burning mouth sensation associated with fusospirochetal infection in edentulous patients

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Burning mouth sensation associated with fusospirochetal infection in edentulous patients Joseph Katz, D.M.D.,* Raphael Benoliel, B.D.S., L.D.S., R.C.S. (Eng.),* and Ephraim Leviner, D.M.D.,** Jerusalem, Israel DEPARTMENTS OF ORAL DIAGNOSIS, ORAL MEDICINE, ORAL RADIOLOGY HEBRCW UNIVERSITY-HADASSAH FACULTY OF DENTAL MEDICINE Six middle-aged and elderly edentulous, systemically ill patients complaining of burning mouth sensation had a fusospirochetal infection of the oral mucosa. Metronidazole was successful in the treatment of three of these patients. (ORAL SURG. ORAL MED. ORAL PATHOL. 62~152-154, 1986) B urning mouth sensation (BMS) is a common disorder in which the patient experiences a constant intraoral pain and irritation of mucosal origin. Although it primarily affects postmenopausal women,’ uncertainty exists as to its cause. In their reviews, Domb and Chole2 and Zegarelli3 outlined the various causes of BMS. They implicated local factors, such as candidal infection, lichen planus, geographic tongue, oral carcinoma, and a variety of irritants that may cause microtrauma to the oral mucosa. The same authors also linked BMS with systemic factors, including diabetes; vitamin B12, iron, or folate deficiency; postmenopausal hypoestro- genism; drug therapy; and lingual artery atheroscle- rosis. Rheumatoid arthritis is also thought to be involved in causing BMS.4 Main and Baske$ report- ed denture faults as the single most tangible cause of BMS in 50% of their cases. In many patients with BMS, however, none of the above conditions is present, and in these instances the disorder may be secondary to a psychologic disturbance3 or is ulti- mately diagnosed as idiopathic.2 Spirochetes and fusiform bacteria usually inhabit the oral cavities of adults with normal dentitions.6 The organisms have been suggestedin the pathogen- esis of oral diseases,among them acute necrotizing ulcerative gingivitis (ANUG),’ dry socket,* and pericoronitis.9 In all of these, there is always accom- *Resident. **Lecturer. panying intraoral pain or irritation.’ However, the presence of fusospirochetes in edentulous adults or in infants prior to tooth eruption is rare.6 Indeed, denture plaque from edentulous patients differs from subgingival plaque of dentulous patients by the absence of spirochetes.” The current case reports describe a possible correlation between BMS and fusospirochetal infection. PATIENTS AND FINDINGS Six edentulous patients (50 to 70 years of age) had complaints of constant burning sensations in the mouth, particularly the tongue, for which they had been unsuccessfully treated in the past with multivi- tamins and minor tranquilizers (medazepam hydro- chloride). The onset of the symptom, which had not been related to any specific event, occurred 4 to 7 months (average, 5 months) before presentation. The burning sensation did not vary in intensity; nor were there any alleviating or aggravating factors. The medical history revealed that the patients suffered from various chronic illnesses (Table I). Intraoral examination revealed that the tongues were coated with an off-white layer, which rubbed off quite easily, leaving an erythematous but non- bleeding area. There were also multiple erosions on the unattached mucosa. Examination of the patients’ dentures for retention, stability, and occlusion showed them to be satisfactory. Direct smears were taken from each tongue and placed on four glass slides; the material was stained 152

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Burning mouth sensation associated with fusospirochetal infection in edentulous patients Joseph Katz, D.M.D.,* Raphael Benoliel, B.D.S., L.D.S., R.C.S. (Eng.),* and Ephraim Leviner, D.M.D.,** Jerusalem, Israel

DEPARTMENTS OF ORAL DIAGNOSIS, ORAL MEDICINE, ORAL RADIOLOGY HEBRCW UNIVERSITY-HADASSAH FACULTY OF DENTAL MEDICINE

Six middle-aged and elderly edentulous, systemically ill patients complaining of burning mouth sensation had a fusospirochetal infection of the oral mucosa. Metronidazole was successful in the treatment of three of these patients. (ORAL SURG. ORAL MED. ORAL PATHOL. 62~152-154, 1986)

B urning mouth sensation (BMS) is a common disorder in which the patient experiences a constant intraoral pain and irritation of mucosal origin. Although it primarily affects postmenopausal women,’ uncertainty exists as to its cause. In their reviews, Domb and Chole2 and Zegarelli3 outlined the various causes of BMS. They implicated local factors, such as candidal infection, lichen planus, geographic tongue, oral carcinoma, and a variety of irritants that may cause microtrauma to the oral mucosa. The same authors also linked BMS with systemic factors, including diabetes; vitamin B12, iron, or folate deficiency; postmenopausal hypoestro- genism; drug therapy; and lingual artery atheroscle- rosis. Rheumatoid arthritis is also thought to be involved in causing BMS.4 Main and Baske$ report- ed denture faults as the single most tangible cause of BMS in 50% of their cases. In many patients with BMS, however, none of the above conditions is present, and in these instances the disorder may be secondary to a psychologic disturbance3 or is ulti- mately diagnosed as idiopathic.2

