Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 6  |  Issue : 2  |  Page : 138-140

Buruli ulcer: Rare presentation of a chronic nonhealing ulcer in India


1 General Surgery Unit 1, CMC Hospital, Vellore, Tamil Nadu, India
2 Department of Vascular Surgery, CMC Hospital, Vellore, Tamil Nadu, India

Date of Web Publication6-Jun-2019

Correspondence Address:
Dr. Ajith John George
General Surgery Unit 1, CMC Hospital, Vellore, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijves.ijves_76_18

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  Abstract 


Buruli ulcer is a rare disabling skin infection caused by Mycobacterium ulcerans. It is essential to consider Buruli ulcer as one of the possible differential diagnoses for a chronic nonhealing ulcer and treat the wounds with antitubercular therapy for at least 2 months before grafting. A young male from Nigeria, which is endemic for Buruli ulcer, presented with a long-standing ulcer with undermined edges. Various differential diagnoses were ruled out, such as venous stasis ulcer, cutaneous leishmaniasis, squamous cell carcinoma, and others. A biopsy and culture was suggestive of an atypical mycobacterial species. Early diagnosis of ulcers of infective etiology is imperative to prevent functional disability. Early debridement and initiation of antitubercular therapy is essential.

Keywords: Buruli ulcer, Mycobacterium ulcerans, wide local excision


How to cite this article:
George AJ, Samuel V, Samuel VM, Gaikwad P. Buruli ulcer: Rare presentation of a chronic nonhealing ulcer in India. Indian J Vasc Endovasc Surg 2019;6:138-40

How to cite this URL:
George AJ, Samuel V, Samuel VM, Gaikwad P. Buruli ulcer: Rare presentation of a chronic nonhealing ulcer in India. Indian J Vasc Endovasc Surg [serial online] 2019 [cited 2019 Oct 19];6:138-40. Available from: http://www.indjvascsurg.org/text.asp?2019/6/2/138/259660




  Introduction Top


Buruli ulcer is a disabling skin infection caused by Mycobacterium ulcerans (M. ulcerans). It is named for the Buruli district in Uganda, a region where many of the early cases in the literature were described. Buruli ulcer begins as localized skin lesions that progress to extensive ulceration, leading to functional disability, loss of economic productivity, and social stigma. Since the 1998 World Health Organization (WHO) Buruli ulcer initiative, there has been increased attention to research efforts for treatment and control of Buruli ulcer.[1] There is no such report from India. There are reports of other disease mimicking a Buruli ulcer.


  Case Report Top


A 16-year-old boy from Nigeria presented to our tertiary care hospital, with history of a large ulcer on the left leg along with pedal edema for 5 months. He had some blisters in the thigh area after which it had turned into an ulcer with pus discharge. There was no history of fever, vomiting, or diarrhea. He had no known comorbid illness. There was no history of trauma or any insect bite.

He was evaluated at Nigeria in April for the same, told to have Buruli ulcer, and started on rifampicin 600 mg once daily + clarithromycin 500 mg twice daily for the same.

On examination, there was a large ulcer overlying the left knee and extending to the thigh and over the left shin. The edges were sloping and undermined in few areas. The floor had minimal slough with granulation tissue. The base was fixed. There was surrounding hyperpigmentation, edema, with isolated smaller ulcers with undermined edges [Figure 1].
Figure 1: Buruli ulcer, left lower limb

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There was a fixed flexion deformity of the left knee joint causing an antalgic gait.

There were few enlarged inguinal nodes.

A biopsy from the edge of the ulcer showed necrotizing granulomatous inflammation, with acid-fast bacilli (AFB) noted. Magnetic resonance imaging and a venous duplex scan were done to rule out a venous ulcer or a malignant infiltrative ulcer. His polymerase chain reaction (PCR) for Mycobacterium ulcers was not conclusive. This may have been due to the initiation of therapy before the culture. Following a debridement of the ulcer, there was healthy granulation tissue. In collaboration with the department of infectious disease at our center, he was initiated on injection streptomycin 1 g intramuscular once daily, tablet rifampicin 600 mg per oral once daily, tablet moxifloxacin 600 mg per oral once daily, and tablet clarithromycin 500 mg per oral twice daily.

