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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 19  |  Issue : 2  |  Page : 130-133

Geographic tongue in 8-month-old monozygotic twins: Case report with review of literature


Department of Dermatology, Venereology and Leprosy, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India

Date of Web Publication26-Mar-2018

Correspondence Address:
Mudita Gupta
Department of Dermatology, Venereology and Leprosy, Indira Gandhi Medical College, Shimla, Himachal Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.IJPD_8_17

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  Abstract 


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 2019 Dec 6];19:130-3. Available from: http://www.ijpd.in/text.asp?2018/19/2/130/216948




  Introduction Top


Geographical tongue is also known by names such as benign migratory glossitis, annulus migrans, lingual erythema migrans, exfoliation linguae areata, areata stomatitis migrans,[1] and wandering rash of the tongue. This lesion usually involves the dorsum of tongue and extends on the lateral borders.[2] 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.[3]


  Case Report Top


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.
Figure 1: First twin showing geographical tongue

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Figure 2: Second twin showing geographical tongue

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  Discussion Top


Epidemiology

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.[4] In India, geographical tongue is reported in children in 0.89%.[2] In a study in Israel, incidence as high as 14.29% has been observed in children up to 2 years of age.[5] It may begin in childhood and most frequently affects children 4–4½ years of age.[6] The prevalence was found to be more in females in a study on 1540 participants, however, a statistical significance could not be established.[7]

Etiopathogenesis

Exact etiopathogenesis of this disorder is not known till date. However, different etiological factors have been implicated such as allergy,[8] asthma, atopy,[9] psoriasis,[10] psychosomatic factors, stress,[11] systemic diseases such as anemia, vitamin deficiencies, gastrointestinal disorders, and hormonal influences.[12] Syndromes associated with the disorder include Reiter's syndrome, Aarskog syndrome, Fetal hydantoin syndrome,[13] Robinow's syndrome,[14] and Down's syndrome.[15] Significant coexistence of geographical tongue with fissured tongue has been reported.[16] Smoking however has been protective against the development of geographical tongue.[17] 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.[18]

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).[19]

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.[20] Recently, geographical tongue has been reported in 5-year-old monozygotic twins supporting the role of genetic factors in the etiopathogenesis.[21] 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.[22] Furthermore, increased incidence in HLA DR5, DRW6, and decreased of HLA DR2 have been observed in these patients.[23] In our cases, genetics and atopy are probably implicated to have an etiological role.

Clinical features

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.[6] 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.[24] 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.[1]

Three patterns of geographical tongue with their evolution have been explained. These include oblate, spiral, and wavy 1D pattern [12],[19] [Figure 3]. The long axis of the lesions lie approximately parallel to the tongue symmetry axis.[19] 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.[25]
Figure 3: Patterns of geographical tongue. (a) Oblong or circular pattern, (b) wavy one-dimensional pattern, and (c) spiral pattern

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Differential diagnosis

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.[1]

Diagnosis

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.[26]

Treatment

Reassurance is all that is required in most of the cases as the condition is benign and self-limiting as seen in previous cases [2] 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.[27] 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,[28] topical anesthetic agents, vitamin A therapy, antihistamines, analytics, steroids, sodium bicarbonate in water and tacrolimus.[29] Successful treatment with cyclosporine [30] has also been reported in resistant cases.


  Conclusion Top


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

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

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2.
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29.
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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.  Back to cited text no. 30
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    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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