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Home , Drooling, Geographic tongue, Glossitis, Lichen planus

Symptomatic benign migratory : CASE REPORTS report of two cases and literature review MichaelJ. Sigal, DDS,MSC, Dip Paed, MRCD(C) DavidMock, DDS, PhD, FRCD(C)

Abstrac~ Benign migratory glossitis (geographic ) is a commonclinical finding in routine pediatric dentistry. The condition usually is discoveredon routine clinical examination,appearing as an asymptomatic, -like region on the dorsumof the tongue. The mayrecur at different sites on the tongue, creating a migratory appearance, and in manycases, will resolve completely. The presentation of symptomatic geographictongue in children is rare. This article presents two cases of symptomaticgeographic tongue. Both children presented with a chief complaintof significant oral pain which wasaffecting daily activity, eating, and sleeping. Both patients presentedwith a classical clinical presentationof ulcer-like regions on the dorsum of the tongue in which the filiform papillae were denuded.Successful managementwas achieved with topical and systemic . The clinician should be aware that this condition may be symptomatic fn children. (Pediatr Dent 14:392-96, 1992)

Introduction Benign migratory glossitis (BMG)is a condition re- account for the increased prevalence observed, since ferred to in the literature by a variety of names,such as: BMGmay be seen more often in children with associ- , migrans, annulus migrans, ated major illnesses. Both articles also suggested that and wandering of the.tongue. 1-4 This inflamma- the increased incidence mayreflect the different racial/ tory condition first was reported by RayerI in 1831. ethnic backgrounds of the samples examined.12, 13 ~. I~is a benign, inflammatory disorder occurring most The etiology of geographic tongue is still unknown. ff6mmonly on the .dorsum of the tongue, possibly ex- Someconsider the condition to be a congenital anomaly tending onto the lateral borders..The characteristic ap- and others believe it to represent an acute inflammatory pearance’includesmultifocal, circinate; irregular reaction. Attempts have been made to demonstrate an erythematous patches bounded by a slightly elevated, association between various systemic and/or psycho- keratotic band or line. The erythematous patches repre- logical conditions and BMG.These conditions include sent loss of filif0rm papillae and a thinning of the ,7, 14, Reiter’s syndrome,14 , gastro- . The white border is composedof regenerat- intestinal disturbances, nutritional disturbances, ing filiform papillae and a mixture of keratin and neu- c a ndid’asis,~ planus, hormonalimbalance,10 psy- trophils. The surface is nonulcerated, but appears ulcer- chological upsets, l~and allergies. 16 A definitive causal ated due to the loss of the surface papillae and keratin. relationship has not been established. These well-defined, elliptical vary in size from a Heredity may play a role in the etiology of BMG. few millimeters to several centimeters. The location and Redman17 postulated a polygenic mode of inheritance pattern undergo change over time, thereby accounting for geographic tongue. Eidelman et al. 18 determined for the name "migratory." This apparent migration is that the prevalence of BMGin parents and sibling com- due to a concurrent epithelial desquamation at one binations was significantly higher than that observed in location5-8 and proliferation at another site. the general population. They concluded that geographic The prevalence of BMGin the general population is tongue was a familial condition in which heredity plays between 1.0 and 2.5%.2-4, 9 Various age groups can be a significant role. affected with no apparent racial predilection; however, Marks and Tait 19 provided additional support for a the condition appears to be more commonin females genetic basis of BMGby demonstrating an increased with reported female-to-male ratios of 5:3 and 2:1.1,2,10 incidence of tissue type HLA-B15in atopic patients Redman11 observed a 1% prevalence of BMGin with geographic tongue. Wysocki and Daley5 investi- schoolchildren, with an equal distribution between gated the prevalence of BMGin patients with juvenile males and females. A similar finding was noted in an , because it is known that HLA-B15occurs investigation4 of university students by Meskin. more frequently in insulin-dependent diabetic pa- Very high rates of occurrence of BMGin children in tients. 20 They discovered a prevalence of 8%for BMGin Japan (8%) and Israel (14%), with a peak age of diabetic patients and concluded that BMGmay be a years, were reported in studies conducted on hospital- clinical marker for insulin-dependent diabetes mellitus.5 ized pediatric patients. 12, 13 This sampling bias could

