Medical Hypotheses 82 (2014) 709–712

Contents lists available at ScienceDirect

Medical Hypotheses journal homepage: www.elsevier.com/locate/mehy

Fissured tongue: A sign of tongue edema? Jaana Järvinen a, Jopi J.W. Mikkonen b,c, Arja M. Kullaa d,e,⇑ a

Municipal Health Centre of Kangasniemi, Kangasniemi, Finland SIB Labs, University of Eastern Finland, Kuopio, Finland c Department of Applied Physics, Faculty of Science and Forestry, University of Eastern Finland, Kuopio, Finland d Department of Diagnostics and Oral Medicine, Faculty of Medicine, University of Oulu, Oulu, Finland e Educational Dental Clinic, Health Centre of Oulu, Oulu, Finland b

a r t i c l e

i n f o

Article history: Received 5 November 2013 Accepted 5 March 2014

a b s t r a c t Fissured tongue (FT) is a condition frequently seen in the general population. Clinically, FT is characterized by grooves that vary in depth and are noted along the dorsal and/or dorsolateral aspects of the tongue. Furthermore, FT presents many enlarged, smooth filiform papillae and subepithelial inflammatory infiltration. Despite of many studies, the etiology of FT remains obscure. FT is believed to be a congenital anomaly associated with several disorders and with geographic tongue (GT). We hypothesize that FT is not a congenital anomaly, and FT with swollen filiform papillae may represent edema in the subepithelial tissue of the tongue. According to the literature, the difference in prevalence among different age groups indicates that FT is not a congenital disorder. FT appears to occur more commonly in adults, and it is very rare or not at all in children younger than 10 years old. An association between FT and GT is well established in the literature, supporting the results of previous authors suggesting that FT might be a consequence of GT. The most remarkable finding in the region of swollen papillae of FT samples has been the subepithelial infiltrates of polymorphonuclear leucocytes and lymphocytes causing the subepithelial edema. The clinically visible grooves and large edematic papillae clustered on the region of the fissures might be caused by the inflammation and edema underneath the epithelium. In the future, FT and GT must be researched together as two different entities of the same disease so that GT is a prestage of FT. The diagnosis of FT must be taken to consideration whether the tongue surface have smooth and swollen papillae or normal-appearing filiform papillae. Ó 2014 Elsevier Ltd. All rights reserved.

Introduction Fissured tongue (FT), also known as lingua fissurata, plicated tongue, scrotal tongue and grooved tongue, is a condition frequently seen in the general population. FT is believed to be a congenital anomaly. Clinically, it is characterized by grooves that vary in depth and are noted along the dorsal and/or dorsolateral aspects of the tongue [1]. Furthermore, FT presents many enlarged, smooth papillae [2–4], which are filiform papillae without hairs seen by scanning electron microscopy [5]. Despite of many studies, the etiology of FT remains obscure, but several conditions associated with FT have been reported including psoriasis [6–11], orofacial granulomatosis [12,13], pernicious Abbreviations: FT, fissured tongue; GT, geographic tongue; MRS, Melkersson– Rosenthal syndrome. ⇑ Corresponding author. Address: Department of Diagnostics and Oral Medicine, Institute of Dentistry, Faculty of Medicine, University of Oulu, Aapistie 3, SF-90220 Oulu, Finland. Tel.: +358 401426319. E-mail address: arja.kullaa@oulu.fi (A.M. Kullaa). http://dx.doi.org/10.1016/j.mehy.2014.03.010 0306-9877/Ó 2014 Elsevier Ltd. All rights reserved.

anemia [14], low serum levels of vitamin A [15], down syndrome [16,17], diabetes mellitus [18] and certain autoimmune diseases [19]. Furthermore, FT is one of the three symptoms of Melkersson–Rosenthal syndrome (MRS) [20,21]. Our review of scientific literature found many studies demonstrating the association between FT and geographic tongue (GT) which is a common immunological tongue disease of unknown etiology [9,22–24]. Furthermore, it is suggested a genetic basis for the development of these two tongue disorders [25–27]. In this study, we hypothesize that FT is not a congenital anomaly, but it is a sequel of GT. Furthermore, FT with swollen papillae may represent edema in the subepithelial tissue of the tongue. Our understanding of this is based on the literature review, our own investigations and long-term clinical follow-up. Diagnosis and clinical appearance of FT The clinical diagnosis of FT is based on deep grooves or fissures on the dorsal and lateral surfaces of the tongue. At present, there is

