ABSTRACT ÖZET

Geographic tongue lesions may also appear, though rarely, on other mucosal areas of the oral cavity. This type of lesion is called as geographic stomatitis. The cause is unknown; yet, emotional stress, nutritional deficiencies, and hereditary factors are suggested. Clinically, it occurs as red annular patches of the labial or buccal mucosa, soft palate; rarely gingiva and floor of mouth. There are mild erosions of the mucosa on these lesions, which are frequently multiple and well-circumscribed. Characteristically lesions change their shape and location. The condition is usually asymp-tomatic. When symptomatic, palliative treatment is recommended.

The purpose of the present paper is to report two cases of geographic stomatitis and to discribe a pos-sible connection between this condition and patients’ emotional status.

Coğrafik dil lezyonları oral kavitenin diğer muko-zal bölgelerinde de nadiren görülebilir. Bu durum coğrafik stomatit olarak adlandırılmaktadır. Nedeni bilinmemektedir. Ancak emosyonel stres, beslenme bozuklukları ve herediter faktörlerin neden olabile-ceği ileri sürülmektedir. Klinik olarak labial ya da bukkal mukozada, nadiren gingiva ve ağız tabanında kırmızı, halka şeklinde yamalar tarzında görülürler. Sıklıkla çok sayıda ve belirgin sınırları olan bu lezyon bölgelerinde mukozanın hafif erozyonuna rastlanır. Karakteristik olarak lezyonlar şekil ve yer değiştirir. Bu durum genellikle asemptomatiktir. Semptomatik olduğunda palyatif tedavi önerilmektedir.

Bu makalenin amacı, iki coğrafik stomatit olgusu sunmak ve bu durum ile hastanın emosyonel statüsü arasındaki olası ilişkiyi tanımlamaktır.

Hacettepe Diş Hekimliği Fakültesi DergisiCilt: 32, Sayı: 2, Sayfa: 73-78, 2008

Geographic Stomatitis

Coğrafik Stomatit

*Nursel AkkAyA DDS, PhD, **Seher kArAGül DDS, PhD, *Aydan kANlı DDS, PhD

*Hacettepe University, Faculty of Dentistry, Department of Oral Diagnosis and Radiology.**Private Dentist

KEYWORDSGeographic stomatitis, migratory stomatitis, stomatitis

areata migrans, erythema circinate migrans, ectopic geographic tongue.

ANAHTAR KELİMELERCoğrafik stomatit, migratuar stomatit, stomatitis areata migrans, eritema sirsinate migrans, ektopik coğrafik dil.

These cases were presented in 3. Scientific Symposium of Oral Diagnosis and Dentomaxillofacial Radiology Society, 21-23 April 2006, Kemer, Antalya

OLGU RAPORU (Case Report)

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INTRODUCTION

Geographic tongue is a condition character-ized by atrophy of the filiform papilae on single or multiple areas of the tongue in an irregular pattern which is frequently accompanied by pe-ripheral keratosis. These geographic lesions oc-cur on the dorsum and lateral borders of tongue in about 1–2 % of general population1. Similar changes may appear on other mucosal areas of the oral cavity. This uncommon condition was first described in 1955 under the term “ery-thema migrans” by Cooke2. It is also named as migratory stomatitis, stomatitis areata migrans, erythema circinate migrans, ectopic geographic tongue, geographic stomatitis, annulus migrans, Cooke’s disease and migratory mucositis3.

The most frequently reported sites of geo-graphic stomatitis (GS) are the buccal mucosa, lower labial mucosa and mucobuccal fold. In-volvement of the gingiva, alveolar mucosa, soft palate and floor of the mouth is unusual4.

Clinical presentation of GS is described as slightly raised, round, erythematous lesions that are circumscribed by well-defined whitish bor-ders. The lesions vary in size from a few millime-ters to several centimeters in diameter5.

Hume6 has made a classification of GS based on its clinical distribution:

Type 1: Lesions on the dorsum, lateral bor-ders and tip of tongue with possible extension to undersurface. The lesions may migrate with time and show both active and remission phases (geographic tongue, without geographic lesions elsewhere in the mouth).

Type 2: Geographic tongue, accompanied by geographic lesions elsewhere in the mouth.

Type 3: Atypical tongue lesions whether or not accompanied by lesions elsewhere in the mouth. Atypical tongue lesions consist of two forms: A- Fixed forms; one or two areas of tongue are affected but movement is not observed. In-stead, they disappear only to recur after a pe-riod of time at the same area. B- Abortive forms; these start as yellow-white patches but disappear

before acquiring the typical appearance of geo-graphic lesion.

