|Year : 2018 | Volume
| Issue : 2 | Page : 130-133
Geographic tongue in 8-month-old monozygotic twins: Case report with review of literature
Saru Thakur, Mudita Gupta, GR Tegta, Kuldeep Verma
Department of Dermatology, Venereology and Leprosy, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India
|Date of Web Publication||26-Mar-2018|
Department of Dermatology, Venereology and Leprosy, Indira Gandhi Medical College, Shimla, Himachal Pradesh
Source of Support: None, Conflict of Interest: None
Geographical tongue is a benign inflammatory disorder of the tongue characterized by map like areas of erythema which are not constant in size, shape, or location. Positive family history may be reported in few cases, but so far there is a single case report of geographical tongue in monozygotic twins 5-year-old. We report a case of geographical tongue in monozygotic twins in infants 8-month-old, which is the first case in twins in infancy to the best of our knowledge.
Keywords: Family history, geographical tongue, monozygotic twins
|How to cite this article:|
Thakur S, Gupta M, Tegta G R, Verma K. Geographic tongue in 8-month-old monozygotic twins: Case report with review of literature. Indian J Paediatr Dermatol 2018;19:130-3
|How to cite this URL:|
Thakur S, Gupta M, Tegta G R, Verma K. Geographic tongue in 8-month-old monozygotic twins: Case report with review of literature. Indian J Paediatr Dermatol [serial online] 2018 [cited 2020 Nov 28];19:130-3. Available from: https://www.ijpd.in/text.asp?2018/19/2/130/216948
| Introduction|| |
Geographical tongue is also known by names such as benign migratory glossitis, annulus migrans, lingual erythema migrans, exfoliation linguae areata, areata stomatitis migrans, and wandering rash of the tongue. This lesion usually involves the dorsum of tongue and extends on the lateral borders. Clinically, erythematous patches with loss of papillae are seen surrounded by white hyperkeratotic borders. Usually, lesions are asymptomatic but sometimes there may be intolerance to hot, spicy food with burning sensation. Rarely, painful geographical tongue has also been described.
| Case Report|| |
An 8-month-old female child was brought to dermatology outpatient department with excessive dryness of skin, recurrent sneezing, and red colored scaly patches over face. A diagnosis of atopic dermatitis was made. The parents revealed that for 3 months, she was also developing white and red lesions on the surface of tongue which appeared off and on with varying sizes and areas of involvement of tongue. On examination, over the dorsum of tongue there were erythematous atrophic patches, which were surrounded by whitish raised hyperkeratotic borders [Figure 1]. And interestingly, her twin sister had similar erythematous oval bald patches with whitish raised borders over the dorsum of tongue [Figure 2]. Clinically, the diagnosis of geographical tongue was made based on migratory history and clinical examination. The first twin had a wavy one-dimensional (1D) pattern, while the other, had an oblate or circular pattern of geographical tongue. Both the girls did not seem to have any discomfort due to the tongue lesions. No such lesions were seen in the parents. Our patient suggested that both atopy and genetic factors may be implicated in the etiopathogenesis. As lesions were asymptomatic, no specific treatment was prescribed and parents were advised to maintain adequate oral hygiene and regular follow-up.
| Discussion|| |
Geographical tongue is a common entity with the prevalence of 1.05%–1.85% in general population. It is common in adults as compared to children. In India, geographical tongue is reported in children in 0.89%. In a study in Israel, incidence as high as 14.29% has been observed in children up to 2 years of age. It may begin in childhood and most frequently affects children 4–4½ years of age. The prevalence was found to be more in females in a study on 1540 participants, however, a statistical significance could not be established.
Exact etiopathogenesis of this disorder is not known till date. However, different etiological factors have been implicated such as allergy, asthma, atopy, psoriasis, psychosomatic factors, stress, systemic diseases such as anemia, vitamin deficiencies, gastrointestinal disorders, and hormonal influences. Syndromes associated with the disorder include Reiter's syndrome, Aarskog syndrome, Fetal hydantoin syndrome, Robinow's syndrome, and Down's syndrome. Significant coexistence of geographical tongue with fissured tongue has been reported. Smoking however has been protective against the development of geographical tongue. Patients with personal or family history of asthma, hay fever are more likely to manifest geographical pattern over tongue. In a study, on one hundred and thirty-two atopic patients, 35.7% had geographical tongue as compared to controls. Hence, suggesting that exfoliatio linguae areata is a possible manifestation of atopy.
