Granulomatous cheilitis secondary to tuberculosis in a child
M Bhattacharya1, K Rajeshwari1, K Sardana2, P Gupta3,
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
Department of Pediatrics, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi -110 002
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.
|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;55:190-192
|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 2023 Mar 31 ];55:190-192
Available from: https://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.  This communication describes the first pediatric case of this condition secondary to tuberculosis.
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  When associated with fissured tongue and facial palsy it is known as Melkersson-Rosenthal syndrome. , Isolated granulomatous cheilitis, also known as Miescher granulomatous cheilitis, is sometimes considered a monosymptomatic variant of Melkersson-Rosenthal syndrome.  Histopathological features consist of epitheloid cell granulomas with Langhans giant cell in a mononuclear inflammatory background. 
The cause is not known in the majority of the cases. But there may be genetic predisposition with increased incidence in siblings.  A few cases represent localized form of sarcoidosis or extraintectinal manifestation of Crohn's disease. , Infective agents implicated in the etiology of granulomatous cheilitis are Toxoplasma gondii, Treponema pallidum, Mycobacterium tuberculosis, Mycobacterium leprae, Herpes simplex and Borrelia burgdorferi. 
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. , 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,  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. ,,
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.
We thank the patient's father for his kind permission to publish the photographs and case summary.
|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.|
|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.|
|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.|
|4||Hornstein OP. Melkersson-Rosenthal syndrome: A neuro-mucocutaneous disease of unknown origin. Curr Prob Dermatol 1973;5:117-56.|
|5||Kauzman A, Quesnel-Mercier A, Lalonde B. Orofacial granulomatosis: 2 case reports and literature review. J Can Dent Assoc 2006;72:325-9.|
|6||Hodgson TA, Buchanan JA, Porter SR. Orofacial granulomatosis. J Oral Pathol Med 2003;33:252.|
|7||Rao TV, Satyanarayana CV, Sundareshwar B, Reddy CR. Unusual form of tuberculosis of lips. J Oral Surg 1977;35:595-6.|
|8||Kavala M, Sudogan S, Can B, Sarigul S. Granulomatous cheilitis resulting from a tuberculide. Int J Dermatol 2004;43:524-7.|
|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.|
|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.|
|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.|
|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.|