392 PEDIATRlC"DENTISTRY" NOVEMBER/DECEMBER, 1992 ~ VOLUME14, NUMBER6 Marks and Simons21 discovered a significantly in- with migration of polymorphonuclear leukocytes and creased frequency of among patients with geo- toward the zone of epithelial degenera- graphic tongue, as compared to the normal population. tion. Munroabscesses also may be seen. The advancing In a study of atopic patients with a history of asthma margin is outlined clearly by a dense, polymorpho- and/or , Marks and Czarny16 found a 50%preva- nuclear infiltration of the acanthotic epithelium and lence of BMG.They also observed that the frequency of corium. The border may demonstrate a zone of geographic tongue increased significantly in the control hyperkeratinization (). Tissues which were group with no clinical history of atopy, but who had a affected previouslly show a chronic inflammatory reac- positive skinprick test to commoninhalant allergens. tion.8, 10, 26, 27 Geographic tongue is characterized by They concluded that a positive association between periods of exacerbation and remission. During remis- geographic tongue and atopy exists, and further postu- sion, lesions resolve without residual formation. lated that geographic tongue and asthma/rhinitis may Whenlesions recur, they tend to occur in a new loca- have a similar pathogenesiso Both conditions are recur- tion, thus producing the migration effect. rent, inflammatory, and can be initiated by contact with Since BMGis asymptomatic in most cases, no treat- external environmental irritants. Geographic tongue is ment is required other than patient reassurance of the probably a sign commonto those individuals who have benign and self-limiting nature of the disorder. If symp- a tendency to develop a recurrent acute inflammatory toms are present, the patient should be instructed to reaction on surfaces which are in contact with the exter- avoid any knownirritants, such as hot, spicy, or acidic nal environment. foods. If treatment is warranted, it should be palliative/ Psoriasis, a cutaneous dermatological condition, ap- symptomatic care using topical anesthetic rinses or pears to be related to an accelerated rate of epithelial gels, , or steroids. 6, 8, 10, 25, 28 A psycho- turnover, resulting in epithelial and is seen logical component may contribute to the development clinically as erythematous and/or white scaly of BMG,and tranquilizers maybe considered in patient plaques. 7 BMGhas been suggested as an oral manifes- management.25 tation of psoriasiso7, 22, 23 A review of the literature on BMGfailed to produce The association between and BMG a reported case of symptomatic BMGin children. This supports a genetic basis for the development of the article presents two cases of therapeutic management conditiono18 The fissures mayact as stagnation areas on of children with symptomatic BMG. the tongue surface in which glossitis maybegin. 8 Geo- graphic tongue appears to be associated with geographic Case Reports 6, 8 which has a clinical and histological ap- Case One pearance similar to BMGbut occurs on an extraglossal A 4-year-old Caucasian boy was seen with a chief intraoral24 site. complaint of oral discomfort and increased salivation. The diagnosis of BMGusually is based solely on the history and clinical presentation which would include Review of his medical history revealed an innocent heart murmur, sleep apnea until the age of 9 months, characteristic migratory pattern and chronic nature. and recurrent otitis media which required placement of The vast majority are asymptomatic and noticed during myringotomytubes at the age of 1 year. At the initial the course of a routine oral examination, or self-exami- nation.6, 8,10, 25 Only Cooke8 reported that a significant visit there was no history of allergy to or environmental factors. number of patients complained of some oral sensitivity The patient’s mother reported that approximately or discomfort, most often described as a "burning sen- one month earlier, her son began to experience oral sation." No oral sensitivity associated with cases of discomfort, evidenced by crying, placing objects in his BMGin pediatric populations has been reported. , and a marked increase in salivation and expec- If a patient presents with suspected BMG,a differen- toration. The child reported that his mouth had an tial diagnosis based on adult studies should include "awful taste." atrophic , psoriasis, Reiter’s syndrome, atro- He was placed on nystatin (Mycostatin ®, Bristol- phic , systemic luaus erythematosus, Myers Squibb Canada, Montreal Quebec, Canada), , and drug reaction. 6, 25 100,000 units, three times a day, by his family physician The histological features of BMGare those of a local- without success. His general dentist suspected that the ized acute glossitiso The central erythematous portions early eruption of his first permanent molars could be represent an area of epithelial degeneration and the the source of the discomfort and referred him to our absence of the stratum corneum, with very little alter- clinic for consultation. The mother also reported that no ation in the basal layer of epithelium. Beneath the epi- other family membershad a similar condition. thelium is a dense infiltration of inflammatory cells

