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 ~  Abstract
 ~ Introduction
 ~ Case Report
 ~ Discussion
 ~  References
 ~  Article Figures
 ~  Article Tables

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  Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 33  |  Issue : 1  |  Page : 168-171
 

Facial lupus vulgaris of bilateral periorbital skin and conjunctiva: A case report and brief review


1 Department of Microbiology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India
2 Department of Dermatology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India
3 Department of Ophthalmology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India

Date of Submission20-Sep-2013
Date of Acceptance08-Mar-2014
Date of Web Publication5-Jan-2015

Correspondence Address:
S Verma
Department of Microbiology, Indira Gandhi Medical College, Shimla, Himachal Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0255-0857.148438

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 ~ Abstract 

A 22-year-old male presented to the Dermatology Department with bilateral plaque lesions distributed symmetrically over malar area, bridge of nose and upper eyelids progressing over 1 year 3 months. Lesion remained unhealed after antibiotic treatment. Microscopy and culture for fungal and mycobacterial infections were negative. The Mantoux test showed an exaggerated response and PCR was positive for Mycobacterium tuberculosis complex. Patient was treated successfully with anti-tubercular therapy.


Keywords: Cutaneous tuberculosis, lupus vulgaris, Mantoux test


How to cite this article:
Verma S, Verma G, Shanker V, Tegta G R, Sharma A, Pandey M L. Facial lupus vulgaris of bilateral periorbital skin and conjunctiva: A case report and brief review. Indian J Med Microbiol 2015;33:168-71

How to cite this URL:
Verma S, Verma G, Shanker V, Tegta G R, Sharma A, Pandey M L. Facial lupus vulgaris of bilateral periorbital skin and conjunctiva: A case report and brief review. Indian J Med Microbiol [serial online] 2015 [cited 2019 Jul 18];33:168-71. Available from: http://www.ijmm.org/text.asp?2015/33/1/168/148438



 ~ Introduction Top


The rising trend of tuberculosis (TB) is a staggering problem the world over. Cutaneous tuberculosis (CTB) constitutes 1-2% of all TB cases. [1],[2] There is diversity of morphological forms and unusual clinical presentation with involvement of face, nose, conjunctiva and buttocks. [3],[4],[5] Lupus vulgaris is the commonest form of CTB characterised by slowly enlarging plaque with slightly elevated border, central atrophy and "apple jelly" crusting. [1] Diagnosis poses a major challenge as results of microscopy and conventional culture are negative consequential to sparse bacilli in the lesion. Clinical presentation is ambiguous as facial lesions of sporotrichosis, chromoblastomycosis and cutaneous leishmaniasis may mimic CTB. [6],[7],[8] This accounts for underestimation of cases and diagnosis may be overlooked. Molecular diagnostic techniques like PCR followed by anti-tubercular treatment helps in resolving diagnostic dilemma and disease.


 ~ Case Report Top


A 22-year-old male visited the Dermatology clinic with non-healing, gradually progressing bilaterally symmetrical facial lesions. The disease spanned over 1 year and 3 months starting with a small solitary pus-discharging papule over left side of nose progressing to involve adjacent malar areas, medial canthi and lower eyelids. Patient complained of painless oozing of pus, itching in lesions and watering of eyes. There was absence of fever, cough, haemoptysis and chest pain. The patient showed no response to antibiotic treatment.

A review of history revealed a similar ulcerated lesion discharging pus over left thigh one year back which healed spontaneous over a few months. Patient denied trauma at site of lesions, self manipulation and insect bites or history of contact with patient of tuberculosis.

