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SHORT COMMUNICATION
Year : 2022  |  Volume : 9  |  Issue : 1  |  Page : 78-81

The possible link between cheilitis granulomatosa and oral health?


1 Department of Dentistry, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India
2 Department of Pathology, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India

Date of Submission14-Dec-2021
Date of Acceptance15-Feb-2022
Date of Web Publication06-Apr-2022

Correspondence Address:
Tulika Wakhloo
Department of Dentistry, All India Institute of Medical Sciences, Rishikesh, Uttarakhand
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdrr.jdrr_188_21

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  Abstract 


This article highlights cheilitis granulomatosa (CG) of the upper lip in a 6-year-old female child with neglected oral hygiene. CG is a rare, idiopathic, chronic inflammatory disorder presenting as a diagnostic dilemma due to a wide array of etiological factors. There is no standard treatment and often unpredictable therapeutic responses and spontaneous recurrences are reported. This article emphasizes on providing specialized dental treatment for active oral infections as the first line of therapy, especially in pediatric patients as it may turn out to be a major shot in the arm.

Keywords: Cheilitis granulomatosa, dental caries, orofacial granulomatosis, pediatric dentistry


How to cite this article:
Wakhloo T, Reddy SG, Chug A, Kumar A. The possible link between cheilitis granulomatosa and oral health?. J Dent Res Rev 2022;9:78-81

How to cite this URL:
Wakhloo T, Reddy SG, Chug A, Kumar A. The possible link between cheilitis granulomatosa and oral health?. J Dent Res Rev [serial online] 2022 [cited 2022 May 23];9:78-81. Available from: https://www.jdrr.org/text.asp?2022/9/1/78/342710




  Case Report Top


A 6-year-old female child was reported to the department of dentistry with a chief complaint of upper lip swelling for 6 months. The persistent swelling was asymptomatic, insidious in onset, and progressively increased to the present size. There was no history of associated itching, food or drug allergy, insect bite, bleeding, pus discharge, fever, and difficulty in mastication or speech. Past family and medical history were noncontributory. Systemic examination was unremarkable with normal vital signs. On extraoral examination, facial asymmetry with diffuse swelling of the upper lip was present with dry, erythematous overlying skin exhibiting scaling [Figure 1]. On palpation, the swelling was firm, nontender, and nonfluctuant with no pulsations or increased local temperature associated with right submandibular lymphadenopathy. Intraoral examination revealed normal mouth opening with neglected oral hygiene. The patient had mixed dentition with grossly decayed primary teeth associated with gingival swellings. Panorex revealed multiple periapical radiolucencies and bone resorption with respect to involved teeth [Figure 2]. A provisional diagnosis of cheilitis granulomatosa (CG) of the upper lip was considered and the patient was referred to the department of pediatrics for thorough investigations. Hemogram, liver function tests, and kidney function tests were within the normal range. No abnormality was detected in serum angiotensin-converting enzyme levels, Chest X-ray, and tuberculin skin test. In addition, a patch test to rule out atopic reaction to allergens was also negative.
Figure 1: Swelling of the upper lip

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Figure 2: Panorex showing periapical radiolucency and bone resorption with respect to grossly carious primary teeth

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Considering the clinical presentation and uncooperative behavior of the patient, a comprehensive dental treatment was planned under GA as the first line of therapy to remove all possible foci of infection. The dental treatment included oral prophylaxis, pit, and fissure sealant application on all erupted permanent first molars, restoration of deciduous canines, and extraction of maxillary and mandibular deciduous incisors and molars associated with gingival swellings. This was followed by a punch biopsy from the upper lip for histopathological examination. The microscopy of the section revealed mildly acanthotic stratified squamous epithelium with pandermis showing edema and moderate-to-dense mixed inflammatory cell infiltrate mainly comprising lymphocytes, few neutrophils, and aggregates of histiocytes along with occasional epithelioid cells.

On follow-up visits, the patient showed satisfactory postoperative healing of the extraction sites with complete resolution of gingival swellings. In addition, a significant improvement in the upper lip swelling and erythema was observed after 3 months without any additional therapy [Figure 3]. The patient was advised meticulous oral hygiene maintenance and clinical follow-up after every 3 months.
Figure 3: Reduction in the upper lip swelling after 3 months

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  Discussion Top


CG is a rare, idiopathic, chronic inflammation, manifesting clinically as persistent, soft to firm, nontender, and diffuse swelling of one or both the lips. It is frequently associated with vertical fissuring of the lip angular cheilitis and has a significant psychological impact on the young child due to the resultant cosmetic deformity.[1],[2],[3] Simultaneous intraoral involvement in the form of diffuse anterior gingival swelling, fissures, erosions, paresthesia, edema, taste alterations, cobblestone appearance of the oral mucosa, and papillary hyperplasia in the palate may be seen.[3] Thus, pediatric dentists can play a pivotal role in the early and correct diagnosis of this rare entity. With an incidence rate of 0.08% and female predilection, it usually affects young adults in the second decade of life.[3] The present case reports CG of the upper lip in a 6-year-old female child with rampant caries and concomitant gingival swellings.