710

J. Järvinen et al. / Medical Hypotheses 82 (2014) 709–712

Fig. 1. (A) Clinically healthy tongue contains filiform papillae interspersed with hairs and smooth fungiform papillae among them. (B) Fissured tongue with enlarged papillae clustering on the region of the fissures along the dorsolateral aspects of the tongue. (C) The dorsal surface of FT contains enlarged smooth papillae.

no consensus regarding the establishment of more universal criteria for the diagnosis of FT. Review of the literature faces considerable diagnostic problems because of the absence of standard protocols. Because the clinical appearance of FT and the pattern of grooves vary widely, it is difficult to estimate the degree of fissuring and the extent to which fissuring must be present before a tongue can actually be called fissured. Two main types of FT have been found clinically: fissures cover the whole surface of the dorsal tongue or they localized on the dorsolateral part of the tongue. In some investigations, attention has been paid to the varied structure of the papillae and the fissures have been combined with unusually large swollen papillae [2–5]. We focused on the papillary structure of FT tongues, and indeed, the enlargement of lingual papillae of the tongue is a main clinical feature of FT (see Fig. 1). The variation in the anatomy of the tongue papillae and especially the size of papillae may itself be the diagnostic criteria of FT. In our own investigations, we have used the following criteria: (a) healthy tongue with filiform papillae (Fig. 1A) and (b) fissured tongue with grooves and smooth-surfaced papillae, which are clustered on the region of the fissures (Figs. 1B, C and 2A). Because the papillary topography and morphology of the tongue is complex, FT may be misdiagnosed in many studies. Some tongue entities as ‘‘Eruptive lingual papillitis’’ [28,29], ‘‘fungiform papillary glossitis’’ [30] and ‘‘chronic lingual papulosis’’ [31] with swollen lingual papillae have been documented in recent years. We believe that almost all of those entities may be classified as fissured tongue.

Epidemiology FT is a common oral lesion occurring in approximately 5–6% of the general population, although the prevalence has been reported to vary from 1% to 42.8% [1,32–35]. FT rarely or not at all occurs before the age of 4 years and has been observed in only 0–2.11% of children younger than 10 years old [36–40]. It has been shown clearly that the prevalence of FT increases with age [1,36]. On the other hand, GT starts in childhood, and it is one of the most common conditions of the oral mucosa observed in children [39,40]. Consequently, the prevalence found for FT varies widely among research groups, like students, dental outpatients, or special clinic referrals. Secondly, the clinical diagnosis of FT may be different and this could be explained by a wide range of differences in sampling and diagnosis as mentioned above.

Histopathology While the true etiology of FT remains unknown, the histopathology of this disorder is different from that of the healthy tongue. The pathophysiological characteristics of FT include intraepithelial and subepithelial infiltrates of polymorphonuclear leucocytes and lymphocytes [4]. Furthermore, clear histoquantitative differences in the epithelium, connective tissue and uppermost muscles layers have been found between FT and healthy tongue samples, suggesting that the subepithelial connective tissues of FT may also differ from the healthy tongue [41]. Inflammation may cause edema, which could explain the increase in the thickness of subepithelial connective tissue. Furthermore, inflammation may induce breaking of the muscle bundles. The inflammatory infiltration in the FT samples is most prominent in the subepithelial connective tissue layers [4], whereas GT presents with edema, particularly within the epithelium [42]. These findings support the results of previous authors suggesting that LF might be a consequence of GT [15,42–45]. FT contains several prominent papillae of various sizes, which are visible with scanning electron microscopy (Fig. 2A and B). These large papillae are filiform papillae without taste buds [5]. The association of smooth and swollen filiform papillae with inflammation is present in this condition [4,41]. Etiology and treatment Scientists have long searched for what is causing FT, delving deeply into epidemiology. Numerous etiological factors, however, have been proposed for FT. FT is suggested to be inherited because the condition is seen clustering in families who are affected. Melkersson–Rosenthal syndrome is a complex neuromucocutaneous disorder of unknown etiology that is characterized by orofacial edema and perivascular lymphocytic infiltrates [20]. FT was seen in 20–40% of MRS patients [21]. The literature suggests that both FT and geographic tongue (GT) are relatively frequent in patients suffering from psoriasis [6–11]. Some reports in the literature indicate that GT may relate to immunological diseases and allergic conditions [37,46,47]. Effective treatment for FT is not known, as its etiology has not been well elucidated. Indeed, FT may be a reversible entity, because some cases have been improved. Our previous observations indicate that there is a deficiency in the defence mechanisms of patients suffering from FT and GT [48]. However, no definitive medication or therapy is suggested for FT. D’Erme and colleques [49]

J. Järvinen et al. / Medical Hypotheses 82 (2014) 709–712

711

Fig. 2. (A) Fissures and smooth, enlarged papillae are visible on the lateral border of the tongue, whereas the middle of the tongue surface looks like normal. (B) Deep fissures separate enlarged rough papillae of fissured tongue; scanning electron microscopic view (bar = 1 mm).

