Oral Medicine

QUINTESSENCE INTERNATIONAL ORAL MEDICINE

Ida M. Kornerup Transient lingual papillitis

Ida M. Kornerup, DDS, Dip Paedo1/Mireya Senye, DDS, MSc TMD/Orofacial Pain, MSc Orthodontics, FRCS (C)2/ Edmund Peters, DDS, MSc, FRCD (C)3

A case of recurrent, clinically innocuous, but painful papules self-limited condition involving the tongue fungiform papillae, involving the tongue dorsum of a 25-year-old man is present- which does not appear to be widely recognized. (Quintessence ed. The lesions were interpreted to represent a transient lin- Int 2016;47: 871–875; doi: 10.3290/j.qi.a36888) gual papillitis. This a poorly understood, but benign and

Key words: lingual, papillitis, tongue lesions, tongue sensitivity

Transient lingual papillitis (TLP) is characterized by recur- anterior tongue dorsum or a more generalized involve- rent, painful, focal or multifocal, red or yellow/white ment, primarily affecting the anterior and lateral tongue papules involving the tongue dorsum, which can occur dorsum.2 A third, non-painful, papulokeratotic variant, over a wide age range from children to adults.1-3 The characterized by multiple, recurrent, more diffusely dis- papules represent inflamed, variably ulcerated, fungi- tributed, hyperkeratotic white/yellow papules has also form papilla. TLP was first formally described by Whita- been described.2,6 A further non-painful condition, ker et al1 in 1996, who suggested, despite lack of earlier termed “chronic lingual papulosis,” characterized by focal documentation, that this was a relatively common con- or diffuse enlargement of lingual papilla has been dition. Since this report, there have been a small number described. In contrast to TLP, this was thought more likely of publications that have further documented the con- to represent fibrotic enlargement of filiform papillae dition.4-13 However, the pathogenesis and management (rather than fungiform papillae), occurring in response to considerations remain poorly defined. low-grade chronic irritation. The relationship of chronic Different patterns of clinical expression have been lingual papulosis to TLP is uncertain. It could represent an described. These include localized involvement of one or end-stage presentation of a long standing TLP or, alterna- more inflamed fungiform papilla, most frequently on the tively, a different, etiologically distinctive, condition.12 In spite of the putative common occurrence of TLP, 1 Assistant Clinical Professor and Acting Head, Division of Pediatric Dentistry; and it does not appear to be well recognized. This paper Director, Pediatric Dentistry Clinic; School of Dentistry, Faculty of Medicine and Dentistry, University of Alberta, Alberta, Canada. presents a case that fit the clinical profile of TLP, and 2 Private Practice, Montreal, Quebec, Canada. discusses the relevant literature. 3 Professor, Division of Oral Pathology and Oral Medicine, School of Dentistry, Faculty of Medicine and Dentistry, University of Alberta, Alberta, Canada.

Correspondence: Dr Ida Kornerup, Division of Pediatric Dentistry, Fac- CASE REPORT ulty of Medicine and Dentistry, University of Alberta; and Acting Divi- sion Head Pediatric Dentistry, Director Pediatric Dentistry Clinic, 5-516 A 25-year-old Caucasian man presented with a com- Edmonton Health Academy, 11405 87 Ave, Edmonton, AB, T6G 1C9, Canada. Email: [email protected] plaint of painful “tongue bumps”, which had been

