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Home , Lupus vulgaris, Mantoux test

PRACTICE IN BRIEF  Cutaneous is on the rise in immigrant population.  Difficulty in diagnosis is due to rarity of condition in developed nations.  Provides differential diagnosis of mid facial cutaneous lesions.  Provides a diagnostic approach for a dental practitioner in the case of vulgaris.

Lupus vulgaris — Report of a case with facial involvement

S. Thomas,1 S. Suhas,2 K. M. Pai3 and A. R. Raghu4

Cutaneous tuberculosis of the orofacial region is a rare condition and when it occurs, can cause confusion regarding the true nature of the lesion. This is compounded by the fact that neither nor histopathology is confirmatory. In this report we discuss a case of with emphasis on the diagnostic approach to be followed by dental practitioners who come across a similar case.

Tuberculosis of the skin is a rare disease size and number despite treatment with top- Examination of the patient (Fig. 1) accounting for approximately 1% of all ical and systemic antibiotics. These nodules revealed erythrematous scaly plaque with reported cases of tuberculosis.1,2 Cutaneous later coalesced to form a plaque on the nose. crusting around the nose extending on to tuberculosis has various clinical manifesta- The lesion was localised on the nose for two the cheeks, inferiorly to the lower lip, angle tions such as plaque, ulcers or mutilating years and in the last six months it had of the mouth and superiorly to the right lesions. One progressive form of cutaneous extended to involve the right lower eyelid lower eye lid. There was destruction of ala tuberculosis that occurs as a post-primary and upper lip, and had lead to the destruc- nasi, nasal septum and columella. The right infection in a person with moderate or high tion of the nasal tip. lower eyelid showed loss of eyelashes with degree of immunity is known as lupus vul- garis.3 Lupus vulgaris (LV) is characterised by plaque with apple-jelly nodule that extends irregularly with scar formation and tissue destruction.3 The lesion typically appears on the head and neck, particularly around the nose.4 Lupus vorax is a variant of LV that affects the nasal cartilage causing ulceration and mutilation.3 We report a case of lupus vulgaris affecting the nose with emphasis on the diagnostic difficulties and treatment protocol.

CASE REPORT A nine-year-old boy presented with a three- year history of multiple nodules appearing on and around the nose. These increased in

1*Department of Oral Medicine and Radiology, College of Dental Surgery, Manipal-576104, Karnataka, India; 2-4Department of Oral Medicine and Radiology, College of Dental Surgery, Manipal-576104, Karnataka, India. *Correspondence to Dr Shibu Thomas E mail: [email protected] Refereed Paper Received 14.10.03; Accepted 06.02.04 Fig. 1 Extra oral photograph showing midfacial destructive lesion showing erythrematous scaly plaque DOI: 10.1038/sj.bdj.4812038 © British Dental Journal 2005; 198: 135–137 with crusting around the nose.

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A review after three months revealed healed lesions on the face (Fig. 3) and improved eye symptoms. ATT was contin- ued for another three months then stopped. The patient is being reviewed every month for assessment of a possible relapse. Recon- structive surgery is planned for a later date.