Fig. 3. (A) Patient with pernicious anemia has fissured tongue with smooth enlarged papillae. (B) Regeneration of filiform papillae on the tongue after treatment with parenteral doses of cobalamin.

reported a case of a patient presenting an important improvement of his FT paralleling the improvement of the cutaneous lesions with infliximab. The improvement of FT is seen in a patient with pernicious anaemia with the treatment B12-vitamin (Fig 3A and B). So, some underlying hematinic deficiencies may be associated with FT.

Hypothesis According to the literature, the difference in prevalence among different age groups indicates that FT is not a congenital disorder. FT appears to occur more commonly in adults, and it is very rare or not at all in children younger than 10 years old. An association between FT and GT is well established in the literature, supporting the results of previous authors suggesting that FT might be a consequence of GT [15,42–45]. In our clinical followup (unpublished), we have observed that GT sometimes turns to FT. The most remarkable finding in the region of swollen papillae of FT samples has been the subepithelial edema [4,36]. The clinically visible fissures and large edematic papillae of the FT samples might be caused by the edema underneath the epithelium. Such edematic changes are typical for orofacial granulomatosis and for Melkersson–Rosenthal syndrome, and it is therefore possible that the development of FT is mediated by the same types of mechanisms as the other lesions found in these conditions. The destruction of the connective tissue of FT is ultimately resulted the formation of furrows and swollen papillae. The

uppermost muscle cells are destroyed, and the mesenchymal cells can penetrate between the uppermost muscle cells [36]. The subepithelial connective tissue breakdown is most likely caused by the pressure of inflammatory edema between the cells. The destruction of the muscle cells could also be due to the long-standing inflammation or to pressure from the edematic mesenchymal tissues. The destruction of the inflamed tongue mucosa could also be explained by the fact that the dorsal tongue mucosa is very thin and that there is no submucosa at all. Marks and Czarny [47] suggested that geographic tongue (GT) often occurs in patients who have a tendency to develop recurrent acute inflammatory diseases, such as asthma or rhinitis (either atopic or not), on surfaces in contact with the external environment. Furthermore, fungiform papillary glossitis with inflammatory swollen papillae has been described as an atopic disease [30]. Zargari [9] found that nail and genital lesions were associated with the presence of tongue lesions. Similarly, the edema may be relevant to other disease entities associated with FT, including diabetes mellitus, vitamin B deficiency, gastrointestinal diseases and autoimmune diseases. Discussion The actual mechanism causing GT and FT is unknown and our hypothesis regarding an inflammatory etiology needs to be study further. The association between these two disease entities can be understood through the histopathological analysis of FT and