Local examination revealed a well-circumscribed plaque, sized about 10 × 5 cm distributed over butterfly area of face extending to both upper and lower eyelids. Lesion presented characteristic raised plaque, erythematous borders with adherent brown scales and crusts topping the surface. There was non-tender induration with few pustules. Symmetrical involvement of conjunctiva of eyelids and lower puncta was perceptible with bilateral cicatricial ectropion [Figure 1]. There was conjunctival congestion. The slit lamp examination of eye and fundus were normal. There was no destruction of underlying bone or cartilage. Vision was 6/6 in both eyes.
Figure 1: Well circumscribed, bilaterally symmetrical plaque lesions involving butterfly area and upper eyelids

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The back of left thigh showed well-defined scar of size 3 × 2 cm. Chest X-ray was normal. Bilateral cervical lymphadenopathy was present. The rest of the systemic findings and vitals were normal.

Considering the possibilities of sporotrichosis, chromoblastomycosis, cutaneous leishmaniasis and CTB, a skin biopsy was taken. The laboratory work-up to rule out deep fungal infections and cutaneous leishmaniasis was non-contributory. The direct microscopy of 10% KOH wet mounts did not show sclerotic or Asteroid bodies and yeast cells. Amastigotes were not visualised in Giemsa-stained smears. The Ziehl-Neelsen stained smears were negative for acid-fast bacilli. The specimen was inoculated on Sabouraud's dextrose agar and Lowenstein-Jensen media for fungal and mycobacterial cultures and incubated at 25°C and 37°C for 1 and 3 months, respectively. Tissue histopathology showed dense dermal granulomatous infiltrate [Figure 2] consisting of multiple giant cells, lymphocytes and epitheloid cells [Figure 3]. Chronic granulomatous reaction with [Figure 4] no definitive evidence of fungal, parasitic or mycobacterial disease and diagnosis remained obscure. A biopsy sample was sent for real-time PCR for Mycobacterium tuberculosis. Considering common occurrence of sporotrichosis in healthy inhabitants of sub-Himalayan regions, empirical treatment with saturated solution of potassium iodide (SSKI) was started while culture reports were awaited but lesions deteriorated.
Figure 2: Photomicrograph showing dense dermal granulomatous infiltrate (×10)

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Figure 3: Photomicrograph showing infiltrate consisting of multiple giant cells, lymphocytes and epitheloid cells (×40)

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Figure 4: Photomicrograph showing hyperkeratosis, acanthosis and crusted epidermis and dermis revealing marked chronic granulomatous infiltrate (×10)

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Mantoux test with 1 TU of PPD showed an exaggerated response relating possibility of CTB [Figure 5]. The treatment was shifted to quadruple antitubercular therapy (ATT) with rifampicin 450 mg, isoniazid 300 mg, ethambutol 800 mg and pyrazinamide 1500 mg daily for 2 months followed by isoniazid and rifampicin in the same doses for 4 months.
Figure 5: Mantoux test showing exaggerated response with fluid-filled bullous reaction

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Subsequently, the PCR was positive for M. tuberculosis complex confirming the diagnosis and we observed remarkable improvement with ATT after four weeks although mycobacterial culture remained sterile. Fungal culture was also sterile. A complete course of directly observed short course of ATT was curative [Figure 6]. Conservative management of cicatricial ectropion was done with plan of surgical reconstruction at a later date.
Figure 6: Lesions showing healing after antitubercular therapy with mild ectropion

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 ~ Discussion Top


Tuberculosis remains a constant threat to humans throughout the world. An estimated 1-2% of all tuberculosis cases have cutaneous disease. [1],[2] CTB has re-emerged in areas with high incidence of HIV infection and multi-drug resistant pulmonary tuberculosis. [9] The sites of predilection are head and neck in European countries and buttocks and trunk in India. [3] Our patient presented with rare facial lesions in butterfly distribution with a healing lesion on left thigh probably due to the same pathology. Extension to eyelids and conjunctiva was seen without visual impairment. Similar exceptional cases of facial CTB with and without ocular involvement have been rarely reported earlier. [4],[5] A brief review is presented in [Table 1] [5],[10],[11],[12],[13],[14],[15],[16],[17] . Ocular involvement may range from minimal conjunctivitis to periostitis, cold abscess, dacryoadenitis and bony involvement which may have grave consequences if diagnosis and therapy is delayed. [10],[18]
Table 1: Review of cases of facial lupus vulgaris with/without involvement of eye


