oral Surg. 1976: 34: 727-730. 5. Barrett, A. R: Selective anaesthesias of the inferior aveolar nerve in leukaemia and lymphoma. J. Oral MaxilloJae. Surg. 1985: 43: 992-994. ' 6. Bhargava, S. & Monga, J. N.: Necrotising sialometaplasia of the parotid. Indian J. Cancer 1975: 12: 9%102. 7. Chaudry, A. R, Yamane, G. M., Salman, L., Salman, S., Saxon, M. & Pierri, L. K.: Necrotising sialometaplasia of the palatal minor salivary glands. A report of two cases. J. Oral Med. 1985: 40:2 6. 8. Dawson, D. M., Rosenthal, D. S. & Moloney, W. C.: Neurological complications
72
L a m e y et al.
of acute leukaemia in adults: changing rate. Ann. Intern. Med. 1973: 79: 541-544. 9. Gibson, J., Lamey, E-J., Watson, W. H. & Tansey, E: The myelodysplastic syndrome presenting with oral symptoms. Br. Dent. J. 1987: 163: 234-235. 10. Grillon, G. L. & Lalley, E. T.: Necrotising sialometaplasia: Literature review and presentation of five cases. J. Oral Surg. 1981: 39: 747-753. 1l. Johnston, W. H.: Necrotising sialometaplasia involving the mucous glands of the nasal cavity. Hum Pathol 1977: 8: 589-592. 12. Kirson, L. E. & Tomato, A. J.: Mental nerve paraesthesia secondary to sickle cell crisis. Oral Surg. 1979: 48: 509-512.
13. Maisel, R. H., Johnston, W. H., Anderson, H. A. & Cantrell, R. W.: Necrotising sialometaplasia involving the nasal cavity. Report of two cases. Laryngoscope 1977: 87: 429-434. 14. Massey, E. W., Moore, J. & Schoid, S. C.: Mental neuropathy from systemic cancer. Neurology (NY) 1981: 31: 1277-1281. 15. Mesa, M. L., Gertler, R. S. & Schneider, L. C.: Necrotising sialometaplasia: frequency of histologic misdiagnosis. Oral Surg. 1984: 75: 71-73. 16. Nubter, M. E: Mental nerve palsy in malignant lymphoma. Cancer 1969: 24: 122-127. 17. Papanaytou, E M., Kayavis, J. G., Epivationos, A. A. & Trigonidis, G.: Necroti-
sing sialometaplasia of the cheek: report of a case and review of the literature. J. Oral Surg. 1980: 38: 538-540. 18. Ruistacher, S. L.: Numbness - a significant finding. Oral Surg. 1973: 36: 22-27. 19. Suckiel, E H., Davis, W. H., Patakas, B. M. & Kaminishi, R. M.: Early and late manifestations of necrotising sialometaplasia. J. Oral Surg. 1978: 36: 902-905. Address: P.-J. Lamey Senior Lecturer in Oral Medicine Glasgow Dental Hospital and School 378 Sauchiehall Street Glasgow, G2 3JZ Scotland UK