Seiden and Curland in 2015 have tried to explain the etiopathogenesis on the basis of laws of physics. The appearance of tongue in erythema migrans has been likened to well-known phenomenon a seen in excitable media such as forest fires, cardiac muscle, and chemically driven reaction-diffusion systems. Their results shed light on the evolution of inflammation which is typically anisotropic on the dorsum of the tongue. Three stages of evolution of inflammation stressed on were: resting state (healed epithelium), excited state (highly inflamed epithelium), and recovering state (healing epithelium).
Genetic factors have also been considered in etiology as seen from positive family history in some reports. Significantly higher prevalence of geographical tongue has been seen in parents and siblings of affected individuals, suggesting a polygenic mode of inheritance. Recently, geographical tongue has been reported in 5-year-old monozygotic twins supporting the role of genetic factors in the etiopathogenesis. Human leukocyte antigen (HLA) associations have also been seen in past. An increased frequency of HLA-B15 was found in patients with geographical tongue as compared to controls. Furthermore, increased incidence in HLA DR5, DRW6, and decreased of HLA DR2 have been observed in these patients. In our cases, genetics and atopy are probably implicated to have an etiological role.
The dorsal surface of tongue is normally covered with tuft like projections called lingual papillae which are associated with taste buds. This gives the tongue a pinkish color and an irregular surface. Geographical tongue is characterized by areas of loss of papillae leaving erythematous atrophic bald patches over the dorsum of the tongue surrounded by well demarcated, slightly raised, white, yellow or greyish serpiginous borders. When observed over a period of hours or days, the bald patches may change in size and shape and involve other areas of the tongue or disappear altogether for varying period. The term “geographic” refers to the resemblance of dorsum of tongue to an aerial view of areas of land masses and ocean on a map. Site most commonly involved is the dorsum of the tongue however sites such as labial mucosa, gingiva, ventral surface of tongue and palate may be involved.
Three patterns of geographical tongue with their evolution have been explained. These include oblate, spiral, and wavy 1D pattern , [Figure 3]. The long axis of the lesions lie approximately parallel to the tongue symmetry axis. Most of the patients are asymptomatic but few may complain of burning sensation, discomfort, sensitivity to hot, and spicy food. Significant oral pain affecting daily activity, eating and sleeping habits has been reported.
|Figure 3: Patterns of geographical tongue. (a) Oblong or circular pattern, (b) wavy one-dimensional pattern, and (c) spiral pattern|
Click here to view
Geographical tongue needs to be differentiated from candidiasis, psoriasis, leukoplakia, lichen planus, Reiter's syndrome, herpes simplex, systemic lupus erythematosus, and drug rash. Solitary geographical tongue lesion may resemble squamous cell carcinoma of tongue. However, it will lack the ulceration, erythroplakia, indurated border seen in carcinoma. Classical lesion with migratory nature, white serpiginous borders is characteristic of geographic tongue.
The clinical diagnosis is confirmatory based on migratory history and clinical examination and histopathology is not always required. When performed, the white areas in geographical tongue show subepithelial mononuclear infiltrates predominantly neutrophilic with abundant exocytosis forming microabscesses. The denuded areas show mononuclear subepithelial infiltrate, suprapapillary hypertrophy with vascular ectasia. Electron microscopy reveals complete loss of filiform papillae in erythematous area and necrotic flaking cells in white borders.
Reassurance is all that is required in most of the cases as the condition is benign and self-limiting as seen in previous cases  and in our patient. However, patients should be motivated to maintain adequate oral hygiene and clean the surface of the tongue with a brush to remove all debris, which may act as irritants. Our patients were also asymptomatic hence only oral hygiene was advised. Treatment may be required only in symptomatic cases. The treatment options include topical steroids, topical tretinoin, topical anesthetic agents, vitamin A therapy, antihistamines, analytics, steroids, sodium bicarbonate in water and tacrolimus. Successful treatment with cyclosporine  has also been reported in resistant cases.
| Conclusion|| |
Geographical tongue is a matter of concern to the patient due to its appearance. However, it is a completely benign and self-limiting condition. Only a single case has been reported in literature about geographical tongue in twins to the best of our knowledge. Hence, this case is being reported as a rare occurrence and only second case report of geographical tongue in twins and first case report in twins in infancy.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Pass B, Brown RS, Childers EL. Geographic tongue: Literature review and case reports. Dent Today 2005;24:54, 56-7.
Desai VD, Phore S, Baghla P. Asymptomatic reversible lesion on tongue: A case series in pediatric patients. Arch Med Health Sci 2015;3:113-6. [Full text]
Dayal S, Sahu P, Jain VK. Painful geographical tongue in two siblings. Indian J Pediatr Dermatol 2016;17:75-6.