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CTB is seen in patients with moderate to high degree of immunity. The lesions are classified based on the mechanism of propagation which includes direct inoculation, contiguous infection or haematogeneous dissemination. Based on bacterial load the disease is categorised as multibacillary with enumerable bacilli and paucibacillary with sparse bacilli. CTB includes a morphological plethora with primary-inoculation TB, tuberculous chancre, scrofuloderma, TB periorificialis and acute military TB in the multibacillary spectrum. At the paucibacillary pole are tuberculosis verrucosa cutis and lupus vulgaris (LV) with sparse bacilli difficult to isolate or visualise. [1],[4] LV is the most common variant approximating 59% of cases of CTB. [3] The morphological patterns may vary; nevertheless certain characteristic findings are very redolent of CTB. LV is characterised by annular plaques with verrucose borders and an "apple jelly" crust in the centre of the healing lesion which were not seen in the present case. Haematogeneous dissemination is implicated in facial LV with a tendency of multiple site involvement as seen in our patient. [1] We were unable to observe mycobacteria in smears or culture of biopsy sample which in all probability was due to shortfall of bacilli for microscopy and culture in lupus. This paucity of acid-fast bacilli is in agreement with findings of other authors. [5] In such cases PCR has an unequivocal role in authenticating diagnosis. The predictive value of Mantoux test is debatable. Ramam et al. document low sensitivity and specificity using cut-off of 10-mm induration. They observed reactions ranging between 0 and 30 mm in normal healthy adults. [19] A strongly positive PPD reaction of >15 mm has been considered of diagnostic value by Bravo et al. [1] A hyperactive skin test in our patient was the only finding suggesting CTB simulating observations made in other documented cases [Table 1]. [5],[10],[11],[12],[13],[14],[15],[16],[17]

In spite of exhaustive laboratory work-up, diagnosis remained incomprehensible. With a view to treat probable sporotrichosis or chromoblastomycosis, both of which are a common occurrence in this region, a therapeutic trial with SSKI was initiated but without improvement. [6],[7] Subsequent to exaggerated Mantoux test, ATT was administered. Later, the diagnosis was confirmed by PCR. A convincing favourable response also assented diagnosis of CTB and resolved the lesions in 4 weeks time with clinical cure after completion of therapy.

Early diagnosis and treatment is crucial in preventing irreparable damage when extension to major organs like eye is inevitable. In clinically ambiguous cases of CTB which may elude judgement, PCR plays a pivotal role in diagnosis. The results of Mantoux test and trial therapy may also be useful where molecular techniques are inaccessible. The conventional diagnostic modalities remain unhelpful and prove to be rather wasteful of resources and time. A therapeutic trial of 5 weeks is adequate to indicate CTB and in our case credible response was seen after 4 weeks. [20] There is a pressing need to make highly sensitive molecular diagnosis, like PCR, available and affordable for confirmation of such cases and discourage improvident use of modalities with low sensitivity.

 
 ~ References Top

1.
Bravo FG, Gotuzzo E. Cutaneous tuberculosis. Clin Dermatol 2007;25:173-80.  Back to cited text no. 1
    
2.
Hernández Solis A, Herrera Gonzalez NE, Cazarez F, Mercadillo Perez P, Olivera Diaz HO, Escobar-Gutierrez A, et al. Skin biopsy: A pillar in the identification of cutaneous Mycobacterium tuberculosis infection. J Infect Dev Ctries 2012;6:626-31.  Back to cited text no. 2
    
3.
Ramesh V, Misra RS, Jain RK. Secondary tuberculosis of the skin. Clinical features and problems in laboratory diagnosis. Int J Dermatol 1987;26:578-81.  Back to cited text no. 3
    
4.
Warin AP, Jones EW. Cutaneous tuberculosis of the nose with unusual clinical and histological features leading to a delay in the diagnosis. Clin Exp Dermatol 1977;2:235-42.  Back to cited text no. 4
[PUBMED]    
5.
Cook CD, Hainsworth M. Tuberculosis of the conjunctiva occurring in association with a neighbouring lupus vulgaris lesion. Br J Ophthalmol 1990;74:315-6.  Back to cited text no. 5
    
