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CORRESPONDENCE
Year : 2022  |  Volume : 67  |  Issue : 6  |  Page : 790-791
Buruli ulcer a diagnostic challenge-A report from non-endemic area


Department of Dermatology, Apollo Gleneagals Hospital, Kolkata, West Bengal, India

Date of Web Publication23-Feb-2023

Correspondence Address:
Abhijit Saha
Department of Dermatology, Apollo Gleneagals Hospital, Kolkata, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.ijd_409_22

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How to cite this article:
Lahiri K, Dhar S, Saha A. Buruli ulcer a diagnostic challenge-A report from non-endemic area. Indian J Dermatol 2022;67:790-1

How to cite this URL:
Lahiri K, Dhar S, Saha A. Buruli ulcer a diagnostic challenge-A report from non-endemic area. Indian J Dermatol [serial online] 2022 [cited 2023 Mar 23];67:790-1. Available from: https://www.e-ijd.org/text.asp?2022/67/6/790/370298




Sir,

Buruli ulcer (BU) is a necrotising, debilitating neglected tropical disease (NTD) mostly reported from different tropical countries of West Africa, and South and Central America.[1] Cases have also been reported from non-endemic areas, which point towards the importance of acquiring more awareness about this entity.[1],[2] Early diagnosis and prompt treatment can prevent lifelong deformities and disabilities.

A 60-year-old female patient presented with an ulcer on her left leg of 1-year duration. She complained of local pain and tenderness. The ulcer started as a painless nodule with oedema of the affected area, which ulcerated in 4 weeks' time. The ulcer had undermined edges and a shaggy necrotic base [Figure 1]. A deep punch biopsy from the ulcer edge was taken and submitted for histopathological examination in 10% buffered formalin. She gave a history of an ulcer on the other leg, which healed with areas of depression and cribriform scar formation. Our differentials were vasculitis, erythema induratum, deep fungal infection, venous stasis ulcer, atypical mycobacterial infection, and pyoderma gangrenosum. Mantoux's test was positive. Pus culture did not show any growth. Anti-nuclear antibody was negative. Colour-doppler ultrasonography of the venous and arterial systems did not show any abnormality. TB BACTEC culture (MGIT system) from pus failed to show any growth at the end of 6 weeks.
Figure 1: Ulcer with undermined edges and a shaggy necrotic base

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Histopathology revealed ulceration of the epidermis. An intense inflammatory response involved the surface of the epidermis as well as the dermis. The dermis was edematous, had degenerative changes, and showed interstitial and perivascular mixed inflammatory cell infiltrate comprising neutrophils, histiocytes, lymphocytes, and plasma cells. There was a zone of ischemic necrosis at the level of subcutaneous fat, which was rimmed by a granulomatous response comprising many multinucleate foreign bodies and Langhans giant cells [Figure 2]. Looking at the granulomatous response, the ZN stain was performed, which revealed bunches of acid-fast bacilli (AFB) in the zone of necrosis in the subcutaneous fat[Figure 3]. Because of this finding of AFB clumps in the typical location of necrosis at the deep level of subcutaneous fat, it was diagnosed as an atypical mycobacterial ulcer due to Mycobacterium ulcerans.
Figure 2: H&E400: Zone of ischemic necrosis at the level of subcutaneous fat, which was rimmed by a granulomatous response comprising many multinucleate foreign bodies and Langhans giant cells

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Figure 3: ZN stain: ZN stain showing bunches of acid-fast bacilli (AFB) in the zone of necrosis in the subcutaneous fat

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Subsequently, the ulcer was sampled for mycobacterial culture.

Meanwhile, the ulcer was treated with rifampicin 600 mg daily and clarithromycin 500 mg twice a day for 6 weeks, and follow-up visits by the patient showed satisfactory healing of the ulcer.

Real-time polymerase chain reaction (PCR) is the gold standard in terms of sensitivity and rapidity for the diagnosis of MU DNA but not widely available, is costly, and requires expertise. The major limitations of isolating the organism from the sample are its slow-growing nature (doubling time >48 h) and low density. The sensitivity of the culture is low, only 35–60% yield positive results and requires an institution of decontamination protocol to avoid contamination with other fast-growing bacteria.[3] Our culture report came out as negative. Our case fulfils histopathological diagnostic criteria for BU, which comprises necrotising granuloma surrounding zones of necrosis and demonstration of AFB in the vicinity. Histopathology is a rapid and highly sensitive method (about 90%) to diagnose Mycobacterium ulcerans.[4]

Our case also supports the effectiveness of the oral rifampicin and clarithromycin combination as described in the literature. BU should be considered in the differential of chronic non-healing ulcers even in a non-endemic area such as ours.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Yotsu. RR, Suzuki K, Simmonds RE, Bedimo R, Ablordey A, Yeboah-Manu D, et al. Buruli ulcer: A review of the current knowledge. Curr Trop Med Rep 2018;5:247-56.  Back to cited text no. 1
    
2.
George AJ, Samuel V, Samuel VM, Gaikwad P. Buruli ulcer: Rare presentation of a chronic nonhealing ulcer in India. Indian J VascEndovascSurg 2019;6:138-40.  Back to cited text no. 2
    
3.
Sakyi SA, Aboagye SY, DarkoOtchere I, Yeboah-Manu D. Clinical and laboratory diagnosis of buruli ulcer disease: A systematic review. Can J Infect Dis Med Microbiol 2016;2016:5310718.doi: 10.1155/2016/5310718.  Back to cited text no. 3
    
4.
Laga AC, Milner DA Jr. Bacterial diseases: Buruliulcer (mycobacterium ulcerans). In: Elder DE, editor. Lever's Histopathology of the Skin. 11thed. Gurugram: Wolters Kluwer; 2019. p. 680-82.  Back to cited text no. 4
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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