PEDIATRIC DENTISTRY: NOVEMBER/DECEMBER,1992 - VOLUME14, NUMBER6 393 On examination, the child appeared normal, healthy, for Cundida.The nystatin was stopped immediately and and well-developed. There were no abnormal extraoral the mother was informed to call if clinical findings. Intraoral examination revealed a com- recurred. plete, caries-hee primary dentition, good , Four weeks later he experienced similar symptoms. and no evidence of gingival . The dorsal Examination revealed irregular, circumscribed surfaceof the tongue demonstrated a patternconsistent erythematous areas on the dorsum of the tongue. These with a resolving geographic tongue, with irregular cir- areas were devoid of filiform papillae. The lesions had cumscribed areas devoid of filiform papillae (Figure). margins with a raised white appearance that could not The surface was not erythematous and there was no be scraped off. The regions were not tender to touch. evidence of leukoplakia or white curd-like Exfoliative cytology was repeated and did not show pseudomembranes, as seen in fungal infections. The any evidence of candidiasis. A complete blood count first permanent molars had not erupted and radio- with differential was within normal limits. graphic examination demonstrated that they were The boy was instructed to rinse with 5 cc of a present at a normal stage of development with no evi- diphenhydramine HCI suspension (BenadryP, Parke- denceofcommunicationbehveen thecryptof themolar Davis, Moms Plains, NJ, 12.5 mg/5 cc) up to four times and the oral cavity. per day, holding it over his tongue and then swallowing it. All the symptoms were relieved within 48 hr of initiation of antihistamine therapy. In the subsequent six months, the condition recurred twice and immediate treatment with the diphenhydramine suspension provided symptomatic relief within 24 hr. Case Two A 3-year-old Caucasiangirl gaveachief complaint of oral pain that prevented eating and drinking. She had multiple environmental allergies and was suspected to have asthma. The remainder of her medical history was unremarkable. No other family members had a similar oral condition. Her father reported that her mouth be- came extremely painful every month for two to three days and this pain would resolve spontaneously with- out treatment. Examination revealed a well-circumscribed, irregu- Figure. l~hrwsurface of the tongue in Case 1 denionstr,iting irrcyqlar areas devoid of filiform papillae consistent with the lar pattern on the dorsum of the tongue devoid of pattern seen in geographic tongue. filiform papillae, consistent with BMG. The areas were asymptomatic and appeared to be resolving, so no treatment was performed. The parent was informed The anti-fungal agent was stopped and the mother about the nature of the condition and instructed to was instructed to contact our clinic if an acute exacerba- return the child to our clinic if the lesions and symp- tion occurred. Nine months later, the mother reported toms recurred. that one month earlier, her son had his DFT booster and The child returned six months after the initial ex- for seven days had a of 101-102°F. At that time, amination with a similar clinical presentation but dur- the tongue lesions reappeared with oral pain, increased ing a period of acute pain. The diagnosis of symptom- salivation with expectoration and a generalized agita- atic BMG was made and the child was instructed to tion. He was placed on nystatin and a lidocaine rinse with 5 cc of diphenhydramine HCI suspension (Xylocaine@,Astra, Mississauga, Ontario, Canada) gel (Benadryl, 12.5 mg/5 cc) up to four times per day, for topical pain relief by his family physician, but this holding it over her tongue for 1-2 min and then swal- provided only temporary relief. Review of his medical lowing it. She was relieved of all her symptoms within history at this time revealed that he was developing 24 hr of initiation of antihistamine therapy. allergies to environmental and food factors. Clinical examination again indicated a resolving pat- Discussion tern on the dorsum on the tongue, and a significant Geographic tongue or BMG is a common finding amount of with a complaint of foul taste. during routine examination of children. In previous Exfoliative cytology was performed and was negative investigations, the condition was asymptomatic. Only