712

J. Järvinen et al. / Medical Hypotheses 82 (2014) 709–712

GT tissue samples. However, further studies will be needed to clarify the molecular mechanism underlying the progression of these two disease states. In conclusion, FT and GT must be researched together as two different entities of the same disease so that GT is a prestage of FT. More thorough multicenter studies are recommended to evaluate the clinical significance of FT diagnosed with the criteria presented in this study. The diagnosis of FT must be taken to consideration whether the tongue surface have smooth and swollen papillae or normal-appearing filiform papillae. Conflict of interest None declared. References [1] Axell T. A prevalence study of oral mucosal lesions in an adult Swedish population. Odontol Revy 1976;27:1–103. [2] Aboyans V, Ghaemmaghami A. The incidence of fissured tongue among 4,009 Iranian dental outpatients. Oral Surg 1973;36:34–8. [3] Kullaa-Mikkonen A, Sorvari T, Kotilainen R. Morphological variations on the dorsal surface of the human tongue. Proc Finn Dent Soc 1985;81:104–10. [4] Kullaa-Mikkonen A, Sorvari T. Lingua fissurata. A clinical, stereomicroscopic and histopathological study. Int J Oral Maxillofac Surg 1986;94:50–6. [5] Kullaa-Mikkonen A, Sorvari T. A scanning electron microscopic study of fissured tongue. J Oral Pathol 1986;15:93–7. [6] Morris LF, Phillips CM, Binnie WH, Sander HM, Silverman AK, Menter MA. Oral lesions in patients with psoriasis: a controlled study. Cutis 1992;49:339–44. [7] Ulmansky M, Michelle R, Azaz B. Oral psoriasis: report of six new cases. J Oral Pathol Med 1995;24:42–5. [8] Bruce AJ, Rogers RS. Oral psoriasis. Dermatol Clin 2003;21:99–104. [9] Zargari O. The prevalence and significance of fissured tongue and geographical tongue in psoriatic patients. Clin Dermatol 2006;31:192–5. [10] Hernández-Pérez F, Jaimes-Aveldafiez A, Urquizo-Ruvalcaba ML, Diaz-Barcelot M, Irigoyen-Camacho ME, Vega-Memije ME, et al. Prevalence of oral lesions in patients with psoriasis. Med Oral Patol Oral Cir Bucal 2008;13:E703–708. [11] Costa SC, Hirota SK, Takahashi MDF, Andrade Jr H, Migliari DA. Oral lesions in 166 patients with cutaneous psoriasis: A controlled study. Med Oral Patol Oral Cir Bucal 2009;14:e371–735. [12] McCartan BE, Healy CM, McCreary CE, Flint SR, Rogers S, Toner ME. Characteristics of patients with orofacial granulomatosis. Oral Dis 2011;17:696–704. [13] Marcoval J, Vinãs M, Bordas X, Juclà A, Servitje O. Orofacial granulomatosis: clinical study of 20 patients. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;113:e12–17. [14] Hjörting-Hansen E, Bertram U. Oral aspects of pernicious anemia. Brit Dent J 1968;125:266–70. [15] Witkop CJ, Barros L. Oral and genetic studies of Chileans 1960 I. Oral anomalies. Amer J Anthrop 1963;21:15–24. [16] Daneshpazhooh M, Nazemi TM, Bigdeloo L, Yoosefi M. Mucocutaneous findings in 100 children with Down syndrome. Pediatric Dermatology 2007;24:317–21. [17] Bilgili SG, Akdeniz N, Karadag AS, Akbayram S, Calka O, Ozkol HU. Mucocutaneous disorders in children with down syndrome: case-controlled study. Genet Couns 2011;22:385–92. [18] Guggenheimer J, Moore PA, Rossie K, Myers D, Mongelluzzo MB, Block HM, et al. Insulin-dependent diabetes mellitus and oral soft tissue pathologies. I. Prevalence and characteristics of non-candidal lesions. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;89:563–9. [19] Scagliusi P, Sisto M, Lisi S, Lazzari A, D’Amore M. Hashimoto’s thyroiditis in Melkersson–Rosenthal syndrome patient: casual association or related diseases? Panminerva Medica 2008;50:255–8. [20] Ratzinger G, Sepp N, Vogetseder W, Tilg H. Cheilitis granulomatosa and Melkersson–Rosenthal syndrome: evaluation of gastrointestinal involvement and therapeutic regimens in a series of 14 patients. JEADV 2007;21:1065–70. [21] Zimmer WM, Rogers RS, Reeve CM, et al. Orofacial manifestations of Melkersson–Rosenthal syndrome. A study of 42 patients and review of 220 cases from the literature. Oral Surg Oral Med Oral Pathol 1992;74:610–9.

