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Table of Contents
CASE REPORT
Year : 2018  |  Volume : 22  |  Issue : 1  |  Page : 33-35

Facial lupus vulgaris neglected for 50 years


1 Department of Dermatology, Mohamed V. Military Hospital, Mohamed V. University, Rabat, Morocco
2 Department of Pathology, Mohamed V. Military Hospital, Mohamed V. University, Rabat, Morocco

Date of Web Publication31-Jan-2018

Correspondence Address:
Prof. Naoufal Hjira
Department of Dermatology, Mohamed V. Military Hospital, Mohamed V. University, Rabat
Morocco
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdds.jdds_4_18

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  Abstract 


Tuberculosis (TB) is still a problem in the developed and underdeveloped countries, and cutaneous TB is a small part of extrapulmonary forms. Lupus vulgaris (LV) is the most common form of cutaneous TB among Western populations. In Morocco, scrofuloderma is the most common form of cutaneous TB followed by LV. LV results both from inoculation and from the endogenous spread through hematogenous or lymphatic route from an underlying infectious focus. We report a case of facial LV with a long delay in diagnosis of 50 years. A 59-year-old man presented with a right cheek slow-growing lesion for 50 years. Skin biopsy showed tuberculoid granulomas without caseous necrosis, and the culture of cutaneous biopsy was negative. The polymerase chain reaction of Mycobacterium complex-specific DNA sequences on a fresh skin biopsy material was positive. The patient was given antituberculous treatment with which lesion improved. The delayed diagnosis of 50 years is atypical, especially that the lesion had a display character. Delayed diagnosis of cutaneous TB will cause a significant increase in morbidity. Early diagnosis and treatment is the key to reduce the morbidity.

Keywords: Cutaneous tuberculosis, lupus vulgaris, Morocco, tuberculosis


How to cite this article:
Hjira N, Frikh R, Boudhas A, Baba N, Oukabli M, Boui M. Facial lupus vulgaris neglected for 50 years. J Dermatol Dermatol Surg 2018;22:33-5

How to cite this URL:
Hjira N, Frikh R, Boudhas A, Baba N, Oukabli M, Boui M. Facial lupus vulgaris neglected for 50 years. J Dermatol Dermatol Surg [serial online] 2018 [cited 2019 Sep 17];22:33-5. Available from: http://www.jddsjournal.org/text.asp?2018/22/1/33/224393




  Introduction Top


Cutaneous tuberculosis (CTB) is the least frequent of the extrapulmonary TB manifestations and it constitutes about 1%–2% of all TB cases.[1]

The disease can present in many different clinical presentations, and diagnosis may often pose difficulties as the clinical appearance of the lesions may not always be characteristic.[2] Lupus vulgaris (LV) is an uncommon type of CTB in Morocco.[3] It occurs in individuals with a moderate-to-high degree of immunity and presents as an asymptomatic, slow-growing plaque on the face. We report a case of facial LV with a long delay in diagnosis of 50 years.


  Case Report Top


A 59-year-old Moroccan man presented with an almost 50-year history of a reddish hyperkeratotic, painless, slowly growing plaque on his right cheek.

Lesion started as a small papule and gradually increased in size and spread in an annular fashion to form a plaque.

He reported that the skin lesion on the cheek had been growing centrifugally 2 years ago [Figure 1]. There was no past or family history of TB.
Figure 1: Erythematous infiltrated plaque involving the right cheek

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On clinical examination, there was a reddish infiltrated plaque involving the right cheek, measured more than 16 cm × 6 cm, with central atrophy and hyperkeratotic borders. On diascopy, apple jelly nodules were demonstrated. There was no lymphadenopathy, and systemic examination also did not reveal any abnormalities.

Considering the possibilities of deep fungal infections, cutaneous sarcoidosis, and CTB, a skin biopsy was taken.

Tissue histopathology showed tuberculoid granulomas with epithelioid cells and Langhans giant cells surrounded by chronic inflammatory cells without caseous necrosis in the dermis [Figure 2], [Figure 3], [Figure 4] (H and E, ×50, PAS, ×40, H and E, ×400)]. Fungal cultures were negative.
Figure 2: Tuberculoid granulomas without caseous necrosis in the dermis (H and E, ×50)

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Figure 3: Granulomas with epithelioid cells and Langhans giant cells without caseous necrosis (PAS, ×40)

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Figure 4: Tuberculoid granulomas with epithelioid cells and Langhans giant cells without caseous necrosis in the dermis (H and E, ×400)

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Conventional culture of fresh biopsy material time-delayed was negative. The polymerase chain reaction (PCR) of Mycobacterium complex-specific DNA sequences was positive. Hematological and biochemical investigations revealed no abnormalities. Chest X-ray was normal. Following CT scan of the thorax, bronchoscopy, and bronchoalveolar lavage. the diagnosis of LV without systemic involvement was established. Antituberculous treatment was initiated with isoniazid, rifampicin, and pyrazinamide.

