Carrie L. Gick, BA,a Ginat W. Mirowski, DMD, MD,a,b John S. Kennedy, MD,c A 73-year-old white male with a 6-month history of glossodynia, unresponsive to clotrimazole troches,cevimeline, triamcinolone dental paste, paroxetine, and lorazepam presented to the dermatology clinic forconsultation. Work-up revealed no oral abnormalities and no underlying systemic disorder. He deniedsymptoms consistent with a psychiatric disorder. A detailed free amnestic assessment by a board certifiedGeriatric Psychiatrist (John S. Kennedy, MD) found that the patient was oppressed by the pain. He did notmeet the criteria for major depression nor did he have any anxiety disorder or delusions. Because of thepresence of dysphoria and anticipatory anxiety secondary to glossodynia, the patient was started onolanzapine. Improvement of pain symptoms were noted within 3 days with full resolution of symptoms at1- and 3-month follow-ups. Dysphoria and anticipatory anxiety remitted fully upon pain relief. (J Am AcadDermatol 2004;51:463-5.) unresponsive to clotrimazole troches, cevimeline, A 73-year-old white male presented with a triamcinolone dental paste, paroxetine, and lora- 6-month history of ‘‘painful burning tongue and zepam. He denied tobacco, alcohol, or other drug bitter taste’’ following acute trauma from eating hot soup. The patient also complained of thick On physical examination, the patient appeared saliva, cotton mouth, and a red tender tongue with physically well. Oral mucosa and conjunctiva were eating. The tenderness and taste perversion re- pink and moist. The tongue appeared normal both sulted in a 20-pound weight loss. The patient before and after eating. The mandible was edentu- denied cancer phobia. Past medical history was lous with a lower complete denture and the maxilla significant for non-Hodgkin’s lymphoma treated was partially edentulous without a partial denture.
with chemotherapy 5 years previously and no There was no angular cheilitis or lymphadenopathy.
evidence of recurrence. He noted that during He was dysphoric and had anticipatory anxiety just chemotherapy he had experienced similar tongue prior to eating each morning. He did not report symptoms for a brief period, but they had remitted symptoms consistent with a depression syndrome.
following completion of treatment. He reported no He expressed a wish not to continue living in his personal or family history of psychiatric disorder.
current state, but denied any active plans of suicide.
His only medication prior to symptom onset was A detailed free amnestic assessment by a board terazosin, which he had taken for 5 years. He was certified Geriatric Psychiatrist (J. S. K.) found thatthe patient was oppressed by the pain. The patientdid not meet the criteria for major depression. He did From the Departments of Dermatologya and Psychiatry and not have any anxiety disorder and he had no de- Medicine,c Indiana University School of Medicine, Indiana University School of Dentistry,b and Lilly Research Laboratories, A review of the patient’s prior laboratory evalua- tion revealed normal blood counts, serum chemistry profile, and erythrocyte sedimentation rate. Vitamin Conflicts of interest: Frank P. Bymaster, MS, PhD is currently employed in the research laboratory of Eli Lilly and Company.
B12 and folate levels were normal. Oral culture was John S. Kennedy, MD was employed by Eli Lilly and Company obtained and was negative for fungus.
from 1997-2002, but is now employed by Indiana University A diagnosis of idiopathic glossodynia was School of Medicine. The other two authors are unaffiliated with rendered. The patient was started on olanzapine Eli Lilly and Company and contacted Frank Bymaster, MS, PhD (Zyprexa, Eli Lilly and Company, Indianapolis, after the patient was seen in follow-up.
Reprint requests: Ginat W. Mirowski, DMD, MD, Department of Ind) 5 mg once daily. He noted improvement of Dermatology, Indiana University School of Medicine, 550 N.
symptoms within 3 days. His taste perception nor- University Blvd, Suite 3240, Indianapolis, IN 46202. E-mail: malized, burning sensation was eliminated, and appetite improved. His mood was euthymic without evidence of anxiety. He remained free of symptoms ª 2004 by the American Academy of Dermatology, Inc.
doi:10.1016/j.jaad.2004.02.016 onset of glossodynia and remitted fully upon pain glossodynia and their associated etiologies Treatment of secondary glossodynia is aimed at correcting the underlying condition. Reassuring that the pain is not related to neoplasia is appropriate in patients with cancerophobia. Topical antifungals are controversial treatments in cases of normal exams and cultures. Topical steroids and viscous lidocaine have been cited as therapy for glossodynia, but often The mainstay of treatment for primary glossodynia has been benzodiazepines and tricyclic antidepres-sants. Low-dose clonazepam was successful in re-ducing symptoms of burning mouth syndrome in67% of 30 patients.Chlordiazepoxide is another long-acting benzodiazepine that has been successful Glossodynia (burning mouth syndrome, glos- in the treatment of glossodynia.Low-dose tricyclic salgia, stomatodynia, glossopyrosis, glossitis) is antidepressants such as amitriptyline and doxepin defined as a spontaneous syndrome of burning have been effective in treating glossodynia, neurop- sensation, discomfort, pain, irritation, or rawness of athy, fibromyalgia, and other chronic pain con- the tongue, lips, or oral cavity often without organic cause.In a cohort of 70 patients with glossodynia, This case is noteworthy because the patient was 80% were female with a mean age of 59 years.The refractory to benzodiazepine and antidepressant reported prevalence of glossodynia is between 0.7% therapy. The patient responded to a medication currently indicated only for psychosis in schizophre- There are no diagnostic criteria for glossodynia.