DISCUSSION LV usually results from an infection of the skin in a person who has been infected else- where with tuberculosis.5 Usually the pathogenic mechanism is con- tiguous, lymphatic or haematogenous, spread from a tuberculous lesion. In rare sit- uations it can appear at the site of primary inoculation or at the site of a BCG vaccina- tion.6 The disease may have different forms of presentation including plaque, , muti- lating lesions, vegetative, tumour like lesion, and papulonodular lesion. The ulcerative and the mutilating form have Fig. 2 Photomicrograph of section stained with hematoxylin and eosin at 10X magnification showing multiple subepithelial without central casseation necrosis. the highest tendency for scarring together with deep tissue involvement.3 LV is commonly seen on the face, ears ectropion of the eyelid. There was conjuncti- defined epitheloid cells interspersed with and neck and may heal with scarring. val congestion and epiphora. Swelling and mononuclear leukocytes. These features Facial involvement may typically involve crusting of upper lip and fissuring of the were suggestive of a chronic granulomatous the nose and may result in destruction of angle of mouth was evident. Mouth opening inflammatory reaction. No typical tubercu- the nasal and septal cartilage. This muti- was limited as upper and lower lip appeared lous follicles or acid-fast bacilli could be lating variant of lupus vulgaris is also fibrosed and could not be everted. identified. Culture was negative for fungi, known as lupus vorax.7,8 Multiple bilateral submandibular lymph bacteria and acid-fast bacilli. Mycobacteri- The nasal lesion of LV may spread nodes were palpable, mobile and non tender. um tuberculosis DNA was identified from either directly or through lymphatic ves- Routine haematological and biochemical the tissue sample of the lesion subjected to sels to the buccal mucosa, palate, gingiva, investigations revealed no abnormalities. polymerase chain reaction (PCR). conjunctiva or oropharynx, giving rise to Serological test results for Leishmania, The patient was put on four drug anti ulcers or vegetation in these areas.7,9 Fur- , and human tubercular therapy (ATT) consisting of ther complications include secondary virus were negative. Para nasal sinus view 450 mg one-and-a-half tablets infection of the ulcerative lesion and and chest radiograph findings were normal. once daily, 300mg one-and-a- scarring deformities such as ectropion Fine needle aspiration cytology of lungs half tablets once daily, 750 and microstomia requiring plastic sur- revealed no evidence of tuberculosis. A mg half a tablet once daily and gery.8,9 purified protein derivative test (Mantoux 200 mg 1 tablet once daily for a period of In our patient the typical nasal lesion was test) was strongly positive, 14 mm diameter two months, following which Isoniazid and evident, along with scarring and involve- after 48 hours. A biopsy of the nasal lesion Rifampacin were continued for the next ment of upper lip and lower eyelid resulting (Fig. 2) revealed a delicate stroma, markedly four months. For the right eye, moistening in ectropion of the eyelid. The differential amorphous, exhibiting areas of necrosis sur- eye drops (0.7% hydroxypropyl methyl cel- diagnosis (Fig. 4) should include lupus ery- rounded by many multinucleated giant cells lulose) and antibiotic eye ointment (0.3% thematosus, lymphocytoma, Spitz naevus, of langhans type containing many small W/W ciprofloxacin) were used for a period syphilis, psoriasis and Bowen’s disease for nuclei disposed to the periphery and a few ill of two weeks. an early nodule or early plaque type. For the

Type of Lupus Vulgaris Differential diagnosis Early plaque or early nodule type • • Lymphocytoma • Spitz naevus • Syphilis • Psoriasis • Bowen’s disease Multinodular or vegetative type • Leishmaniasis • • • Acne rosacea • Wegener’s granulomatosus Fig. 3 Post treatment photograph of face showing healed lesion and residual defect. Fig. 4 Differential diagnosis for lupus vulgaris.