An improvement of skin infiltration and incipient healing was observed 8 weeks later [Figure 5]. The patient had a complete healing after 6 months of therapy [Figure 6].
Figure 5: Improvement of skin infiltration after 8 weeks of treatment

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Figure 6: Complete healing after 6 months of antituberculous therapy

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  Discussion Top


TB is still a problem in the developed and underdeveloped countries, and cutaneous TB is a small part of extrapulmonary forms.[1]

Scrofuloderma is the most common form of cutaneous TB in Morocco (73% cases) followed by LV in 13%, TB verrucosa cutis in 7%, and tuberculids in 7% cases.[3]

The recent classification based on bacterial load and was classified into paucibacillary forms (LV, TB verrucosa cutis, and tuberculids) and multibacillary forms (scrofuloderma, tuberculous chancre, and acute miliary TB).[4] LV results both from inoculation and from endogenous spread through hematogenous or lymphatic route from an underlying infective focus. Rarely, it may develop following direct inoculation of the bacilli into the skin or at the site of Bacille Calmette–Guerin vaccination.[5]

The word “lupus” meaning wolf was given to the lesion because of the ulcerating and devouring character of the lesion.[6]

LV usually affects females more than males and could be presented as plaque, nodule, ulcer, mutilating, and vegetative lesions.[7],[8] It occurs on the face, ear, neck, and less common extremities and trunk.

Histopathology shows tuberculoid granulomas composed of lymphocytes, plasma cells, epithelioid cells and giant cells, and scant or absent central caseation, in the superficial dermis. The epidermis is usually hyperplastic but may be atrophic or ulcerated.[9]

Diagnosis of CTB may often pose difficulties as the clinical appearance of the lesions may not always be characteristic. Demonstration of acid-fast bacilli may be difficult with the high immune status of the patient.

In TB, there is a granulomatous inflammatory reaction in which the tuberculoid granulomas composed of lymphocytes, plasma cells, epithelioid cells and giant cells, and central caseous necrosis which is diagnostic, but its absence may not rule out the diagnosis of TB. In this case too, there was no evidence of caseous necrosis in the biopsy specimen. The absolute criteria for the diagnosis of CTB include a positive culture of Mycobacterium tuberculosis from the lesion or deoxyribonucleic acid amplification by PCR. The culture was negative; however, PCR was positive in our case. A therapeutic trial with antitubercular therapy is justified if clinical suspicion is strong and the diagnosis can be made based on the therapeutic response. The delay diagnosis of 50 years is atypical, especially that the lesions had a display character. To our knowledge, two cases had been reported in the literature with a delay diagnosis of 50 and 60 years.[10],[11] Delayed diagnosis of cutaneous TB results increasing rate of squamous or basal cell carcinoma, and hence, early diagnosis is very important.[12]

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.
Bravo FG, Gotuzzo E. Cutaneous tuberculosis. Clin Dermatol 2007;25:173-80.  Back to cited text no. 1
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2.
El Fekih N, Fazaa B, Kerkeni N, Sfia M, Zeglaoui F, Zermani R, et al. Chronic tubercular lupus. Med Mal Infect 2009;39:409-12.  Back to cited text no. 2
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3.
Zouhair K, El Moutaoui L, Benchikhi H, Lakhdar H. Cutaneous tuberculosis: a pathology still relevant in Morocco (study of 223 cases). Ann Dermatol Venereol 2007;134:1S30-62.  Back to cited text no. 3
    
4.
Yates VM. Mycobacterial infections. In: Tony B, Stephen B, Neil C, Christopher G, editors. Rook's Textbook of Dermatology. 8th ed., Vol. 31. Oxford, UK: Wiley-Blackwell; 2010. p. 10-29.  Back to cited text no. 4
    
5.
Marcoval J, Servitje O, Moreno A, Jucglà A, Peyrí J. Lupus vulgaris. Clinical, histopathologic, and bacteriologic study of 10 cases. J Am Acad Dermatol 1992;26:404-7.  Back to cited text no. 5
    
6.
Findlay GH, editor. Bacterial infections. In: The Dermatology of Bacterial Infections. 1st ed. London: Blackwell Scientific; 1987. p. 71-83.  Back to cited text no. 6
    
7.
Senol M, Ozcan A, Mizrak B, Turgut AC, Karaca S, Kocer H, et al. A case of lupus vulgaris with unusual location. J Dermatol 2003;30:566-9.  Back to cited text no. 7
    
8.
Thomas S, Suhas S, Pai KM, Raghu AR. Lupus vulgaris – Report of a case with facial involvement. Br Dent J 2005;198:135-7.  Back to cited text no. 8
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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
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10.
Amouri M, Ben Salah R, Chaaben H, Meziou TJ, Mseddi M, Turki H. Chronic tubercular lupus. Ann Dermatol Venereol 2011;138:637-8.  Back to cited text no. 10
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11.
Flohr C, Khan M, Leach IH, Johnston ID, English JS. Cutaneous tuberculosis due to Mycobacterium bovis lasting for more than 60 years. Clin Exp Dermatol 2009;34:921-3.  Back to cited text no. 11
[PUBMED]    
12.
Masmoudi A, Ayadi L, Turki H, Boudaya S, Bouassida S, Abdelmaksoud W, et al. Squamous cell carcinoma on tuberculous lupus. Ann Dermatol Venereol 2005;132:591-2.  Back to cited text no. 12
[PUBMED]    


    Figures

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



 

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