394 PEDIATRICDENTISTRY: NOVEMRE~DECEMRER, 1992 -VOLUME 14, NUMRER6 Cooke,8 in 1962, reported that a significant number of The etiology of BMGis unknown, but the condition adults with BMGhad varying degrees of oral sensitiv- maybe linked to allergies. 21 It is interesting to note that ity associated with the condition. To our knowledge, both of these children presented with various environ- these are the first two cases of symptomatic geographic mental allergies. tongue reported in pediatric patients. In both cases, symptoms were severe enough to interfere with sleep- Dr. Sigal is associate professor, paediatric dentistry, Faculty of Den- ing and eating. tistry, University of Toronto; head, Dentistry for the Disabled, Mt. Sinai Hospital; and chief of dentistry, Queen Elizabeth Hospital. Dr BMGis capable of producing symptoms in children Mockis professor, Oral , Faculty of Dentistry, University of that are significant enough to require management.The Ontario; chief of dentistry and head, Oral Pathology, Mt. Sinai Hospi- of BMGin children should in- tal. All are located in Toronto, Ontario, Canada. Reprint requests clude atrophic candidia~is, drug-induced reactions, lo- should be sent to: Dr. MichaelJ. Sigal, Mt. Sinai Hospital 600 Univer- sity Avenue, Toronto, Ontario, Canada M5G1X5. cal trauma and a severe neutropeniao Psoriasis, Reiter’s syndrome, atrophic lichen planus, , and sys- 1. Prinz H: Wandering rash of the tongue (geographic tongue). temic erythematosus can produce similar lesions, Dent Cosmos 69:272-75, 1927. but are rare in children. 2. Halperin V, Kolas S, Jefferies KR, Haddleston SO, Robinson HBG:Occurrence of , benign migratory glossitis, The child with symptomatic tongue ulcerations median rhomboid glossitis, and fissured tongue in 2,478 den- should have a complete medical and dental history tal patients. Oral Surg 6:1072--77, 1953. taken, followed by a comprehensive extra- and intraoral 3. Richardson ER: Incidence of geographic tongue and median examination. If a diagnosis of BMGcannot be made rhomboid glossitis. Oral Surg 26:623-25, 1968. 4. Meskin LH, Redman RS, Gorlin RJ: Incidence of geographic based on the history and examination due to an atypi- tongue among3,668 students at the University of Minnesota. cal, symptomatic presentation, then a complete blood J Dent Res 42:895, 1963. count with differential should be obtained to rule out 5. WysockiGP, Daley TD: Benign migratory glossitis in patients neutropenia and to assess the general state of health. In with juvenile diabetes. Oral Surg 63:68-70, 1987. 6. Brooks JK, Balciunas BA: Geographic stomatitis: review of the addition, exfoliative cytology of the area should be literature and report of five cases. J AmDent Assoc 115:421- performed to rule out candidiasis. If a definitive diag- 24, 1987. nosis still cannot be made, a of a representative 7. Pogrel MA,Cram D: Intraoral findings in patients with pso- region of the lesion would be warranted. riasis with special reference to ectopic geographic tongue (erythema circinata) Oral Surg 66:184-89, 1988. Most patients require no definitive treatment other 8. Cooke BED: Median rhomboid glossitis and benign glossitis than observation and reassurance of the benign nature migrans (geographic tongue). Br Dent J 112:389-93,1962. of the condition. For painful BMG,recommended sup- 9. Kullaa-Mikkonen A, Mikkonen M, Kotilainen R: Prevalence portive and symptomatic management would include of different morphologic forms of the human tongue in young Finns. Oral Surg 53:152-56, 1982. a bland diet, plenty of fluids, acetaminophen for sys- 10. BanoczyJ, Szabo L, Csiba A: Migratory glossitis: a clinical- temic pain relief, and a topical anesthetic agent such as histologic review of seventy cases. Oral Surg 39:113 -21,1975. viscous lidocaine or (TantumTM, Riker/ 11. RedmanRS: Prevalence of geographic tongue, fissured tongue, 3M, London, Ontario, Canada) rinse for local pain re- median rhomcoid glossitis and hairy tongue among 3,611 Minnesota schoolchildren. Oral Surg 30:390-95, 1970. lief. If available, benzydamine may be preferred be- 12. Togo T: Clinical study on geographic tongue. KurumeMed J cause of a reported combined and anti-in- 24:1156-72, 1961. flammatory29 effect that lasts for up to 3 hr. 13. Rahamimoff P, Muhsam HV: Some observations of 1,246 If the lesions should recur, or their severity is such cases of geographic tongue. AmJ Dis Child 93:519-25, 1957. 14. Weathers DR, Baker G, Archard HO, Burkes EJ: Psoriasis- that the child does not have adequate relief from the form lesions of the with emphasis on ectopic symptomatic therapy, then an antihistamine, such as geographic tongue. Oral Surg 37: 72-88, 1974. diphenhydramine HC1 (Benadryl) should be used. The 15. Redman RS, Vance FL, Gorlin RJ, Peagler FD, Meskin LH: child should rinse with 12.5-25 mg (1 to 2 teaspoons) Psychological componentin the etiology of geographic tongue. J Dent Res 45: 1403-8, 1966. depending on age and weight, holding it over the tongue 16. Marks R, Czarny D: Geographic tongue: sensitivity to the for a few minutes, and then swallowing, three to four environment. Oral Surg 58:156-59, 1984. times29 per day for up to seven days. 17. RedmanRS, Shapiro BL, Gorlin RJ: Hereditary component in If the lesions do not respond to antihistamine therapy, -the etiology of benign migratory glossitis. AmJ HumGenet 15:124-30, 1972. then a , such as betamethasoneas a 500-~tg 18. Eidelman E, Chosack A, Cohen T: Scrotal tongue and geo- tablet dissolved in water, can be used as a rinse for a few graphic tongue: polygenic and associated traits. Oral Surg minutes and swallowed, twice daily for seven to 14 42:591-96, 1976. days.29 The steroid only should be used in patients who 19. Marks R, Tait B: HLAantigens in geographic tongue. Tissue Antigens 15:60-62, 1980. do not respond to either supportive/symptomatic or 20. Murrah VA: Diabetes mellitus and associated oral manifesta- antihistamine therapy. The child’s physician should be tions: a review. J Oral Path 14:271-81, 1985. consulted when using steroids. 21. Marks R, Simons M: Geographic tongue - as manifestation of atopy. Br J Dermato1101:159~2, 1979.