Spirochetes and fusiform bacteria usually inhabit the oral cavities of adults with normal dentitions.6 The organisms have been suggested in the pathogen- esis of oral diseases, among them acute necrotizing ulcerative gingivitis (ANUG),’ dry socket,* and pericoronitis.9 In all of these, there is always accom-

*Resident. **Lecturer.

panying intraoral pain or irritation.’ However, the presence of fusospirochetes in edentulous adults or in infants prior to tooth eruption is rare.6 Indeed, denture plaque from edentulous patients differs from subgingival plaque of dentulous patients by the absence of spirochetes.” The current case reports describe a possible correlation between BMS and fusospirochetal infection.

PATIENTS AND FINDINGS

Six edentulous patients (50 to 70 years of age) had complaints of constant burning sensations in the mouth, particularly the tongue, for which they had been unsuccessfully treated in the past with multivi- tamins and minor tranquilizers (medazepam hydro- chloride). The onset of the symptom, which had not been related to any specific event, occurred 4 to 7 months (average, 5 months) before presentation. The burning sensation did not vary in intensity; nor were there any alleviating or aggravating factors. The medical history revealed that the patients suffered from various chronic illnesses (Table I).

Intraoral examination revealed that the tongues were coated with an off-white layer, which rubbed off quite easily, leaving an erythematous but non- bleeding area. There were also multiple erosions on the unattached mucosa. Examination of the patients’ dentures for retention, stability, and occlusion showed them to be satisfactory.

Direct smears were taken from each tongue and placed on four glass slides; the material was stained

152

Volume 62 Number 2

Burning mouth sensation associated with fusospirochetal infection 153

Table I. Data of the patients witn ournmg mouth sensation

I 51/F Hypertension, mental Burning mouth, bad Tetracycline mouthwash Partial improvement

depression taste, halitosis

2 50/F Lupus erythematosus Burning mouth, oral Tctracyclinc mouthwash Partial improvement

discoides ulceration

3 74/M Glomerulonephritis Burning mouth, dry Tetracycline mouthwash Partial improvement

mouth, orolingual pain

4 60/F Chronic active Burning mouth, dry Tetracycline mouthwash, Partial improvement,

hepatitis, mouth. orolingual pain systemic mctronidarolc followed by resolution

rheumatoid arthritis

5 78/M Malignant lymphoma Burning mouth, dry Tetracycline mouthwash, Partial improvcmcnt. mouth systemic metronidazole followed by resolution

6 64/M Diabetes mellitus Burning mouth, smoky Tetracycline mouthwash, Partial improvement, taste, oral ulceration systemic metronidazole followed by resolution

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with Giemsa dye and examined under the light microscope. Spirochetes and fusiform bacteria con- stituted the predominant picture. With this finding, the patients were given prescriptions for tetracycline mouthwashes (250 mg/5 ml water, four times daily for 1 week). A decrease in the severity of the burning sensation was reported by all the patients following this regimen. In three patients, further treatment with metronidazole (250 mg, three times daily for 1 week) was instituted in an attempt to totally elimi- nate the symptoms, and it did result in a total remission. The subsequent clinical examination revealed normal mucous membranes, and smears revealed no fusospirochetes. At reexamination, 2 and 4 weeks later, these three patients reported no recurrence of symptoms and had intact mucous membranes. Recent recall (1 year after treatment) revealed no symptoms or signs.

DISCUSSION

The rationale behind this article was the singular finding of a fusospirochetal infection in the mouths of edentulous patients suffering from BMS. More noteworthy is the fact that treatment with metroni- dazole resulted in resolution of the BMS, and smears taken after this treatment showed disappearance of the fusospirochetes, thus establishing a direct con- nection between BMS and these organisms in such cases. To the best of our knowledge, such a relation- ship has not been reported previously.

It is probable that infection with fusospirochetes causes a local inflammation which, in turn, evokes a burning sensation. The constant association between oral fusospirochetal infection and pain raises the possibility that these organisms produce an enzyme or cascade a series of events producing a pain-

initiating factor, a supposition that has been investi- gated in dry socket.”