Type 4: Geographic lesions elsewhere in the mouth, without the presence of a geographic tongue.

Although its etiology is unknown, many dif-ferent conditions, such as pustular psoriasis, Re-iter’s syndrome, atopy, stress may be associated with GS. The condition is more common in men than women. No particular age group shows an increased tendency to GS7.

The purpose of the present paper is to re-port two additional cases of GS and to describe a possible connection between this condition and patients’ emotional status.

CASE 1

33 year-old woman applied to our clinic for routine dental examination. Her medical his-tory was unremarkable. Extraoral examination revealed hyperkeratosis on her left hand. There were no skin lesions present except the hyper-keratosis of hand. Intraoral examination showed multiple erythematous lesions surrounded by a narrow white margin on the right buccal muco-sa, mucobuccal fold, the right upper labial mu-cosa and the dorsum of the tongue (Figure 1–3). The lesions varied in size and had well-defined, white, slightly raised, circinate borders. No evi-dence of vesicle formation was noted. There was fissuring of the dorsum of the tongue. She was unaware of mucosal lesions but she was suffering from burning of tongue after consuming hot or spicy foods. Patient expressed that she has been under stressful condition during the last three months because of her mother’s health problems and intensity of symptoms has increased during this period. Patient was referred to Dermatology Clinic for the evaluation of lesions on her hand and possible relationship between skin and oral mucosal lesions. Skin lesions of her hand were diagnosed as “contact dermatitis”. Therefore, it was concluded that skin and oral mucosal lesions were not related.

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Oral lichen planus and acute atrophic candidia-sis were considered as differential diagnoses. Lichen planus could be excluded as the clinical appearance was not consistent with this condi-tion. A smear from the lesions of oral mucosa for the evaluation of oral candidiasis was nega-tive. Her dermatologist decided to take biopsy, but the mucosal lesions disappeared on appoint-ment day. However, no scars were left at the sites of the previous lesions. An incisional biopsy was performed for tongue lesions. Microscopic examination of the biopsy specimen showed acanthosis in the surface epitelial layer, subepi-thelial neutrophil infiltrates and the formation of microabscesses and chronic inflammatory infil-trate in submucosa. Microscopic diagnosis was “geographic tongue”. Benzydamin HCl and tri-amcinolone acetonide were used for symptomat-ic treatment. We offered to avoid topical factors that exacerbate her symptoms, such as very hot, spicy, or acidic foods. A two weeks follow-up conducted with the patient revealed only the mi-gratory behavior of these asymptomatic lesions. The clinical diagnosis of geographic stomatitis was made.

FIGURE 1

Multiple, well-demarcated erythematous lesions of geographic stomatitis on the maxillary labial mucosa in Case 1.

FIGURE 2

Involvement of the mandibular mucobuccal fold with discrete, annular lesions in Case 1.

FIGURE 3

Deep grooves and fissures on the dorsum of the tongue accompanied by geographic lesions in Case 1.

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CASE 2

43 year-old man had applied to our clinic for toothache. In his medical history, patient ex-pressed that he has been under medical treat-ment until he was 19 years old, due to congenital hypothyroidism. Currently he is not using any medication. He has been undergoing psychologi-cal treatment since 1993 because of over ner-vousness, instability to control himself and prob-lematic relations in the working environment.

Extraoral examination was unremarkable. During intraoral examination it was detected that he had fissured tongue and there were bilater-ally, slightly raised, erythematous lesions on the buccal mucosa (Figure 4). The lesions had white, circinate borders. In these regions, there was no symptom in the burning or pain characteristics. Also the patient expressed that he was unaware of this condition. Result of the smear taken from the lesion region in order to detect possible exis-tence of candida albicans showed normal flora. Full blood test results, vitamin B12 and folate lev-els were normal. 15 days later when he applied to clinic with these results, it was seen that there were same type of lesions in the nearby regions of lower labial mucosa and comissura labiorum. However, mucosa at the sites of previous lesions was completely normal (Figure 5). Although le-

sions were migrating, they were similar in sight. Due to all these characteristic attributions, no biopsy was taken and the disease diagnosed as “geographic stomatitis”. Nonetheless, the patient was directed to internal medicine department. Due to detection of hypercalsuri in urinalysis examination the patient is directed to urology department. In his abdomen CT and renal USI, renal cortical cysts were seen in both of his kid-neys. Suspected of internal hernia in the right hemi region of the abdomen, radiology depart-ment requested intestinal passage examination. Results were normal.

