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 ::  Abstract
 ::  Case Report
 ::  Discussion
 ::  Acknowledgement
 ::  References
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CASE REPORT
Year : 2009  |  Volume : 55  |  Issue : 3  |  Page : 190-192

Granulomatous cheilitis secondary to tuberculosis in a child


1 Department of Pediatrics, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi -110 002, India
2 Department of Dermatology, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi -110 002, India
3 Department of Pathology, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi -110 002, India

Date of Submission10-Jul-2009
Date of Decision22-Aug-2009
Date of Acceptance04-Sep-2009
Date of Web Publication2-Nov-2009

Correspondence Address:
M Bhattacharya
Department of Pediatrics, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi -110 002
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0022-3859.57400

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 :: Abstract 

Granulomatous cheilitis is a chronic granulomatous inflammation of the lips that can be secondary to various etiologies. A few infectious agents including Mycobacterium tuberculosis have been implicated in its etiology. It can be the clinical presentation of a tuberculide resulting from a hypersensitivity reaction to an underlying focus of tuberculosis such as pulmonary tuberculosis.
This case report describes a child with granulomatous cheilitis with pulmonary tuberculosis, who responded to anti-tubercular treatment. This is probably the first pediatric case of this rare condition.


Keywords: Granulomatous cheilitis, tuberculide, tuberculosis


How to cite this article:
Bhattacharya M, Rajeshwari K, Sardana K, Gupta P. Granulomatous cheilitis secondary to tuberculosis in a child. J Postgrad Med 2009;55:190-2

How to cite this URL:
Bhattacharya M, Rajeshwari K, Sardana K, Gupta P. Granulomatous cheilitis secondary to tuberculosis in a child. J Postgrad Med [serial online] 2009 [cited 2019 Jul 20];55:190-2. Available from: http://www.jpgmonline.com/text.asp?2009/55/3/190/57400


Granulomatous cheilitis is a granulomatous inflammation with mononuclear cell infiltration of the lips and peri-oral skin that presents as a diffuse painless local swelling of the affected areas. It can be secondary to numerous conditions including Crohn's disease, sarcoidosis and infections such as tuberculosis. [1] This communication describes the first pediatric case of this condition secondary to tuberculosis.


 :: Case Report Top


A five-year-old male child presented with progressive, painless swelling of both lips for four months. It was more in the morning during the initial 15-20 days, but subsequently became persistent throughout the day and spread to the adjacent peri-oral area. It was not accompanied by fever, atopic manifestations, swelling at any other site, urinary complaints, dysentery, cough or respiratory distress. There was no history of contact with tuberculosis. The swelling did not subside with anti-histamines. On general physical examination, he had cervical lymph nodes that were < 1 cm in diameter, pallor and right-sided phlyctenular conjunctivitis. There was no abnormality in the systemic examination. He had no systemic signs of sarcoidosis or Crohn's disease.

Local examination revealed grossly enlarged upper and lower lips with fissures. The mucosal surface was edematous and hyperemic. There were no ulcers or discharge. The swelling extended to the adjoining perioral skin. The nasal mucosa, floor of mouth, throat, teeth and tongue were normal. There was no facial nerve palsy [Figure 1].

The initial investigations revealed a normal total and differential leucocyte count and hemoglobin concentration of 8 gm/dL with hypochromia and microcytosis on the peripheral smear. The levels of serum urea and creatinine, electrolytes, serum proteins and albumin, serum calcium and serum angiotensin-converting enzyme were normal. Urine and stool analyses were normal too. Serological tests for syphilis were negative.

Chest radiograph showed ill-defined bilateral upper zone opacities. There was no hilar lymphadenopathy. There was no radiological improvement following a course of antibiotics. Tuberculin skin testing with purified protein derivative (PPD) was strongly positive. Gastric aspirate was positive for acid-fast bacilli (AFB). The abdominal ultrasound was normal.

Histopathological examination of the tissue biopsy obtained from the mucosal surface of the upper lip revealed hyperplastic stratified squamous epithelium with underlying dense mononuclear inflammatory infiltrate with a few epitheloid cell granulomas with Langhans giant cells with caseation. AFB could not be demonstrated [Figure 2].

A diagnosis of pulmonary tuberculosis with granulomatous cheilitis was made and four-drug anti-tubercular treatment (isoniazid, rifampicin, ethambutol and pyrazinamide) was initiated. No systemic or intra-lesional steroids were prescribed. After two months, the lower lip swelling had resolved, upper lip swelling had significantly reduced, the fissures had healed, the perioral skin swelling had resolved and the phlyctenular conjunctivitis had healed [Figure 3].


 :: Discussion Top


Granulomatous cheilitis is clinically characterized by diffuse and painless swelling of the lips. [1] When associated with fissured tongue and facial palsy it is known as  Melkersson-Rosenthal syndrome More Details. [1],[2] Isolated granulomatous cheilitis, also known as Miescher granulomatous cheilitis, is sometimes considered a monosymptomatic variant of Melkersson-Rosenthal syndrome. [1] Histopathological features consist of epitheloid cell granulomas with Langhans giant cell in a mononuclear inflammatory background. [3]

The cause is not known in the majority of the cases. But there may be genetic predisposition with increased incidence in siblings. [4] A few cases represent localized form of sarcoidosis or extraintectinal manifestation of Crohn's disease. [5],[6] Infective agents implicated in the etiology of granulomatous cheilitis are Toxoplasma gondii, Treponema pallidum, Mycobacterium tuberculosis, Mycobacterium leprae, Herpes simplex and Borrelia burgdorferi. [4]

The course of our patient was unlike that of orofacial tuberculosis which usually affects subjects in poor general health with depressed immunity and pulmonary tuberculosis. It starts with a yellowish nodule which breaks down into a painful ulcer. AFB may be demonstrable in the lesion.

