OLGU RAPORU (Case Report) Hacettepe Diş Hekimliği Fakültesi Dergisi Cilt: 32, Sayı: 2, Sayfa: 73-78, 2008

Geographic

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

ABSTRACT ÖZET

Geographic lesions may also appear, though Coğrafik dil lezyonları oral kavitenin diğer muko- rarely, on other mucosal areas of the oral cavity. This zal bölgelerinde de nadiren görülebilir. Bu durum type of lesion is called as geographic stomatitis. The coğrafik stomatit olarak adlandırılmaktadır. Nedeni cause is unknown; yet, emotional stress, nutritional bilinmemektedir. Ancak emosyonel stres, beslenme deficiencies, and hereditary factors are suggested. bozuklukları ve herediter faktörlerin neden olabile- Clinically, it occurs as red annular patches of the labial ceği ileri sürülmektedir. Klinik olarak labial ya da or buccal mucosa, soft ; rarely gingiva and floor bukkal mukozada, nadiren gingiva ve ağız tabanında of mouth. There are mild erosions of the mucosa on kırmızı, halka şeklinde yamalar tarzında görülürler. these lesions, which are frequently multiple and well- Sıklıkla çok sayıda ve belirgin sınırları olan bu lezyon circumscribed. Characteristically lesions change their bölgelerinde mukozanın hafif erozyonuna rastlanır. shape and location. The condition is usually asymp- tomatic. When symptomatic, palliative treatment is Karakteristik olarak lezyonlar şekil ve yer değiştirir. recommended. Bu durum genellikle asemptomatiktir. Semptomatik olduğunda palyatif tedavi önerilmektedir. The purpose of the present paper is to report two cases of geographic stomatitis and to discribe a pos- Bu makalenin amacı, iki coğrafik stomatit olgusu sible connection between this condition and patients’ sunmak ve bu durum ile hastanın emosyonel statüsü emotional status. arasındaki olası ilişkiyi tanımlamaktır.

KEYWORDS ANAHTAR KELİMELER Geographic stomatitis, migratory stomatitis, stomatitis Coğrafik stomatit, migratuar stomatit, stomatitis areata areata migrans, circinate migrans, ectopic migrans, eritema sirsinate migrans, ektopik coğrafik dil. geographic tongue.

These cases were presented in 3. Scientific Symposium of Oral Diagnosis and Dentomaxillofacial Radiology Society, 21-23 April 2006, Kemer, Antalya 74

INTRODUCTION before acquiring the typical appearance of geo- graphic lesion. Geographic tongue is a condition character- ized by of the filiform papilae on single Type 4: Geographic lesions elsewhere in the or multiple areas of the tongue in an irregular mouth, without the presence of a geographic pattern which is frequently accompanied by pe- tongue. ripheral keratosis. These geographic lesions oc- Although its etiology is unknown, many dif- cur on the dorsum and lateral borders of tongue ferent conditions, such as pustular , Re- in about 1–2 % of general population1. Similar iter’s syndrome, , stress may be associated changes may appear on other mucosal areas of with GS. The condition is more common in men the oral cavity. This uncommon condition was than women. No particular age group shows an first described in 1955 under the term “ery- increased tendency to GS7. thema migrans” by Cooke2. It is also named as The purpose of the present paper is to re- migratory stomatitis, stomatitis areata migrans, port two additional cases of GS and to describe a erythema circinate migrans, ectopic geographic possible connection between this condition and tongue, geographic stomatitis, annulus migrans, patients’ emotional status. Cooke’s disease and migratory mucositis3.

The most frequently reported sites of geo- CASE 1 graphic stomatitis (GS) are the buccal mucosa, lower labial mucosa and mucobuccal fold. In- 33 year-old woman applied to our clinic for volvement of the gingiva, alveolar mucosa, soft routine dental examination. Her medical his- palate and floor of the mouth is unusual4. tory was unremarkable. Extraoral examination revealed on her left hand. There Clinical presentation of GS is described as slightly raised, round, erythematous lesions that were no skin lesions present except the hyper- are circumscribed by well-defined whitish bor- keratosis of hand. Intraoral examination showed ders. The lesions vary in size from a few millime- multiple erythematous lesions surrounded by a ters to several centimeters in diameter5. narrow white margin on the right buccal muco- sa, mucobuccal fold, the right upper labial mu- Hume6 has made a classification of GS based cosa and the dorsum of the tongue (Figure 1–3). on its clinical distribution: The lesions varied in size and had well-defined, Type 1: Lesions on the dorsum, lateral bor- white, slightly raised, circinate borders. No evi- ders and tip of tongue with possible extension dence of vesicle formation was noted. There was to undersurface. The lesions may migrate with fissuring of the dorsum of the tongue. She was time and show both active and remission phases unaware of mucosal lesions but she was suffering (geographic tongue, without geographic lesions from burning of tongue after consuming hot or elsewhere in the mouth). spicy foods. Patient expressed that she has been Type 2: Geographic tongue, accompanied by under stressful condition during the last three geographic lesions elsewhere in the mouth. months because of her mother’s health problems Type 3: Atypical tongue lesions whether or and intensity of symptoms has increased during not accompanied by lesions elsewhere in the this period. Patient was referred to Dermatology mouth. Atypical tongue lesions consist of two Clinic for the evaluation of lesions on her hand forms: A- Fixed forms; one or two areas of tongue and possible relationship between skin and oral are affected but movement is not observed. In- mucosal lesions. Skin lesions of her hand were stead, they disappear only to recur after a pe- diagnosed as “contact dermatitis”. Therefore, it riod of time at the same area. B- Abortive forms; was concluded that skin and oral mucosal lesions these start as yellow-white patches but disappear were not related. 75