Shulman JD. Prevalence of oral mucosal lesions in children and youths in the USA. Int J Paediatr Dent 2005;15:89-97.
Rahamimoff P, Muhsam HV. Some observations on 1246 cases of geographic tongue: The association between geographic tongue, seborrheic dermatitis, and spasmodic bronchitis; transition of geographic tongue to fissured tongue. AMA J Dis Child 1957;93:519-25.
Shah N, Kariya P, Dave B, Thomas P. Geographic tongue: A case report with review of literature. Adv Hum Biol 2016;6:142-4. [Full text]
Khozeimeh F, Rasti G. The prevalence of tongue abnormalities among the school children in Borazjan, Iran. Dent Res J 2006;3:1-6.
Goregen M, Melikoglu M, Miloglu O, Erdem T. Predisposition of allergy in patients with benign migratory glossitis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;110:470-4.
Marks R, Simons MJ. Geographic tongue – A manifestation of atopy. Br J Dermatol 1979;101:159-62.
Singh S, Nivash S, Mann BK. Matched case-control study to examine association of psoriasis and migratory glossitis in India. Indian J Dermatol Venereol Leprol 2013;79:59-64.
] [Full text]
Ebrahimi H, Pourshahidi S, Tadbir AA, Shyan SB. The relationship between geographical tongue and stress. IRCMJ 2010;12:313-5.
Nandini DB, Bhavana SB, Deepak BS, Ashwini R. Paediatric geographic tongue: A case report, review and recent updates. J Clin Diagn Res 2016;10:ZE05-9.
Nanda A, Kaur S, Bhakoo ON, Kapoor MM, Kanwar AJ. Fetal hydantoin syndrome: A case report. Pediatr Dermatol 1989;6:130-3.
Cerqueira DF, de Souza IP. Orofacial manifestations of Robinow's syndrome: A case report in a pediatric patient. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:353-7.
Erics M, Balci S, Atakan N. Dermatological manifestations of 71 Down syndrome children admitted to a clinical genetics unit. Clin Genet 1996;37:872-88.
Jainkittivong A, Langlais RP. Geographic tongue: Clinical characteristics of 188 cases. J Contemp Dent Pract 2005;6:123-35.
Miloglu O, Göregen M, Akgül HM, Acemoglu H. The prevalence and risk factors associated with benign migratory glossitis lesions in 7619 Turkish dental outpatients. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;107:e29-33.
Ullmann W. Correlation between exfoliatio linguae areata and atopy. Hautarzt 1982;32:629-31.
Seiden G, Curland S. The tongue as an excitable medium. New J Phys 2015;17:1-8.
Shekhar MG. Geographic tongue in monozygotic twins. J Clin Diagn Res 2014;8:ZD01-2.
Eidelman E, Chosack A, Cohen T. Scrotal tongue and geographic tongue: Polygenic and associated traits. Oral Surg Oral Med Oral Pathol 1976;42:591-6.
Marks R, Taitt B. HLA antigens in geographic tongue. Tissue Antigens 1980;15:60-2.
Fenerli A, Papanicolaou S, Papanicolaou M, Laskaris G. Histocompatibility antigens and geographic tongue. Oral Surg Oral Med Oral Pathol 1993;76:476-9.
Bajaj P, Kapoor C, Garg D, Mohammed PK, Sabharwal R, Vaidya S. Geographical tongue in a 6 year old child: A case report with review of literature. Dent J 2013;1:112-7.
Sigal M, Mock D. Symptomatic benign migratory glossitis: Report of two cases and literature review. Paediatr Dent 1992;14:392-6.
Picciani BL, Domingos TA, Teixeira-Souza T, Santos Vde C, Gonzaga HF, Cardoso-Oliveira J, et al.
Geographic tongue and psoriasis: Clinical, histopathological, immunohistochemical and genetic correlation – A literature review. An Bras Dermatol 2016;91:410-21.
Shahzad M, Sattar A, Ali SM. Geographical tongue: Case report and literature review. Pak Oral Dent J 2014;34:409-10.
Helfman RJ. The treatment of geographic tongue with topical retin – A solution. Cutis 1975;50:41-6.
Ishibashi M, Tojo G, Watanabe M, Tamabuchi T, Masu T, Aiba S. Geographic tongue treated with topical tacrolimus. J Dermatol Case Rep 2010;4:57-9.
Abe M, Sogabe Y, Syuto T, Ishibuchi H, Yokoyama Y, Ishikawa O. Successful treatment with cyclosporin administration for persistent benign migratory glossitis. J Dermatol 2007;34:340-3.
[Figure 1], [Figure 2], [Figure 3]