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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 9  |  Issue : 2  |  Page : 223-225

Long-term ulcerations caused by Mycobacterium lepromatosis


1 Dermatology Unit, Department of Health Sciences (DISSAL), University of Genoa, San Martino Policlinic Hospital, IRCCS, Genoa, Italy
2 Dermatology Unit, San Martino Policlinic Hospital, IRCCS, Genoa, Italy

Date of Web Publication29-May-2020

Correspondence Address:
Ilaria Trave
Department of Health Science, University of Genoa, San Martino Polyclinic Hospital, Via Pastore 1, 16132 Genoa
Italy
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmy.ijmy_40_20

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  Abstract 


Patients with leprosy rarely present ulcerated lesions that can appear during reactional states like Lucio's phenomenon (LP), as in our case. LP is a rare complication of multibacillary leprosy due to massive bacilli invasion of endothelial cells causing a thrombotic syndrome. The initial macular lesion is purpuric followed by multiple infiltrated papules and nodules, some of them ulcerated, associated to loss of sensation on lower limbs. The importance of recognizing ulcers as a specific cutaneous manifestation of leprosy allows early diagnosis and treatment, and therefore avoiding the development of disabilities and persistence of illness. Infection by Mycobacterium lepromatosis is associated with LP and it should be especially sought in patients from endemic areas.

Keywords: Leprosy, Lucio's phenomenon, Mycobacterium lepromatosis, ulcerations


How to cite this article:
Trave I, Barabino G, Cavalchini A, Parodi A. Long-term ulcerations caused by Mycobacterium lepromatosis. Int J Mycobacteriol 2020;9:223-5

How to cite this URL:
Trave I, Barabino G, Cavalchini A, Parodi A. Long-term ulcerations caused by Mycobacterium lepromatosis. Int J Mycobacteriol [serial online] 2020 [cited 2020 Jul 9];9:223-5. Available from: http://www.ijmyco.org/text.asp?2020/9/2/223/285229




  Introduction Top


Leprosy (Hansen's disease) is probably the oldest human infection historically caused by Mycobacterium leprae.[1] It is a worldwide problem with 208,619 new cases reported globally in 2018. The WHO identified India (120,334 cases), Brazil (28,660 cases), and Indonesia (17,017 cases) as regions with global highest incidence of new cases in 2018.[2] Although Italy has a lower incidence of leprosy cases than endemic areas (five cases notified during 2018),[3] it shares a delay between onset of symptoms and appropriate treatment.[4] The early diagnosis of leprosy and the strong adherence of therapy, that it should be monitored by serum anti-phenolic glycolipid-I and Il-17A expression in lesions, are important key pillars of leprosy management to avoid disability.[4],[5] We present a case of long-term ulcerations caused by infection of Mycobacterium lepromatosis.


  Case Report Top


A 41-year-old woman native of Cuba presented to our dermatologic department with diffuse generalized cutaneous infiltration and ulcerations localized in the abdominal region and in the upper and lower extremities. Four years before, she had developed recurrent painful, well-shaped erythematous macules in the lower extremities, later becoming necrotic. Histologically, through a biopsy of the lower extremity, the diagnosis was compatible with granulomatous and necrotic cutaneous vasculitis. A therapy based on systemic corticosteroids was partially successful. When we ran a physical examination, multiple reddish-purple lesions with irregular borders evolving in multiple ulcers were present in upper and lower extremities, some with necrotic eschars other with achromatic scars, associated to cutaneous infiltration, extremities' loss of sensation, and eyebrows/eyelashes loss [Figure 1]. The histopathological analysis showed extensive infiltration of histiocytes and Virchow cells that involved derma, subcutaneous tissue, small nerves, and blood vessels with vascular thrombosis. A Fite-Faraco stained examination of the skin was positive to acid-fast bacilli [Figure 2]. The molecular search of leprosy agents led to the conclusion that only M. lepromatosis was present in the sample. These results allowed the diagnosis of diffuse lepromatous leprosy (DLL) associated to Lucio's phenomenon (LP) caused by infection of M. lepromatosis. According to the WHO guidelines, a multidrug therapy (MDT) was setup consisting to combination of rifampicin (600 mg once a month), clofazimine (300 mg once a month and 50 mg/die), and dapsone (100 mg/die) up to achieve zero bacterial index.
Figure 1: Multiple ulcers in lower extremities

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Figure 2: Acid-fast bacilli in the skin. Fite stain, (magnification, ×100)