[22] Shulman JD, Carpenter WM. Prevalence and risk factors associated with geographic tongue among US adults. Oral Dis 2006;12:381–6. [23] 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. [24] Honarmand M, Farhad Mollashahi L, Shirzaiy M, Sehhatpour M. Geographic tongue and associated risk factors among Iranian dental patients. Iran J Public Health 2013;42:215–9. [25] Singh S, Nivash S, Mann BK. Matched case-control study to examine association of psoriasis and migratory glossitis in India. Indian J Dermatol Venerol Leprol 2013;79:59–64. [26] Kullaa-Mikkonen A. Familial study of fissured tongue. Scand J Dental Res 1988;96:366–71. [27] Guimaräes ALS, Correia-Silva JF, Diniz MG, Xavier GM, Horta MCR, Gomez RS. Investigation of functional gene polymorphisms: IL- 1B, IL-6 and TNFA in benign migratory glossitis in Brazilian individuals. J Oral Pathol Med 2007;36:533–7. [28] Lacour JP, Perrin C. Eruptive familial lingual papillitis: a new entity? Pediatr Dermatol 1997;14:13–6. [29] Roux O, Lacour JP. Eruptive lingual papillitis with household transmission: a prospective clinical study. Br J Dermatol 2004;150:299–303. [30] Marks R, Scarff CE, Yap LM, Verlinden V, Jolley D, Campbell J. Fungiform papillary glossitis: atopic disease in the mouth? Br J Dermatol 2005;153:740–5. [31] Bouquot JE, Adibi SS, Sanchez M. Chronic lingual papulosis: new, independent entity or ‘‘mature‘‘ form of transient lingual papillitis? Oral Surg O Med Oral Path Oral Radiol 2012;113:111–7. [32] Yarom N, Cantony U, Gorsky M. Prevalence of fissured tongue, geographic tongue and median rhomboid glossitis among Israeli adults of different ethnic origins. Dermatology 2004;209:88–94. [33] Bhatnagar P, Rai S, Bhatnagar G, Kaur M, Goel S, Prabhat M. Prevalence study of oral mucosal lesions, mucosal variants, and treatment required for patients reporting to a dental school in North India: in accordance with WHO guidelines. J Family Community Med 2013;20:41–8. [34] Fernandes AM. Oral mucosa alterations in a socioeconomically deprived region: prevalence and associated factors. Braz Oral Res 2011;25:393–400. [35] Parlak AH, Koybasi S, Yavuz N, Anul H, Aydogan I, Cetinkaya R, et al. Prevalence of oral lesions in 13- to16-year-oldstudents in Duzce. Turkey. Oral Dis 2006;12:553–8. [36] Darwazeh AM, Almelaih AA. Tongue lesions in a Jordanian population. Prevalence, symptoms, subject’s knowledge and treatment provided. Med Oral Pathol Oral. Cir Bucal 2011;16:e745–749. [37] Vörös-Balog T, Vincze N, Bánóczy J. Prevalence of tongue lesions in Hungarian children. Oral Dis 2003;9:84–7. [38] Bessa CF. SantosPJ, Aguiar MC, do CArmo MA. Prevalence of oral mucosal alterations in children from 0 to 12 years old. J Oral Pathol Med 2004;33:17–22. [39] Shulman JD. Prevalence of oral mucosal lesions in children and youths in the USA. Int J Paediatric Dent 2005;15:89–97. [40] Yilmaz AE, Gorpelioglu C, Sarifakioglu E, Dogan DG, Bilici M, Calik N. Prevalence of oral mucosal lesions from birth to two years. Nigerian J Clin Pract 2011;14:349–53. [41] Järvinen J, Kullaa-Mikkonen A, Pesonen E. Histoquantitative study of inflamed tongue mucosa. Scand J Dent Res 1991;99:424–30. [42] Assimakopoulos D, Patikakos G, Fotika C, Elisaf M. Benign migratory glossitis or geographic tongue: an enigmatic oral lesion. Am J Med 2002;113:751–5. [43] Hume WJ. Geographic stomatitis: a critical review. J Dent. 1975;3:25–43. [44] Hernández-Pérez F, Jaimes-Aveldañez A, Urquizo-Ruvalcaba ML, Diaz-Barcelot M, Irigoyen-Camacho ME, Vega-Memije ME, et al. Prevalence of oral lesions in patients with psoriasis. Med Oral Pathol Oral Cir Bucal 2008;13:E703–708. [45] van der Wal, van der Kwast, van Dijk E, van der Waal I. Geographic stomatitis and psoriasis. Int J Oral Maxillofac Surg 1988;17:106–9. [46] Marks R, Simons MJ. Geographic tongue – a manifestation of atopy. Br J Dermatol 1979;101:159–62. [47] Marks R, Gzarny D. Geographic tongue: sensitivity to the environment. Oral Surg Oral Med Oral Pathol 1984;58:156–9. [48] Kullaa-Mikkonen A, Penttilä I, Kotilainen R, Puhalainen E. Haematological and immunological features of patients with fissured tongue syndrome. Br J Oral Maxillofac Surg 1987;25:481–7. [49] D’Erme AM, Agnoletti AF, Prignano F. Fissured tongue responding to biologics during the treatment of psoriasis: the importance of detecting oral involvement of psoriasis. Dermatol Ther 2013;26:364–6.

Fissured tongue: a sign of tongue edema?

Fissured tongue (FT) is a condition frequently seen in the general population. Clinically, FT is characterized by grooves that vary in depth and are n...
900KB Sizes 1 Downloads 3 Views