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intermittently developing for over 2.5 years. He indi- Empirical management included patient education cated that between 5 to 10 painful papules would to improve awareness of the possible role of factitial develop on the lateral tongue dorsum about once a injury associated with oral parafunctional activity and month. He would obtain immediate profound relief by the potential role of topical allergenic agents. The removing the papules with nail clippers. However, the patient was encouraged to refrain from surgical lesions would spontaneously recur in about 2 to 3 self-management. Fabrication of an occlusal stabiliza- weeks following this self-management. There was no tion appliance for sleep bruxism was recommended previous diagnosis and because of the persistent recur- and a 0.12% chlorhexidine solution was prescribed to rent nature of the condition, he indicated increasing sponge on affected areas to help manage possible sec- concerns this might represent a cancer. ondary infection. Review of local associated or eliciting factors indi- The patient presented for follow-up after 13 months cated the lesions were exacerbated or possibly induced about a week after an exacerbation. He had not with tomato sauce. There was a history of clenching. obtained the splint but indicated that application of The medical history was unremarkable. There was the chlorhexidine solution afforded mild relief within 1 no history of allergy. or 2 days after an exacerbation and he also felt this At the initial examination, the glossal lesions were improved resolution when he performed surgical in a quiescent phase. However, localized multifocal self-management. He noted that development of the white slightly raised lesions were evident on the left lesions occurred approximately once every 1 or 2 lateral aspect of the tongue and, to a lesser extent, on months and seemed to correlate with stress. He had the right lateral aspect of the tongue. Focal resolving become aware of his habit of forcefully pressing his ulcerations were also noted at the sites of earlier surgi- tongue against his palate and maxillary teeth. Again, cal self-management (Fig 1). lateral tongue crenations were not noted, possibly In spite of the clenching history, there was no evi- because the parafunctional glossal stresses were not dence of lateral tongue crenations or other diffuse directed against mandibular teeth. Figure 2 shows an inflammatory change involving the anterior or lateral erythematous sensitive papule and recent focal lesions, aspects of tongue, which could occur in response to which had been clipped and appeared to be develop- chronic parafunctional irritation involving the tongue. ing again. Further empirical management with topical There was no evidence of malocclusion, including open corticosteroid application to be used at inception of the bite, which might suggest tongue thrusting. There was lesions has proven useful to help control the develop- no evidence of anterior gingival inflammatory changes, ment of the lesions, limit duration to about 1 day, and which might suggest a mouth breathing habit. In this to moderately reduce sensitivity. However, lesions regard, the patient did not indicate any concerns occasionally still develop after a 2-year follow-up and regarding breathing difficulties and no breathing diffi- thus, there is not definitive resolution. culties were noted during the course of the interview and clinical exam. DISCUSSION Findings from the remaining exam were unremark- able although a “lip licking” habit was noted. The lip The tongue lesions described in this report were inter- licking was in response to a perception of dryness of preted as a reactive condition, preferentially affecting the vermilion. fungiform papilla for unknown reasons. The condition A smear obtained from the tongue dorsum to fit the profile of a TLP and the patient was reassured assess the unlikely possibility of a secondary candidal that there were no features suggesting malignancy. infection was negative. No further testing, including Categorizing the case as TLP was useful to remove the culture or biopsy, was obtained. stress associated with a potential cancer diagnosis and

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Fig 1 TLP in a quiescent stage. The Fig 2 The same patient shown in Fig 1 Fig 3 A 21-year-old Oriental woman with arrows show representative papules, after 13 months. A circle indicates a sensi- acutely painful small white papules which from the history, appeared to be in tive erythematous papule. Arrows show (arrows) on the tongue dorsum. A very mild different stages of development. The cir- white papules, which had been excised by diffuse erythema is evident anteriorly, sug- cles show healing focal ulcers in sites the patient before his reevaluation. gestive of inflammation possibly caused by which had been previously clipped by the parafunctional tongue activity. The painful patient. Centrally, the tongue dorsum papules were confirmed with the patient’s shows a moderate coating. active participation in the exam. to exclude other inappropriate diagnoses. However, predictably limited benefit. Subsequent empirical man- TLP is still poorly defined and the diagnosis was of lim- agement with topically applied 0.2% chlorhexidine ited value with respect to identifying definitively an with topical corticosteroids was useful to control pain etiologic basis or in terms of directing further treat- and minimize duration of the occasional recurrence to ment. The evidence implicating fungiform papilla is about a day in a subsequent 4-year follow-up period. primarily based on the clinical inference that the sensi- The unusual sensitivity associated with these lesions tive papules, which characterize this condition, mimic could be because fungiform papillae are architecturally the distribution and anatomic structure of fungiform more fragile than the surrounding robust, heavily kera- papilla. Even though this interpretation has been tinized filiform papillae. As previously noted, mature widely accepted and appears reasonable, we were not fungiform papillae have a thin non-keratinized strati- able to find any photomicrographs in reported cases, fied squamous epithelial lining, which supports taste which showed convincing histologic features of a fun- buds. There is evidence, in a mouse model, that both giform papilla. These features should include a thin, epithelial and connective tissue components of taste non- or lightly keratinized, layer of stratified squamous buds are embryonically derived from neural crest.16,17 epithelium and the presence of taste buds.14,15 Structurally, the taste buds have external pores to per- A striking feature of the present case was the mit ingress of chemical substances.18 Thus, the lining marked sensitivity, which appeared completely dispro- would be relatively susceptible to injurious stimuli, and portionate to the presenting clinical features. This dis- the resultant inflammation with attendant edema crepancy is further illustrated in Fig 3, which shows would involve a relatively well-innervated fibrous core, painful recurrent tongue lesions in a 21-year-old which uniquely supports taste bud function. Possibly, healthy Oriental woman. Examination, with the these considerations explain the selective inflammation patient’s active assistance, confirmed that the incon- involving the fungiform papilla and the consequent spicuous white dorsal tongue papules corresponded to localized sensitivity. In this regard, in those cases where the painful lesions. There had been five previous acute the reactive change involves thickening of the epithe- episodes over a 2-year period, each lasting about 8 to 9 lial lining (the papulokeratotic variant), sensitivity does days. Previous diagnoses had included “thrush” and an not occur. unspecified viral infection. The earlier management A variety of injurious pro-inflammatory stimuli or had included nystatin and fluconazole, which were of predisposing conditions have been described. Different