nia and acute mania in bipolar disorder. This is the Patients often complain of pain, tenderness, or first reported case in the literature of olanzapine burning with symptoms often worsening throughout successfully relieving the symptoms of glossodynia.
the day. Patients may also complain of dysgeusia Recent case reports demonstrate the potential suc- (altered taste) or xerostomia (dry mouth). In a pro- cess of olanzapine in treating patients with other pain spective study of 96 patients, dysgeusia was detected in 44.8% of patients with glossodynia, approximately The mechanism responsible for this effect of half of which did not present with a complaint of olanzapine is unknown. Olanzapine is a potent dysgeusia.Xerostomia has been reported in more antagonist at a number of neuronal receptors in- than 20% of patients with glossodynia.On exam- cluding dopamine and serotonin receptors and ination, the tongue appears normal, with erythema, produces antipsychotic effects with reduced risk of edema, or atrophy present in rare cases.
The work-up for glossodynia should be guided by that the efficacy of olanzapine in treating glossodynia clinical suspicion and may include: detailed patient is due, in part, to its blockade of a subset of these history, complete blood count, serum iron, serum serotonin receptors. Olanzapine may exhibit anxio- B12 and folate levels, mucosal culture, serum tests lytic, antipsychotic, and gastrointestinal anti-motility ¨gren’s syndrome antibodies (SS-A, SS-B), properties secondary to other serotonin receptor glycosylated hemoglobin, patch testing, and biopsy (if exam is abnormal).Further consultation may be or involvement of alpha2-adrenoreceptorsby helpful. In the case presented above, no underlying olanzapine may be partially responsible for the pain systemic conditions, nutritional deficiencies, adverse relief observed in our patient. Possible side-effects drug reactions, or oral disorders were detected of olanzapine include weight gain, which may be to explain the etiology of the patient’s continued substantial in a small subset of patients. Minimal anticholinergic side effects may be observed in orders, particularly those with depressive or anxiety geriatric patients.Olanzapine, a newer anti- symptoms, have been reported in 30%-72% of psychotic and dopamine modifier, may induce or un- patients with glossodynia.However, in the case cover extrapyramidal syndromes such as parkinson- of our patient, symptoms of dysphoria and anxiety ism, akathisia, tardive dyskinesia, and neuroleptic were felt to be secondary to oppression associated malignant syndrome. The risk of the atypical anti- with pain since these symptoms developed after the cardiotoxicity is much lower than has been reported 9. Regezi JA, Sciubba JJ, Jordon RCK, eds. Oral pathology: clinical for the older typical medications including pimozide.
pathologic correlations (4th ed). St. Louis, MO. Saunders; 2003;123-125.
Glossodynia is a challenge for many health care 10. Bogetto F, Maina G, Ferro G, Carbone M, Gandolfo S.
providers. Etiology often cannot be identified and Psychiatric comorbidity in patients with burning mouth success of treatment is currently patient-specific and syndrome. Psychosom Med 1998;60:378-85.
unpredictable. When the side effect profile or safety 11. Grushka M, Epstein J, Mott A. An open-label, dose escalation concerns of benzodiazepines and tricyclic anti- pilot study of the effect of clonazepam in burning mouthsyndrome. Oral Surg Oral Med Oral Path Oral Radiol Endod depressants outweigh the benefits of their use, or the patient is refractory to such medications, 12. Godfrey RG. A guide to the understanding and use of tricyclic olanzapine may prove to be the next successful antidepressants in the overall management of fibromyalgia alternative in providing relief to the patient with and other chronic pain syndromes. Arch Int Med 1996;156: glossodynia. Therefore, further experience with 13. Sindrup SH, Jensen TS. Pharmacologic treatment of pain in olanzapine in glossodynia, confirming the effect polyneuropathy. Neurology 2000;55:915-20.
observed in such patients as this, is warranted by 14. Kiser RS, Cohen HM, Freedenfeld RN, Jewell C, Fuchs PN.
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15. Khojainova N, Santiago-Palma J, Kornick C, Breitbart W, Gonzales GR. Olanzapine in the management of cancer pain.
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19. Schreiber S, Getslev V, Backer MM, Weizman R, Pick CG. The 6. Tanaka M, Kitago H, Ogawa S, Tokunaga E, Ikeda M, Tomita H.
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7. Gorsky M, Silverman S Jr, Chinn H. Clinical characteristics and 20. Bymaster FP, Falcone JF. Decreased binding affinity of olan- management outcome in the burning mouth syndrome: an zapine and clozapine for human muscarinic receptors in intact open study of 130 patients. Oral Surg Oral Med Oral Pathol clonal cells in physiological medium. Eur J Pharmacol 2000; 8. Drage LA, Rogers RS III. Glossodynia. In: Lebwohl MG, 21. Kennedy JS, Bymaster FP, Schuh L, Calligaro DO, Nomikos G, Heymann WR, Berth-Jones J, Coulson I, eds. Treatment of Felder CC, et al. A current review of olanzapine’s safety in the skin disease: comprehensive therapeutic strategies. London.
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