FIGURE 3

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

Oral and acute atrophic candidia- FIGURE 1 sis were considered as differential diagnoses. Multiple, well-demarcated erythematous lesions of geographic Lichen planus could be excluded as the clinical stomatitis on the maxillary labial mucosa in Case 1. appearance was not consistent with this condi- tion. A smear from the lesions of for the evaluation of was nega- tive. Her dermatologist decided to take , 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- FIGURE 2 gratory behavior of these asymptomatic lesions. Involvement of the mandibular mucobuccal fold with discrete, The clinical diagnosis of geographic stomatitis annular lesions in Case 1. was made. 76

CASE 2 sions were migrating, they were similar in sight. Due to all these characteristic attributions, no 43 year-old man had applied to our clinic biopsy was taken and the disease diagnosed as for toothache. In his medical history, patient ex- “geographic stomatitis”. Nonetheless, the patient pressed that he has been under medical treat- was directed to internal medicine department. ment until he was 19 years old, due to congenital Due to detection of hypercalsuri in urinalysis hypothyroidism. Currently he is not using any examination the patient is directed to urology . He has been undergoing psychologi- department. In his abdomen CT and renal USI, cal treatment since 1993 because of over ner- renal cortical cysts were seen in both of his kid- vousness, instability to control himself and prob- neys. Suspected of internal hernia in the right lematic relations in the working environment. hemi region of the abdomen, radiology depart- Extraoral examination was unremarkable. ment requested intestinal passage examination. During intraoral examination it was detected that Results were normal. he had and there were bilater- Patient monitored for a year with regular ally, slightly raised, erythematous lesions on the periods. It was observed that the intensity and buccal mucosa (Figure 4). The lesions had white, localization of the lesions changed and this situa- circinate borders. In these regions, there was no tion was attributed to his emotional stress. symptom in the burning or pain characteristics. Also the patient expressed that he was unaware DISCUSSION of this condition. Result of the smear taken from the lesion region in order to detect possible exis- In the literature limited numbers of GS cases tence of candida albicans showed normal flora. were reported, however some of the researchers Full blood test results, vitamin B12 and folate lev- think that its real incidence is more than expected els were normal. 15 days later when he applied since the disease mostly proceed in asymptom- to clinic with these results, it was seen that there atic nature and easy to diagnose when encoun- were same type of lesions in the nearby regions tered together with geographic tongue8. Never- of lower labial mucosa and comissura labiorum. theless, Bouquot and Gundlach9 reviewed data However, mucosa at the sites of previous lesions on 23616 white American and found no patient was completely normal (Figure 5). Although le- with geographic stomatitis. Daneshpazhooh et.