Differential diagnosis

CG presents as a diagnostic dilemma and requires thorough systemic examination and investigations to exclude other granulomatous lesions having similar clinical and histopathological features.[3],[4] The differential diagnosis of CG includes angioedema, foreign body reaction, mycobacterial infection, sarcoidosis, Crohn's disease, leprosy, Wegener's granulomatosis, histoplasmosis, amyloidosis, rosacea, Ascher's syndrome, MEN-IIb, Non-Hodgkin lymphoma, intake of drugs such as angiotensin-converting enzyme inhibitors and calcium channel blockers, and atopic reaction to allergens, and diseases such as C1 esterase deficiency.[1],[3],[4]

Etiology and pathogenesis

The precise etiology remains unknown; however, some reported mechanisms include chronic antigenic stimulation leading to monoclonal lymphocytic infiltration and cell-mediated hypersensitivity with lymphatic blockage,[1] alteration in the function of autonomic nervous system localized to facial skin resulting in increased vascular permeability and edema.[3] Although genetic evidence is minimal, autosomal dominant inheritance pattern has been demonstrated. The responsible gene has been mapped to chromosome 9 p11[4] and FATP1 mutation has been reported to cause Melkersson–Rosenthal syndrome in a Han Chinese family.[4] In addition, CG is associated with autoimmune mechanisms and allergic reactions to food items and additives such as benzoates, monosodium glutamate, carmoisine; toothpaste and dental materials such as amalgam and mercury.[2],[3],[4] The possible mechanism in our case is the random influx of inflammatory cells without involvement of a specific antigen[3] which could have been triggered by the chronic periapical infection of the deciduous dentition.

Histopathology

The characteristic histologic features of CG are those of dermal noncaseating granulomatous inflammation.[5] In the present case, pandermis is edematous and shows an ill-formed granuloma along with few Langhans type of giant cells and mixed inflammatory cell infiltrate reaching up to the deep dermis. Stain for acid-fast bacilli was negative [Figure 4]a and [Figure 4]b.
Figure 4: (a) Epidermis showing mild acanthosis with pandermal interstitial moderate inflammatory cell infiltrate with Langhans type of giant cell (Hematoxylin-eosin stain, original magnification × 200), (b) Mixed inflammatory cell infiltrate comprising lymphocyte and few neutrophils with aggregates of histiocytes (Hematoxylin-eosin stain, original magnification × 400)

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Management

The management should aim at improving the patient's esthetics and preventing recurrences which are otherwise common.[6] In the current case, after ruling out the differentials, chronic periapical infection of the deciduous dentition was considered the possible etiology and comprehensive dental treatment was done as the first line of therapy [Figure 5]. The patient did not receive any supplemental medication and showed marked improvement in clinical appearance with no signs of recurrence on 3 and 6 months follow-up.
Figure 5: Flowchart illustrating the present case

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On the literature review, CG cases have been mostly managed by oral and intralesional corticosteroids and various other combination regimens; however, many studies report unpredictable therapeutic responses and spontaneous recurrences. Moreover, there is no standard treatment protocol since the comparative trials for these drugs are lacking.[2],[7] Elimination diets such as cinnamon-free and benzoate-free diets have been recommended,[3],[8] whereas surgery (cheiloplasty) and helium–neon laser radiation treatment is reserved only for patients with impaired function and major lip deformity.[3],[5] It is worthwhile to note that several studies report cases of chronic granulomatous disease including steroid-resistant cases associated with poor oral hygiene, carious teeth, gingivitis, periodontitis, and periapical infections of variable intensity;[9],[10] however, the exact correlation still requires further exploration.[10] Therefore, a multidisciplinary approach for early diagnosis with prompt treatment and follow-up of active intraoral infections is emphasized in the management, especially in children with CG.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Khan Mubeen BS. Granulomatous cheilitis of the upper lip in a child. Int J Biomed Adv Research 2016;7:306-9.  Back to cited text no. 1
    
2.
Chintagunta SR, Sana SN, Bukka KS, Padma S. Cheilitis granulomatosa: Case series. J Dr. NTR Univ Health Sci 2017;6:174.  Back to cited text no. 2
    
3.
Nair PA, Patel TM. Granulomatous cheilitis involving the lower lip. Egypt J Dermatol Venerol 2017;37:85.  Back to cited text no. 3
  [Full text]  
4.
Sharma YK, Chauhan S, Deo K, Agrawal P. Granulomatous cheilitis: Report of three cases and systematic review of cases and case series reported from India. Clin Dermatol Rev 2020;4:12.  Back to cited text no. 4
  [Full text]  
5.
Banks T, Gada S. A comprehensive review of current treatments for granulomatous cheilitis. Br J Dermatol 2012;166:934-7.  Back to cited text no. 5
    
6.
Bansal M, Singh N, Patne S, Singh SK. Orofacial granulomatosis affecting lip and gingiva in a 15-year-old patient: A rare case report. Contemp Clin Dent 2015;6 Suppl 1:S94-6.  Back to cited text no. 6
    
7.
Shanmukhappa AG, Shivaram B, Budamakuntala L, Samynathan A. Idiopathic granulomatous cheilitis of Miescher in a young patient: A rare entity and its successful treatment. Indian J Paediatr Dermatol 2017;18:217.  Back to cited text no. 7
  [Full text]  
8.
Critchlow WA, Chang D. Cheilitis granulomatosa: A review. Head Neck Pathol 2014;8:209-13.  Back to cited text no. 8
    
9.
Dar-Odeh NS, Hayajneh WA, Abu-Hammad OA, Hammad HM, Al-Wahadneh AM, Bulos NK, et al. Orofacial findings in chronic granulomatous disease: Report of twelve patients and review of the literature. BMC Res Notes 2010;3:37.  Back to cited text no. 9
    
10.
Ukichi K, Yabushita M, Kimura Y, Iguchi N, Kouno M, Tanaka YI, et al. CR0327 Six cases of cheilitis granulomatosa improved after dental treatment. Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:e375.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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