Patient monitored for a year with regular periods. It was observed that the intensity and localization of the lesions changed and this situa-tion was attributed to his emotional stress.

DISCUSSION

In the literature limited numbers of GS cases were reported, however some of the researchers think that its real incidence is more than expected since the disease mostly proceed in asymptom-atic nature and easy to diagnose when encoun-tered together with geographic tongue8. Never-theless, Bouquot and Gundlach9 reviewed data on 23616 white American and found no patient with geographic stomatitis. Daneshpazhooh et.

FIGURE 4

Geographic stomatitis appearing as erythematous lesions on the buccal mucosa in Case 2.

FIGURE 5

Multiple small lesions of the geographic stomatitis on the mandibular labial mucosa in Case 2.

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al.10 denote that this lesion is rarely encountered in the population.

Etiology of GS is not understood complete-ly. There is no certain difference between geo-graphic tongue lesions and GS but GS defined as the type of these lesions encountered in the oral mucosa. Geographic tongue can be related to atopic conditions (hay fever, eczema, and asthma), reactive bronchitis, allergy, hormonal disturbances (pregnancy, juvenile diabetes mel-litus), nutrition deficiencies, anemia, gastroin-testinal anomalies, infectious agents and lithium therapy. Psychological status as an etiological factor has considerable effects11. Both of these patients had psychological problems. We have observed that alteration in the intensity of the le-sions was correlated with the patient’s emotional status. Patients having a psychological treatment supported this view.

Differential diagnosis includes atrophic can-didiasis, erosive lichen planus, psoriasis, Reiter’s syndrome, lupus erythematosus, leukoplakia, allergic or hypersensitivity reaction to a food, flavoring or drug12. Diagnosis of GS is usually made without biopsy. Medical history and physi-cal examination may be helpful in differentiating GS from the oral lesions of psoriasis and Reiter’s syndrome, which have similar histological chang-es1,13. Reiter’s syndrome characterized by the triad of conjunctivitis, urethritis, arthritis. The histopa-tological appearance of specimen from patient with geographic tongue and GS has been well described as a psoriasiform pattern. Psoriasiform lesions of the oral mucosa described by Weathers et al.14 in 1974 are characterized by psoriasiform mucositis and can be divided into three distinct clinical entities; geographic tongue, geographic stomatitis, and intraoral psoriasis. Diagnosis of GS is usually based on their clinical appearance; pattern of migration, lack of symptoms, chronic-ity of lesions3. Ralls and Warnock15 claim that GS may represent an incomplete form of either psoriasis or Reiter’s syndrome. It was also shown that geographic tongue and psoriasis are associ-ated with human leukocyte antigen HLA Cw616.

Pogrel and Cram17 reported GS occurring in 19 of 100 patients with severe relapse of cutane-ous psoriasis. Therefore, exposed skin should be examined in patients with GS. However, associa-tion between GS and psoriasis is not yet under-stood completely and may be coincidental.

The lesions tend to change location, pattern, and size within minutes to hours. The disease is characterized by exacerbation and remission peri-ods. As regards duration of exacerbation, lesions show variation. While in some patients lesions are healing within two weeks, in some patients they continue to develop for more than a year8. Lesions resolve without residual scar formation1. These may last days, months, or years. When lesions recur, they tend to appear at a new location, thus producing migration effect18. However, Weathers et al.14 have reported that all geographic lesions are not migrated and they divided these lesions two subgroup as erythema circinate perstans and erythema circinate migrans. Brooks and Balci-unas3 showed that only 34% of the patients had a history of migration of the lesions. Therefore, we have preferred using the term “geographic stoma-titis” rather than the term “migration”, which is being used up to date.

Although GS has not appeared within all geo-graphic tongue patients, almost all patients with GS, geographic tongue has been observed. Fis-sured tongue is also frequently observed among GS patients4,19. Both our cases had fissured tongue, but only one of them had geographic tongue as convenient with the literature,.

The majority of patients are asymptomatic but occasionally patients may suffer from pain, itching or burning sensation. Patients should be told to avoid topical factors that exacerbate their symptoms, such as smoking, very hot, spicy, acidic foods, and dried, salty nuts7,12. Symptom-atic treatment may include mouth rinsing with topical anesthetics, antihistamines, steroids or combination of these. Anemia should be ruled out with the appropriate laboratory tests. Re-placement of iron or zinc, if these elements are deficient, may help relief of symptoms20. The

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disease may cause anxiety and fear of cancer5. Patients who have a fear of cancer may refer to medical help for control of emotional stress.