On review in the outpatient department after 1 month of antitubercular therapy and physiotherapy, there was a significant improvement in the healing of the ulcer. There was adequate granulation with visible decrease in the size of the ulcer. All adjacent ulcers had healed. He was able to straighten his knee and assume normal gait. He was planned for a split-thickness skin graft after 2 months of therapy [Figure 2].
Figure 2: Postsplit-thickness skin grafting, Buruli ulcer

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  Discussion Top


In a tropical country like India, there are various differential diagnoses that we can consider for a chronic nonhealing ulcer, for example, venous ulcer, eumycetoma, cutaneous leishmaniasis, squamous cell carcinoma, and sickle-cell ulcers. The country of origin and history of treatment are as important as the diagnostic tests. The pathogenesis of the disease is not clearly understood, but there are characteristics of M. ulcerans which enable it to cause such a devastating disease process. There are three notable characters as follows:

  1. M. ulcerans is dependent on the temperature of the area where the bacteria were inoculated
  2. Its inability to penetrate intact skin and its inability to infect abraded skin, as demonstrated in an experimental infection of guinea pigs and mice. These results suggest that Buruli ulcer is dependent on the passive inoculation of M. ulcerans through intact skin as an alternative to ineffective passive passage through abraded skin, with the precise role of “biological needles,” such as mosquitoes and other insects, remaining to be studied in comparison with the effectiveness of mechanical needles
  3. The presence of cell damage in the absence of an acute inflammatory response [2]


The various diagnostic tools are AFB smears, AFB cultures, and PCR for Mycobacterium ulcers. A debridement must be done if there are secondary microbial infection and secondary ulcers. The WHO recommendations for the treatment are different combinations of antibiotics given for 8 weeks which are used to treat the Buruli ulcer irrespective of the stage. One of the following combinations may be used depending on the patient. (1) A combination of rifampicin (10 mg/kg once daily) and streptomycin (15 mg/kg once daily) or (2) a combination of rifampicin (10 mg/kg once daily) and clarithromycin (7.5 mg/kg twice daily).

A randomized controlled trial concluded in 2017, and preliminary results show no difference in cure rates between the two treatments.

For pregnant women, the combination of rifampicin and clarithromycin is considered the safer option because of the contraindication to streptomycin.

In Australia, a combination of rifampicin (10 mg/kg once daily) and moxifloxacin (400 mg once daily) is routinely used with good results, but its effectiveness has not been proven by the randomized trial.[3],[4] The WHO has set up guidelines for treatment, including early detection and antibiotic treatment. There is a lot of focus on community-level activities, strengthening of the health systems, standardized case management, and supportive activities.[4] There is no consensus on wound cover. It is presumed that once the course of antibiotics has been completed, with a negative wound culture, it is safe to proceed with skin grafts or flaps.


  Conclusion Top


It is important to diagnose these ulcers of infectious etiology early to prevent the functional disability.[5]

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.

Acknowledgment

I would like to thank the surgical and infectious disease teams from CMC, Vellore.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Tai AY, Athan E, Friedman ND, Hughes A, Walton A, O'Brien DP. Mycobacterium ulcerans in mosquitoes captured during outbreak of buruli ulcer, Southeastern Australia. Emerg Infect Dis 2018;24:58-64.  Back to cited text no. 1
    
2.
Zingue D, Bouam A, Tian RB, Drancourt M. Buruli ulcer, a prototype for ecosystem-related infection, caused by Mycobacterium ulcerans. Clin Microbiol Rev 2018;31. pii: e00045-17.  Back to cited text no. 2
    
3.
Bertolotti A, Izzo A, Grigolato PG, Iabichella ML. The use of ozone therapy in buruli ulcer had an excellent outcome. BMJ Case Rep 2013;2013. pii: bcr2012008249.  Back to cited text no. 3
    
4.
Global Buruli Ulcer Initiative. WHO Buruli Ulcer; 2018.  Back to cited text no. 4
    
5.
Stienstra Y, van Roest MH, van Wezel MJ, Wiersma IC, Hospers IC, Dijkstra PU, et al. Factors associated with functional limitations and subsequent employment or schooling in buruli ulcer patients. Trop Med Int Health 2005;10:1251-7.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2]



 

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