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  Discussion Top


Leprosy (Hansen's disease) is probably the oldest human infection historically caused by M. leprae.[1] It is a worldwide problem with 208,619 new cases reported globally in 2018. The WHO identified India (120,334 cases), Brazil (28,660 cases), and Indonesia (17,017 cases) as regions with global highest incidence of new cases in 2018.[2] In 2008, a novel Mycobacterium species named M. lepromatosis was recognized as the cause of death of two Mexican patients.[6]M. lepromatosis has a genome-scale mismatch of ~13% with M. leprae and there is 9.1% genetic difference between the two leprosy bacilli.[7] In addiction to Mexico, countries where nested polymerase chain reaction assay reported a high prevalence of M. lepromatosis were Canada, Brazil, Costa Rica, Singapore, and Myanmar.[6] Clinically, M. Lepromatosis mainly causes diffuse leprosy of Lucio and Latapi.[8] This form of leprosy shows a diffuse cutaneous infiltrate and frequent skin ulceration in the late stage with no nodule or plaque formation.[6] DLL is predominantly seen in patients from western and central Mexico and from Caribbean countries, as for our patient.[8] DLL can develop severe reactions called Lucio reactions (erythema necroticans). Lucio reaction is a vasculopathy that results in cutaneous infarcts which produce severe necrosis and desquamation of the skin.[8] Histologically, it is characterized by the existence of immune complexes, necrotizing vasculitis on superficial and medium-sized vessels.[8] LP could be confused with an atypical form of type 2 reaction called reactive perforating leprosy characterized by painful punch-out ulcers, similar to our patient's lesions, but with a different histology.[9] The term LP should only be used when there is a correlation of clinical (skin ulceration) and histological findings (vascular thrombosis and invasion of blood vessels by leprosy bacilli).[10] The classic treatment of DLL consists of the WHO MDT characterized by combination of rifampicin, clofazimine, and dapsone for 12 months. LP is treated with systemic corticosteroids and topical management. Although the exact source of M. lepromatosis infection by the patient is very uncertain. We suppose that she may have contracted it from her father when she lived to Cuba.


  Conclusion Top


We describe a case of DLL associated to LP caused by infection of M. lepromatosis. In our opinion, LP should be included in the differential diagnoses in patients presenting long-term ulcerations also in nonendemic areas. M. lepromatosis is often associated with DLL and LP. It should be sought every time, especially in patients from endemic areas.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Monot M, Honoré N, Garnier T, Zidane N, Sherafi D, Paniz-Mondolfi A, et al. Comparative genomic and phylogeographic analysis of Mycobacterium leprae. Nat Genet 2009;41:1282-9.  Back to cited text no. 1
    
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3.
Riccò M, Vezzosi L, Balzarini F, Mezzoiuso AG, Ranzieri S, Vaccaro FG, et al. Epidemiology of leprosy in Italy (1920-2019): A comprehensive review on existing data. Acta Biomed 2019;90:7-14.  Back to cited text no. 3
    
4.
Naaz F, Mohanty PS, Bansal AK, Kumar D, Gupta UD. Challenges beyond elimination in leprosy. Int J Mycobacteriol 2017;6:222-8.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Gunawan H, Annissa MN, Dwiyana RF, Avriyanti E, Suwarsa O. Increased expression of interleukin-17A in the lesional skin indicates increase of serum antibody anti-phenolic glycolipid-I in leprosy patients. Biomed Biotechnol Res J 2017;1:147-50.  Back to cited text no. 5
  [Full text]  
6.
Han XY, Quintanilla M. Diffuse lepromatous leprosy due to Mycobacterium lepromatosis in quintana roo, mexico. J Clin Microbiol 2015;53:3695-8.  Back to cited text no. 6
    
7.
Han XY, Mistry NA, Thompson EJ, Tang HL, Khanna K, Zhang L. Draft genome sequence of new leprosy agent Mycobacterium lepromatosis. Genome Announc 2015;3:e00513-15.  Back to cited text no. 7
    
8.
Nunzie E, Cabrera LV, Moncayo FM, Espinosa PF, Clapasson A, Massone C. Lucio leprosy with Lucio's phenomenon, digital gangrene and anticardiolipin antibodies. Lepr Rev 2014;85:194-200.  Back to cited text no. 8
    
9.
Gunawan H, Yogya Y, Hafinah R, Marsella R, Ermawaty D, Suwarsa O. Reactive perforating leprosy, erythema multiforme-like reactions, sweet's syndrome-like reactions as atypical clinical manifestations of type 2 leprosy reaction. Int J Mycobacteriol 2018;7:97-100.  Back to cited text no. 9
[PUBMED]  [Full text]  
10.
Curi PF, Villaroel JS, Migliore N, Albertengo A, Aquino ML, Ceccato F, et al. Lucio's phenomenon: Report of five cases. Clin Rheumatol 2016;35:1397-401.  Back to cited text no. 10
    


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