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reports have noted that since the common end point in ciencies, which result in dorsal tongue atrophy.6 TLP is an inflamed fungiform papilla, there is no reason Well-defined clinical management protocols have not that this could not be mediated through a range of been established and treatment remains empirical and pro-inflammatory factors.1,3,7 palliative.1-3 Among the proposed treatments are use of The suggested eliciting factors have included irrita- topical anesthetics, corticosteroids, coating agents, tion associated with chronic trauma or parafunctional chlorhexidine rinse, change of oral hygiene products, glossal activity,1,19 chemical damage described in an identification and elimination of potential allergenic individual licking envelopes,16 hypersensitivity to foods agents, and control of parafunctional habits. The use of or oral hygiene products,2,3,8,9 guttate psoriasis,21 and nail clippers to remove the lesions, which was practiced hormonal predisposition.2 The possibility of viral infec- by our patient, has been previously documented and is tion has also been suggested in a similar condition not recommended.1,2 In the present cases, manage- termed “eruptive lingual papillitis”.4,7 These reports ment with topical corticosteroids in conjunction with described a condition characterized by recurrent topical application of 0.2% nonalcoholic chlorhexidine inflammation of the fungiform papillae of the tip and solution has proven useful. Patient reassurance regard- lateral tongue dorsum, primarily in children. There was ing the benign nature of the condition was important. variable association with lymphadenopathy, fever, and angular cheilitis. Possible transmission to family mem- CONCLUSION bers was noted. Diagnosis is made on the basis of the clinical pre- In summary, although there has been a suggestion that sentation, and biopsy is generally not necessary. In the TLP is a common condition, the incidence is not known. occasional cases where a biopsy is obtained, the histo- Retrospective surveys, attempting to assess the fre- pathologic presentation is nonspecific. The papillae quency of presentation based on patient recollection, show chronic inflammation with edema, possibly with should be treated with caution. Diagnosis should be ulceration or mild hyperkeratosis. The non-painful pap- restricted to those cases in which it is obvious that ulokeratotic variant shows epithelial hyperplasia, there is selective involvement of fungiform papillae hyperkeratosis, and chronic inflammation.1,2,4-6 after examination by qualified clinicians. The condition Clinical exclusion of other possible diagnoses is is benign and self-limiting although often painful and necessary and the differential diagnosis could include recurrent. Management involves efforts to identify and herpetiform aphthous stomatitis and recurrent herpetic eliminate any potential local or systemic factors and infection.3 However, recurrent herpetic ulcers initially empirical conservative topical palliative treatment. In present as vesicles, typically on masticatory mucosa. the present cases, patient education with reassurances Herpetiform aphthous ulcers are very unusual and regarding the benign nature of the condition was present as multiple punctate ulcers involving erythem- important. atous mucosa, usually on non-keratinized mucosa. In both cases, these evolve into obvious ulcerations, which are distinct from TLP. Fungiform papilla enlarge- REFERENCES ment has also been noted in other clinical contexts, 1. Whitaker SB, Krupa JJ 3rd, Singh BB. Transient lingual papillitis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1996;82:441–445. which appear distinctive from TLP. These include 2. Neville B, Damm D, Allen C, Bouquot J. Oral and maxillofacial pathology. 3rd asymptomatic fungiform papilla enlargement, which ed. Philadelphia: WB Saunders, 2008:330–332. 22 3. Flaitz CM, Chavarria C. Painful tongue lesions associated with a food allergy. can occur with ciclosporin A use and in neurofibroma- Pediatr Dent 2001;23:506–507. tosis.23 Other systemic conditions that can show fungi- 4. Lacour JP, Perrin C. Eruptive familial lingual papillitis: a new entity? Pediatr form papilla pathosis include scarlet fever, familial Dermatol 1997;14:13–16. 5. Flaitz CM. Oral and maxillofacial pathology case of the month. Fungiform dysautonomia, some anemias, and with vitamin defi- papillae. Tex Dent J 1999;116:47,88.

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