6.
Verma S, Verma GK, Singh G, Kanga A, Shanker V, Singh D, et al. Sporotrichosis in sub-Himalayan India. PLoS Negl Trop Dis 2012;6:e1673.  Back to cited text no. 6
    
7.
Verma S, Verma GK, Singh G, Kanga A, Sharma V, Gautam N. Facial chromoblastomycosis in sub-Himalayan region misdiagnosed as cutaneous leishmaniasis: Brief report and review of Indian literature. Dermatol Online J 2012;18:3.  Back to cited text no. 7
    
8.
Verma GK, Verma S, Shanker V, Singh G, Tegta GR. A rare case of diffuse cutaneous leishmaniasis in an immunocompetent patient from sub-Himalayan, India. Trop Doct 2012;42:237-9.  Back to cited text no. 8
    
9.
Barbagallo J, Tager P, Ingleton R, Hirsch RJ, Weinberg JM. Cutaneous tuberculosis: Diagnosis and treatment. Am J Clin Dermatol 2002;3:319-28.  Back to cited text no. 9
    
10.
El-Ghatit AM, El-Deriny SM, Mahmoud AA, Ashi AS. Presumed periorbital lupus vulgaris with ocular extension. Ophthalmology 1999;106:1990-3.  Back to cited text no. 10
    
11.
Khandpur S, Reddy BS. Lupus vulgaris: Unusual presentations over the face. J Eur Acad Dermatol Venereol 2003;17:706-10.  Back to cited text no. 11
    
12.
Daabek B, Issaoui B, Siala M, Cherif S, Koubaa J, Sayadi SJ, et al. Cervical-facial skin tuberculosis. Three case reports. Tunis Med 2004;82:51-4.  Back to cited text no. 12
    
13.
Ghosh SK, Bandyopadhyay D, Ghoshal L. Facial swelling and ulceration with nasal destruction. Cleve Clin J Med 2011;78:289-90.  Back to cited text no. 13
    
14.
Padmavathy L, Rao LL, Ethirajan N, Krishnaswami B. Ulcerative lupus vulgaris of face: An uncommon presentation in India. Indian J Tuberc 2007;54:52-4.  Back to cited text no. 14
    
15.
Garg A, Wadhera R, Gulati SP, Singh J. Lupus vulgaris of external nose with septal perforation -A rarity in antibiotic era. Indian J Tuberc 2010;57:157-9.  Back to cited text no. 15
    
16.
Ljubenovic MS, Ljubenovic DB, Binic II, Jankovic AS, Jancic SA. Cutaneous tuberculosis and squamous-cell carcinoma. An Bras Dermatol 2011;86:541-4.  Back to cited text no. 16
    
17.
Mlika RB, Tounsi J, Fenniche S, Hajlaoui K, Marrak H, Mokhtar I. Childhood cutaneous tuberculosis: A 20-year retrospective study in Tunis. Dermatol Online J 2006;12:11.  Back to cited text no. 17
    
18.
Madge SN, Prabhakaran VC, Shome D, Kim U, Honavar S, Selva D. Orbital tuberculosis: A review of the literature. Orbit 2008;27:267-77.  Back to cited text no. 18
    
19.
Ramam M, Malhotra A, Tejasvi T, Manchanda Y, Sharma S, Mittal R, et al. How useful is the Mantoux test in the diagnosis of doubtful cases of cutaneous tuberculosis? Int J Dermatol 2011;50:1379-82.  Back to cited text no. 19
    
20.
Ramam M, Tejasvi T, Manchanda Y, Sharma S, Mittal R. What is the appropriate duration of a therapeutic trial in cutaneous tuberculosis? Further observations. Indian J Dermatol Venereol Leprol 2007;73:243-6  Back to cited text no. 20
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
 
 
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