The apparent failure of the tetracycline mouth- wash may be ascribed to the short contact between this bacteriostatic drug and the affected areas, whereas systemically administered (per orally) met- ronidazole proved efficient. Metronidazole is a high- ly specific bactericidal drug for most anaerobic bacteria.‘*, I3 Concentrations obtained in saliva are as high as those in serum and may be more prolonged,14 making it a useful antibiotic for oral infections. Side effects include mild gastrointestinal symptoms, occa- sional but reversible neutropenia, and a temporary peripheral neuropathy on high-dose, long-term ther- apy.‘* A disulfiram effect with alcohol can be expect- ed in patients taking metronidazole, which may be a weak mutagen and, under certain circumstances, weakly tumorigenic and carcinogenic.‘* Even though these and clinical side effects are minimized on low-dose, short-term therapy (as was used in our patients), metronidazole should not be administered during pregnancy.

The questions remain as to why fusospirochetes were present in the mouths of edentulous systemical- ly ill persons, and what is the clinical significance of this finding. Further double-blind controlled trials are necessary to answer these questions.

CONCLUSIONS

The unusual presence of fusospirochetes in the mouths of six edentulous patients complaining of BMS is discussed. In three of these patients a direct relationship between BMS and fusospirochetal infec- tion is demonstrated. We recommend that smears be taken routinely to search for these organisms in patients suffering from BMS.

154 Katz, Benoliel, and Leviner

REFERENCES

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Brooke RI, Segansky DP: Aetiology and investigation of sore mouth. Can Dent Assoc J 10: 504-506, 1977. Domb GH, Chole RA: The burning mouth and tongue. Ear Nose Throat J 60: 310-314, 1981. Zegarelli DJ: Burning mouth: An analysis of 57 patients. ORAL SURC ORAL MED ORAL PATHOL 58: 34-38, 1984. Fcrguson MM, Carter J, Boyle P, Hart DM, Linsay R: Oral complaints related to climacteric symptoms in oophorectom- ized women. J R Sot Med 74: 492-498, 198 I. Main DMG, Basker RM: Patients complaining of burning mouth. Br Dent J 154: 206-212. 1983. Burnert G, Scherp BS. Schuster G: Oral microbiology and infectious diseases. Baltimore. 1978, Williams & Wilkins Company, p. 238. Hampp EC, Mergcnhagen SE: Experimental intracutaneous fusobacterial and fusospirochetal infections. J Infect Dis 122: 84-99, 1963. Nitzan DW: On the genesis of “dry socket.” J Oral Maxillo- Tat Surg 41: 706-710, 1983. Nitzan DW, Tal 0, Sela MN, Shteyer A: Pericornitis: A reappraisal of its clinical and microbiological aspects. J Oral Maxillofac Surg 43: 510-516, 1985.

Oral Surg. August, 1986

IO. Thcilade E, Jorgensen B, Theilade J: Predominant cultivable microflora of plaque on removable dentures in patients with healthy oral n&&a. Arch Oral Biol 28: 675-680. 19X3.

I I. Birn H: Kinines and pain in dry socket. Int J Oral Surg 1: 34-42, 1972.

12. Finegold SM: Metronidazole: Proceedings of the Internation- al Metronida7ole Conference, Montreal, Quebec. Canada, May 26-28. 1977, Princeton, N.J. 1977. Excerpta Medica, pp. 3-l I. 112-144.

13. Goodman LS, Gilman A: The pharmacological basis of therapeutics, ed. 6, New York, 1980, Macmillan Publishing Company, pp. 10751077.

14. vonKonow L, Nord CE: Concentrations of tinidazole and mctronidarole in serum saliva and alveolar bone. J ,4ntimi- crab Chemother 10 (Suppl. A): I65- 172, 1982.

Kepint requests to: Dr. Joseph Katl- Dcpartmcnts of Oral Diagnosis, Oral Medicine, Oral Radiology Hebrew University-Hadassah Faculty of Dental Medicine P.O. Box II72 Jcrusnlem. 91010 Israel

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Bound volumes of ORAL SURGERY, ORAL MEDICINE and ORAL PATHOLOGY are available to subscribers (only) for the 1986 issues from the Publisher, at a cost of $38.00 ($50.00 international) for Vol. 61 (January-June) and Vol. 62 (July-December). Shipping charges are included. Each bound volume contains a subject and author index and all advertising is removed. Copies are shipped within 60 days after publication of the last issue in the volume. The binding is durable buckram with the journal name, volume number, and year stamped in gold on the spine. Payment must accompany all orders. Contact The C. V. Mosby Company, Circulation Department, II830 Westline Industrial Drive, St. Louis, Missouri 63 146, USA; phone (800) 325-4 177, ext. 35 I. Subscriptions must be in force to qualify. Bound volumes are not available in place of a regular journal subscription.