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more mutinodular or vegetative type the Investigations for Lupus Vulgaris 1. Chen C H, Shih J F, Wang L S, Perng R P. Tuberculous differential diagnosis will include leishma- subcutaneous : analysis of seven cases. • Chest radiograph Tuber Lung Dis 1996; 77: 184-187. niasis, leprosy, sarcoidosis, acne rosacea and 2. Grosset J H. Present status of chemotherapy for 3,8 • Mantoux test Wegener’s granulomatosus. tuberculosis. Rev Infect Dis 1989; II: 347-352. • Incisional biopsy Diagnosis of cutaneous tuberculosis is 3. Gawkrodger D J. Mycobacterial infections. In: Rook at times difficult because the clinical • Acid fast bacilli culture A, Wilkinson DS, Ebling FJG (Eds). Textbook of th appearance of the lesion may not always • Polymerase chain reaction Dermatology, 6 ed. Oxford; Boston: Blackwell Scientific, 1998; V.2, Chap. 28, p1194. be characteristic and will require multiple 4. El-Gatit A L, EL-Deriny, Mahmoud A A and Ashi A S. investigations (Fig. 5). At times culture for Presumed periorbital lupus vulgaris with ocular Fig. 5 Investigations for lupus vulgaris. the bacilli may not yield positive results extension. Ophthalmology 1999; 106: 1990-1993. and demonstration of acid-fast bacilli may 5. Sehgal V N, Jain M K and Srivastava G. Changing pattern of cutaneous tuberculosis — A prospective be difficult especially in patients with strong but cannot be confirmed with biopsy study. Int J Dermatol 1989; 28: 231-236. chronic lesions and with a high degree of or the Mantoux test. Then diagnosis can be 6. Marcoval J, Servitje O, Moreno A, et al. Lupus immunity against the infection.6,7,10,11 In done based on the therapeutic response of vulgaris. Clinical, histopathologic, and bacteriologic the present case acid fast bacilli could not the lesion to anti tubercular drugs.14 study of 10 cases. J Am Acad Dermatol 1992; 26: be demonstrated in the biopsy specimen The absolute criteria for diagnosis of 404-407. 7. Farina M C, Gegundez M I, Pique E, et al. Cutaneous and culture for acid fast bacilli yielded cutaneous tuberculosis are a positive culture tuberculosis. Clinical, histopathologic, and negative results. of M.tuberculosis from the lesion or success- bacteriologic study of 10 cases. J Am Acad Dermatol Tuberculosis infection is a - ful guinea-pig inoculation, or DNA identifi- 1995; 33: 433-440. tous inflammatory reaction in which the cation by PCR. In PCR, specific fragments of 8. Freitag D S, Chin R. Facial granuloma with nasal destruction. Chest 1998; 92-3: 422-423. granuloma shows central caseous necrosis mycobacterial DNA can be amplified and 9. Kounis N G, Constantinidis K. Lupus vulgaris: a rare which is diagnostic, but its absence may not hence, can be detected even when present dermal involvement. Practice of medicine 1980; rule out the diagnosis of tuberculosis.11,12,13 only in tiny amounts. It has sensitivity that 224: 1284-5. In this case there was no evidence of is equal to or higher than culture, and a high 10. Serfling U, Penneys N S, Leonardi C L. Identification caseation necrosis in the biopsy specimen. specificity.3,11 Diagnosis in the present case of Mycobacterium tuberculosis DNA in case of lupus vulgaris. J Am Acad Dermatol 1993; 28: 318-22. The Mantoux test is positive in most cases was based on the positive PCR test that 11. Sehgal V N, Jain M K and Srivastava G. Changing of LV6,11,13 as in our case. A negative reaction revealed presence of mycobacterial DNA. pattern of cutaneous tuberculosis — A prospective provides strong evidence against tuberculo- study. Int J Dermatol 1989; 28: 231-236. sis.14 The diagnostic value of a positive CONCLUSION 12. Walter J B, Hamilton M C, Israel M S. Tuberculosis, Leprosy, Syphilis and . In principles of test is blurred if the subject has a Since facial lesions of LV occur particular- pathology for dental students. 4th edition. Churchill history of BCG vaccination in early life or ly around the nose, the oral structures in living stone. pp213–223. previous mycobacterial infection. Thus con- close proximity are naturally involved. 13. Kumar B, Rai R, Kaur I, et al. Childhood cutaneous firmation is not always possible. Lupus vulgaris must be considered in the tuberculosis: a study over 25 years from northern A therapeutic trial of anti tubercular differential diagnosis of ulcerative and India. Int J Dermatology 2001; 40: 26-32. 14. Kakakhel K, Fritsch P. Cutaneous Tuberculosis. Int J drugs is justified if clinical suspicion is mutilating lesions of the oro-nasal region. Dermatol 1989; 28: 355-362.

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