Two reports of granulomatous cheilitis secondary to tuberculosis have been described in adults. [7],[8] This report constitutes the first published case of granulomatous cheilitis in a child. Our diagnosis was based not only on exclusion of other conditions (such as syphilis, Crohn's disease, sarcoidosis), on the basis of clinical and laboratory workup but also on positive evidence of tuberculosis, in the form of a persistent radiological lesion in the lungs, demonstration of AFB in gastric lavage, positive tuberculin skin test and response to anti-tubercular therapy. The caseating granuloma did not demonstrate the presence of AFB. We feel that the lesion represented a tuberculide, which being a hypersensitivity phenomenon is not associated with AFB. The characteristic features of a tuberculide are histopathological appearance of tuberculosis, absence of AFB and response to anti-tubercular therapy, [9] the criteria fulfilled by the reported case. We also wish to reiterate that due to the low microbiological yield in skin tuberculosis and poor correlation with polymerase chain reaction in cutaneous tuberculosis, therapeutic improvement is a well-established diagnostic tool. [10],[11],[12]

In conclusion, our case represents a rare presentation of granulomatous cheilitis in the pediatric age group secondary to underlying pulmonary tuberculosis. It also highlights the importance of infective conditions, especially tuberculosis, in the etiology of this rare clinical entity.


 :: Acknowledgement Top


We thank the patient's father for his kind permission to publish the photographs and case summary.

 
 :: References Top

1.Ghorbel IB, Sioud Dhrif A, Lamloum M, Trabelsi S, Habib Houman M. Melkersson-Rosenthal syndrome: Report of 5 cases and review of the literature. Tunis Med 2006;84:816-20.  Back to cited text no. 1      
2.Allen CM, Camisa C, Hamzeh S, Stephens L. Cheilitis granulomatosa: Report of six cases and review of literature. J Am Acad Dermatol 1990;23:444-50.  Back to cited text no. 2  [PUBMED]  [FULLTEXT]  
3.Wiesenfield D, Ferguson MM, Mitchell DN, MacDonald DG, Scully C, Cochran K, et al. Oro-facial granulomatosis: A clinical and pathological analysis. Q J Med 1985;54:101-13.  Back to cited text no. 3      
4.Hornstein OP. Melkersson-Rosenthal syndrome: A neuro-mucocutaneous disease of unknown origin. Curr Prob Dermatol 1973;5:117-56.  Back to cited text no. 4      
5.Kauzman A, Quesnel-Mercier A, Lalonde B. Orofacial granulomatosis: 2 case reports and literature review. J Can Dent Assoc 2006;72:325-9.  Back to cited text no. 5  [PUBMED]  [FULLTEXT]  
6.Hodgson TA, Buchanan JA, Porter SR. Orofacial granulomatosis. J Oral Pathol Med 2003;33:252.  Back to cited text no. 6      
7.Rao TV, Satyanarayana CV, Sundareshwar B, Reddy CR. Unusual form of tuberculosis of lips. J Oral Surg 1977;35:595-6.  Back to cited text no. 7  [PUBMED]  [FULLTEXT]  
8.Kavala M, Sudogan S, Can B, Sarigul S. Granulomatous cheilitis resulting from a tuberculide. Int J Dermatol 2004;43:524-7.  Back to cited text no. 8      
9.Champion RH, Burton JL, Burns DA. Mycobacterial infections. In: Gawkroder DJ. Textbook of dermatology, 6 th ed. Oxford: Blackwell Science Ltd; 1998. p. 1198-9.  Back to cited text no. 9      
10.Sehgal VN, Sardana K, Sharma S. Inadequacy of clinical and/or laboratory criteria for the diagnosis of lupus vulgaris, re-infection cutaneous tuberculosis: Fallout/implication of 6 weeks of anti-tubular therapy (ATT) as a precise diagnostic supplement to complete the scheduled regimen. J Dermatolog Treat 2008;19:164-7.  Back to cited text no. 10  [PUBMED]  [FULLTEXT]  
11.Sehgal VN, Sardana K, Sehgal R, Sharma S. The use of anti-tubercular therapy (ATT) as a diagnostic tool in pediatric cutaneous tuberculosis. Int J Dermatol 2005;44:961-3.  Back to cited text no. 11  [PUBMED]  [FULLTEXT]  
12.Sehgal VN, Sardana K, Bajaj P, Bhattacharya SN. Tuberculosis verrucosa cutis: Antitubercular therapy, a well-conceived diagnostic criterion. Int J Dermatol 2005;44:230-2.  Back to cited text no. 12  [PUBMED]  [FULLTEXT]  


    Figures

  [Figure 1], [Figure 2], [Figure 3]

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2004 - Journal of Postgraduate Medicine
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