PEDIATRICDENTISTRY: NOVEMBER/DECEMBER, 1992 N VOLUME14, NUMBER6 395 22. DawsonTAJ: Tongue lesions in generalized pustular psoria- 26. Shafer WG,Hine MK,Levy B: A Textbook of Oral Pathology. sis. Br J Dermato191:419-24,1974. Philadelphia: WBSaunders, 1983, pp 28-29. 23. Van der Wal N, Van der Kwast AM, Van Dijk E, Van der Wal 27. DawsonTAJ: Microscopic appearance of geographic tongue. h Geographic stomatitis and psoriasis. Int J Oral Maxillofac Br J Dermato181: 827-28, 1969. Surg 17:106-9, 1988. 28. Brightman VJ: Diseases of the tongue, in Burkett’s Oral Medi- 24. Rhyne TR, Smith SW, Minier AL: Multiple annular, cine. MLynch, V Brightman, MGreenberg eds. Philadelphia: erythematous lesions of the oral mucosa. J Am Dent Assoc JB Lippincott, 1984, pp 453. 116:217-18, 1988. 29. Compendiumof Pharmaceuticals and Specialties. CMEKrogh 25. Raghoebar GM, de Bont LGM,Schoots CJF: ed. Ottawa, Ontario, Canada: Canadian Pharmaceutical As- of the oral mucosa. Report of two cases. Quintessence Int 119: sociation, 1992, pp 1119, 125, 136. 809-11,1988.

Consultants for the 1993 AmericanBoard of Pediatric Dentistry Section Examinations

Jeffrey R. Blum -- Wynnewood, PA Larry S. Luke -- Simi Valley, CA Robert A. Boraz -- Kansas City, KS James W. McCourt -- San Antonio, TX Steven K. Brandt -- Temple, TX William A. Mueller- Denver, CO Charles M. Brenner -- Rochester, NY John E. Nathan -- Oak Brook, IL Paul S. Casamassimo -- Columbus, OH MamounM. Nazif-- Pittsburgh, PA George J. Cisneros -- Pelham Manor, NY Peter W.H. Ngan -- Columbus, OH Robert O. Cooley -- Flossmoor, IL Melvyn N. Oppenheim -- Scarsdale, NY Joseph C. Creech, Jr. -- Mesa, AZ James W. Preisch -- Columbus, OH Diane C.H. Dilley -- Chapel Hill NC Richard A. Pugliese -- Cranston, RI Burton L. Edelstein -- New London, CT Michael W. Roberts -- Chapel Hill, NC Terrance Fippinger -- Evanston, IL Peter J. Ross -- Lancaster, PA Donald O. French -- Tucson, AZ Howard S. Schneider --Jacksonville, FL Robert R. Gatehouse -- Middletown, CT Robert S. Sears -- Sterling, VA Roger D. Gausman -- Hutchinson, KS Andrew L. Sonis -- Newton Highlands, MA Norman L. Goldberg- Concord, MA Robert L. Stonerock -- Federal Services David L. Good -- Canoga Park CA Nilsa Toledo -- Hato-Rey, Puerto Rico Douglas E. Holmes -- Shiprock, NM Kenneth C. Troutman -- Scarsdale, NY Herschel L. Jones -- Fort Lewis, WA Erwin G. Turner -- Lexington, KY Paul E. Kittle -- Fort Lewis, WA Paul L. Vitsky -- Fredericksburg, VA Doron Kochman -- Pittsford, NY Joseph H. Wenner -- St. Cloud, MN Donald W. Kohn -- New Haven, CT

396 PEDIATRICDENTISTRY: NOVEMBER/DECEMBER, 1992 N VOLUME14, NUMBER6