CONClUSION

Geographic lesions have been recorded for a long time but our knowledge of this condition is still inadequate. The reason of this insufficiency may be harmless nature of the vast majority of lesions. Dental practitioners should be aware that geographic lesions are not only confined to the tongue, but also can affect occasionally oral mucosa elsewhere in the mouth.

REFERENCES

1. Lucas VS, Challacombe SJ, Morgan PR. Erythema migrans: an unusal presentation. Br Dent J 1993; 175(7): 258–259.

2. Cooke BE. Erythema migrans affecting the oral mucosa. Oral Surg Oral Med Oral Pathol 1955; 8(2): 164–167.

3. Brooks JK, Balciunas BA. Geographic stomatitis: review of the literature and report of five cases. J Am Dent Assoc 1987; 115(3): 421–424.

4. Van der Wal N, van der Kwast WA, van Dijk E, van der Waal I. Geographic stomatitis and psoriasis. Int J Oral Maxillofac Surg 1988; 17(2):106–109.

5. Littner MM, Dayan D, Gorsky M, Moskona D, Harel-Raviv M. Migratory Stomatitis. Oral Surg Oral Med Oral Pathol 1987; 63(5): 555–559.

6. Hume WJ. Geographic stomatitis: a critical review. J Dent 1975; 3(1): 25–43.

7. Zunt SL, Tomich CE. Erythema migrans-a psoriasiform lesion of the oral mucosa. J Dermatol Surg Oncol 1989; 15(10): 1067–1070.

8. Warnock GR, Correll RW, Pierce GL, Hatch CL. Multiple, shallow, circinate mucosal erosions on the soft palate and base of uvula. J Am Dent Assoc 1986; 112(4): 523–524.

9. Bouqout JE, Gundlach KK. Odd tongues. The prevalence of common tongue lesions in 23,616 white Americans over 35 years of age. Quintessence Int 1986; 17(11): 719–730.

10. Daneshpazhooh M, Moslehi H, Akhyani M, Etesami M. Tongue lesions in psoriasis: a controlled study. BMC Dermatol 2004; 4(1): 16.

11. Saprio SM, Shklar G. Stomatitis areata migrans. Oral Surg Oral Med Oral Pathol 1973; 36(1): 28–33.

12. Assimakopoulos D, Patrikakos G, Fotika C, Elisaf M. Benign migratory glossitis or geographic tongue: an enigmatic oral lesion. Am J Med 2002; 113(9): 751–755.

13. Rhyne TR, Smith SW, Minier AL. Multiple, annular, erythematous lesions of the oral mucosa. J Am Dent Assoc 1988; 116(2): 217–218.

14. Weathers DR, Baker G, Archard HO, Burkes EJ Jr. Psoriasiform lesions of the oral mucosa (with emphasis on “ectopic geographic tongue”). Oral Surg Oral Med Oral Pathol 1974; 37(6): 872–888.

15. Ralls SA, Warnock GR. Stomatitis areata migrans affecting the gingiva. Oral Surg Oral Med Oral Pathol 1985; 60(2): 197–200.

16. Gonzaga HF, Torres EA, Alchorne MM, Gerbase-Delima M. Both psoriasis and benign migratory glossitis are associated with HLA-Cw6. Br J Dermatol 1996; 135(3): 368–370.

17. Pogrel MA, Cram D. Intraoral findings in patient with psoriasis with a special reference to ectopic geographic tongue (erythema circinata). Oral Surg Oral Med Oral Pathol 1988; 66(2): 184–189.

18. Sigal MJ, Mock D. Symptomatic benign migratory glossitis: report of two cases and literature review. Pediatr Dent 1992; 14(6): 392–396.

19. Özbayrak S. Dildeki değişiklikler ve hastalıklar. Ed. Özbayrak S. Ağız hastalıkları atlası tanı kriterleri, ayırıcı tanı ve tedavi yaklaşımları. 1. Baskı. İstanbul: Quintessence Yayıncılık Ltd. Şti. 2003: 122-131.

20. Borrie F, Musthyala R, Macintyre D. Ectopic geographic tongue-a case report. Dent Update 2007; 34(2): 121–122.

CORRESPONDING ADRESS

Dr. Nursel AKKAYAHacettepe University Faculty of Dentistry Department of Oral Diagnosis and Radiology 06100 Sıhhiye-Ankara/Turkey

Phone : + 90 312 3052205 Fax : + 90 (312) 3113741 E-mail: ynursel@hacettepe.edu.tr.

Geliş Tarihi : 02.05.2008 Received Date : 02 May 2008 Kabul Tarihi : 17.07.2008 Accepted Date : 17 July 2008