FIGURE 4 FIGURE 5

Geographic stomatitis appearing as erythematous lesions on Multiple small lesions of the geographic stomatitis on the the buccal mucosa in Case 2. mandibular labial mucosa in Case 2. 77 al.10 denote that this lesion is rarely encountered Pogrel and Cram17 reported GS occurring in 19 in the population. of 100 patients with severe relapse of cutane- Etiology of GS is not understood complete- ous psoriasis. Therefore, exposed skin should be ly. There is no certain difference between geo- examined in patients with GS. However, associa- graphic tongue lesions and GS but GS defined tion between GS and psoriasis is not yet under- as the type of these lesions encountered in the stood completely and may be coincidental. oral mucosa. Geographic tongue can be related The lesions tend to change location, pattern, to atopic conditions (hay fever, eczema, and and size within minutes to hours. The disease is asthma), reactive bronchitis, allergy, hormonal characterized by exacerbation and remission peri- disturbances (pregnancy, juvenile mel- ods. As regards duration of exacerbation, lesions litus), nutrition deficiencies, , gastroin- show variation. While in some patients lesions are testinal anomalies, infectious agents and lithium healing within two weeks, in some patients they therapy. Psychological status as an etiological continue to develop for more than a year8. Lesions factor has considerable effects11. Both of these resolve without residual scar formation1. These patients had psychological problems. We have may last days, months, or years. When lesions observed that alteration in the intensity of the le- recur, they tend to appear at a new location, thus sions was correlated with the patient’s emotional producing migration effect18. However, Weathers status. Patients having a psychological treatment et al.14 have reported that all geographic lesions supported this view. are not migrated and they divided these lesions two subgroup as erythema circinate perstans and includes atrophic can- erythema circinate migrans. Brooks and Balci- didiasis, erosive lichen planus, psoriasis, Reiter’s unas3 showed that only 34% of the patients had a syndrome, , , history of migration of the lesions. Therefore, we allergic or hypersensitivity reaction to a food, have preferred using the term “geographic stoma- flavoring or drug12. Diagnosis of GS is usually titis” rather than the term “migration”, which is made without biopsy. Medical history and physi- being used up to date. cal examination may be helpful in differentiating GS from the oral lesions of psoriasis and Reiter’s Although GS has not appeared within all geo- syndrome, which have similar histological chang- graphic tongue patients, almost all patients with es1,13. Reiter’s syndrome characterized by the triad GS, geographic tongue has been observed. Fis- of , , . The histopa- sured tongue is also frequently observed among 4,19 tological appearance of specimen from patient GS patients . Both our cases had fissured with geographic tongue and GS has been well tongue, but only one of them had geographic described as a psoriasiform pattern. Psoriasiform tongue as convenient with the literature,. lesions of the oral mucosa described by Weathers The majority of patients are asymptomatic et al.14 in 1974 are characterized by psoriasiform but occasionally patients may suffer from pain, mucositis and can be divided into three distinct itching or burning sensation. Patients should be clinical entities; geographic tongue, geographic told to avoid topical factors that exacerbate their stomatitis, and intraoral psoriasis. Diagnosis of symptoms, such as smoking, very hot, spicy, GS is usually based on their clinical appearance; acidic foods, and dried, salty nuts7,12. Symptom- pattern of migration, lack of symptoms, chronic- atic treatment may include mouth rinsing with ity of lesions3. Ralls and Warnock15 claim that topical anesthetics, , steroids or GS may represent an incomplete form of either combination of these. Anemia should be ruled psoriasis or Reiter’s syndrome. It was also shown out with the appropriate laboratory tests. Re- that geographic tongue and psoriasis are associ- placement of iron or zinc, if these elements are ated with HLA Cw616. deficient, may help relief of symptoms20. The 78 disease may cause anxiety and fear of cancer5. 8. Warnock GR, Correll RW, Pierce GL, Hatch CL. Multiple, shallow, circinate mucosal erosions on the and Patients who have a fear of cancer may refer to base of uvula. J Am Dent Assoc 1986; 112(4): 523–524. medical help for control of emotional stress. 9. Bouqout JE, Gundlach KK. Odd . The prevalence of common tongue lesions in 23,616 white Americans over Conclusion 35 years of age. Quintessence Int 1986; 17(11): 719–730. 10. Daneshpazhooh M, Moslehi H, Akhyani M, Etesami M. Geographic lesions have been recorded for a Tongue lesions in psoriasis: a controlled study. BMC long time but our knowledge of this condition is Dermatol 2004; 4(1): 16. still inadequate. The reason of this insufficiency 11. Saprio SM, Shklar G. Stomatitis areata migrans. Oral Surg may be harmless nature of the vast majority of Oral Med Oral Pathol 1973; 36(1): 28–33. lesions. Dental practitioners should be aware 12. Assimakopoulos D, Patrikakos G, Fotika C, Elisaf M. that geographic lesions are not only confined to Benign migratory or geographic tongue: an enigmatic oral lesion. Am J Med 2002; 113(9): 751–755. the tongue, but also can affect occasionally oral 13. Rhyne TR, Smith SW, Minier AL. 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Geliş Tarihi : 02.05.2008 Received Date : 02 May 2008 Kabul Tarihi : 17.07.2008 Accepted Date : 17 July 2008

CORRESPONDING ADRESS

Dr. Nursel AKKAYA Hacettepe University Faculty of Dentistry Department of Oral Diagnosis and Radiology 06100 Sıhhiye-Ankara/Turkey Phone : + 90 312 3052205 Fax : + 90 (312) 3113741 E-mail: [email protected].