Autochthonous lepromatous leprosy in a Spanish woman with burns on both feet and skin lesions.
Leprosy may be defined as a chronic contagious granulomatous disease that usually involves the skin and peripheral nervous system. The current incidence of leprosy is very low in developed countries, (1-4) but some autochthonous cases could be observed worldwide. Lepromatous leprosy is a type of leprosy characterised by many lesions with bacteria, hair loss, nerve involvement, limb weakness and disfigurement. (5) Due to its rarity, physicians could not be aware of the possibility of autochthonous transmission in developed countries, so the knowledge of clinical manifestations and diagnostic procedures is still necessary. In this report, we present a case of autochthonous lepromatous leprosy in a woman from Spain that was diagnosed due to the presentation with burns on both feet.
A 55 year old Spanish female was admitted at the Plastic Surgery Department to our hospital for performing skin grafts on both feet due to severe burns from hot water. During the initial interview, the patient reported some non-pruritic skin lesions in both legs, arms and trunk for 4 years. A physical examination revealed numerous lesions in both legs, of vascular aspect, that fuse forming plaques with whitish centre. The lesions were arranged asymmetrically, irregular edges, resizable and with conservation of rude tactile sensitivity (Figure 1A). Moreover, the patient had madarosis in the eyelashes and eyebrows (Figure 1B). In the left eye, ectropion and lagophthalmos was observed and keratitis punctata was discovered by the ophthalmologist. On both feet, the patient had second-degree burns and deformities compatible with neuropathic arthropathy (Figure 1C). No thickening of the radial, medianus, peroneal and ulnar nerves was detected.
After debridement and performing of skin grafts, some cutaneous biopsies were taken for both histopathologic and microbiologic study. Histopathologic examination by means of a Haematoxyllin-eosin stain showed chronic inflammation signs with a perivascular infiltrate of neutrophils and histiocytes without the presence of granulomas. No Ziehl-Neelsen stain was performed in the pathology laboratory.
However, in the microbiology laboratory a Ziehl-Neelsen stain with a small sample obtained from a cutaneous biopsy was carried out, but no fast-acid bacilli (FAB) were observed. Due to the high index of suspicion of leprosy, the biopsy was placed on saline solution with glass beads and a mechanical rupture by vortexing was performed and then centrifugated. A new Ziehl-Neelsen stain was then carried out from the pellet, showing FAB and globi (Figure 1D). A specific polymerase chain reaction (PCR) for Mycobacterium leprae from the biospsy was performed and a positive result was obtained. Molecular study for resistance was carried out and the microorganism was susceptible to rifampicin, dapsone and ofloxacin (Genotype LepraeDR, Hain-Lifescience, Nehren, Germany) (performed in the Mycobacteria Reference Centre, Oviedo, Spain). A diagnosis of lepromatous leprosy was established and treatment with rifampicin (600mg/day), dapsone (100mg/day) and clofazimine (100mg/48 hours) was started and prescribed during 2 years. At 6 months of follow-up, the patient remained clinically stable with a favourable outcome.
The incidence of leprosy is currently decreasing worldwide, (1-4) although the new case detection rate remains high, with about 250,000 new cases being registered each year. (2) In Spain, according the leprosy registration state, 11 new cases were diagnosed in 2014 as well as eight in 2015, and the majority of them were seen in the immigrant population. (3,4) In 2015, the number of prevalent cases was 34, lower than the previous year (n = 40), being the prevalence of 0-0073 cases per 10,000 inhabitants. These data show that leprosy in Spain is mainly an imported disease, although some autochthonous cases have been also seen.
Leprosy diagnosis in developed countries requires now a high index of suspicion, due to being a rare and chronic disease whose symptomatology is appearing slowly and progressively. Diagnosis may be difficult, because the direct stain from the sample can be negative, as in our case, so several procedures such as the mechanical rupture of the biopsy could be necessary for the correct visualization. Moreover, histopathologic findings are usually non-specific due to the absence of granulomas, as occurred in the lepromatous form.
Global leprosy eradication is difficult, (1,2) although its control involves an early diagnosis and a rapid and correct therapy. Some days after the treatment has begun, the patient is not contagious.
The treatment of choice of leprosy is based on rifampicin and dapsone association; clofazimine may be added in the cases of lepromatous leprosy. Although drug resistance does not represent a threat, the experts in the field advise about a treatment monitoring in all cases. (1,2) With regard to drug resistance study, the most appropriate method for its detection is the DNA sequencing, because mutations in three genes involved on the rifampicin, ofloxacin and dapsone resistance have been already identified. (1,2)
In summary, although leprosy is decreasing worldwide and is an unusual disease in developed countries, physicians should be still aware about this entity, including autochthonous population, in order to make a correct diagnosis and avoiding severe complications.
(1) Renault CA, Ernst JD. Mycobacterium leprae (Leprosy). In: Bennett JE, Dolin R, Blaser MJ (eds). Mandell, Douglas and Bennett's principles and practice of infectious diseases. 8[TM] edition, Philadelphia: Elsevier, 2015; pp. 2819-2831.e2.
(2) Rodrigues LC, Lockwood DN. Leprosy now: epidemiology, progress, challenges, and research gaps. Lancet Infect Dis, 2011; 11: 464-470.
(3) Rodriguez E, Diaz O. Situacion epidemiologica de la lepra en el ano 2014. Bol Epidemiol Semanal, 2015; 22: 236-239.
(4) Diaz O, Rodriguez E. Vigilancia de la lepra en Espana en 2015 y situation mundial. Bol Epidemiol Semanal, 2016; 24: 20-33.
(5) Walker SL, Lockwood DN. Leprosy. Clin Dermatol, 2007; 25: 165-172.
CRISTINA GOMEZ-CAMARASA *, JAVIER RODRIGUEZ-GRANGER *, OBDULIA CANADAS-MORENO **, ANTONIO SAMPEDRO *, LUIS ALIAGA-MARTINEZ *** & FERNANDO COBO *
* Department of Microbiology, University Hospital Virgen de las Nieves, Granada, Spain
** Department of Plastic Surgery, University Hospital Virgen de las Nieves, Granada, Spain
*** Department of Internal Medicine, University Hospital Virgen de las Nieves, Granada, Spain
Accepted for publication 5 October 2016
Correspondence to: Fernando Cobo, Department of Microbiology, University Hospital Virgen de las Nieves, Avda Fuerzas Armadas, 2, 18014 Granada, Spain (Tel: + 34958020364; Fax: + 34958241245; e-mail: fernando. firstname.lastname@example.org)
Caption: Figure 1. (A) Leprosy chronic skin lesions at the lower member. (B) Madarosis can be seen in the eyelashes and eyebrows, as well as the hypertrophy of the eyebrows folds. (C) Second-degree burns and deformities compatible with neuropathic arthropathy. (D) Ziehl-Neelsen stain from the pellet of the skin biopsy after mechanical rupture with glass beads. The photograph shows FAB and some globi (see the arrows).
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|Title Annotation:||CASE REPORT|
|Author:||Gomez-Camarasa, Cristina; Rodriguez-Granger, Javier; Canadas-Moreno, Obdulia; Sampedro, Antonio; Ali|
|Date:||Dec 1, 2016|
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