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De novo histoid leprosy in a Colombian patient with multiple skin nodules on the ears and extremities

De novo histoid leprosy in a Colombian patient with multiple skin nodules on the ears and... Histoid leprosy is an uncommon form of lepromatous leprosy with distinct clinical, histopathological, immunological, and bacteriological features. This variant usually occurs in multibacillary patients who have irregular or inadequate treatment. Herein, we report a case of de novo histoid leprosy diagnosed in a patient from Cali, Colombia. In endemic areas, histoid leprosy should be in the differential diagnosis of any patient presenting with skin nodules. Early diagnosis and appropriate treatment are recommended for mitigating the impact of histoid leprosy cases, which are important reservoirs of Mycobacterium leprae. Keywords: Leprosy. Histoid leprosy. Mycobacterium leprae. Physical examination revealed skin phototype VI, and multiple INTRODUCTION firm, keloid-like, skin-colored cutaneous nodules of various sizes Leprosy, also known as Hansen’s disease, is a chronic localized on the ears (Figure 1), arms, thighs, and legs (Figure 2). infectious disease caused by the non-cultivable, acid-fast, rod- The lesions were not tender, and there were no sensory deficits. shaped bacterium Mycobacterium leprae, which has affected Complete blood count and renal function tests were normal. 1, humans since ancient times . Leprosy mainly affects the skin Rapid plasma reagin and HIV serology results were non-reactive. and peripheral nerves in most cases, apart from some other De novo histoid leprosy was highly suspected on the basis of structures, depending on the disease subtype . Histoid leprosy clinical findings. Considerations in the differential diagnosis for is an uncommon variant of lepromatous leprosy that is generally this patient included spontaneous keloids, dermatofibroma, and reported in multibacillary patients who have irregular or 2-4, lobomycosis. An slit-skin smear for acid-fast bacilli (AFB) taken inadequate treatment . Herein, we describe a case of de novo histoid leprosy in a Colombian patient. from the earlobes and nodules revealed a bacteriological index of 1+. Examination of a skin biopsy specimen from one nodule CASE REPORT demonstrated a dense inflammatory infiltrate within the dermis consisting primarily of histiocytes, admixed with occasional A 29-year-old unemployed man from Cali, Colombia perivascular plasma cells. Histoid leprosy was confirmed by presented to our hospital with a 1-year history of non-pruritic nodular skin lesions, which started on the anterior aspect of his skin biopsy examination using the Fite-Faraco stain, which right leg and progressed to involve his left leg, thighs, arms, demonstrated the presence of abundant easily identifiable neck, and ears. The patient’s medical history was otherwise AFB predominantly arranged in clumps within histiocytes unremarkable. He did not recall any contact with animals or (Figure 3). The patient was started on multibacillary-multidrug sick people. He had not received any treatment for his lesions. therapy (MB-MDT) consisting of dapsone 100mg/d, clofazimine 50mg/d, and a monthly course of clofazimine 300mg and rifampin 600mg for 1 year. The patient had a good response Corresponding author: Dr. Claudia J. Díaz to antimicrobials with no leprosy reactions or relapses. The Orcid: 0000-0003-3582-7748 Leprosy Control Program of the Secretaría de Salud Municipal e-mail: clajudiaz@yahoo.com of Cali performed monthly follow-up of the patient. Extensive Received 8 February 2017 Accepted 27 December 2017 education was provided by both the healthcare providers that 1/3 Piedrahíta-Rojas LM et al. - De novo histoid leprosy in a Colombian patient FIGURE 1: Nodular lesions involving the pinna of both ears (arrows). Earlobe infi ltration was a remarkable fi nding. FIGURE 3: Skin biopsy sample showing many acid-fast bacilli singly and in clumps inside histiocytes in the dermis (Fite-Faraco stain; original magnifi cation, ×1,000). immigration from endemic areas. According to offi cial reports from 143 countries and territories, the global registered prevalence of leprosy by the r fi st quarter of 2017 was 0.23 cases per 10,000 population (171,948 cases) . The clinical-histopathological spectrum based on the Ridley- Jopling classifi cation refl ects the interplay between M. leprae and the host’s cell-mediated immunity, and includes the five presentations: polar tuberculoid (TT), borderline tuberculoid (BT), borderline-borderline (BB), borderline lepromatous (BL), and polar lepromatous (LL); indeterminate leprosy (I) and pure neuritic leprosy are not considered in this spectrum 6, classifi cation . A simplifi ed classifi cation of leprosy based on the number of skin lesions and the bacillary load is used for the purpose of treatment. Multibacillary disease (BB, BL, and LL FIGURE 2: Multiple nodular lesions on the anterior aspect of the legs. forms), which typically correlates with having more than five skin lesions, represents the most infectious form of the disease first evaluated the case and the medical staff from the local and needs to be treated with MB-MDT, which is a combination program following the patient. of dapsone, rifampin, and clofazimine, for at least 12 months. Conversely, paucibacillary disease (TT, BT, and I forms) There were no other leprosy cases among the patient’s family 1, requires treatment with dapsone and rifampin for 6 months . members and close contacts based on signs and symptoms; nevertheless, Bacillus Calmette-Guérin (BCG) vaccination Histoid leprosy, first described by Dr. Herbert Windsor Wade was recommended. in the 1960s , is an uncommon variant of LL, affecting 1.1-3.6% 2- of all patients with leprosy and occurring predominantly in men DISCUSSION 4, . To date, there are no epidemiological data regarding histoid leprosy in Colombia, but a few cases have been described . Leprosy continues to be an important health problem in 1,5 Histoid leprosy usually occurs in multibacillary patients who several countries of southeast Asia, Africa, and the Americas . have irregular or inadequate treatment, particularly dapsone Cases in countries elsewhere are rare and usually linked to 2/3 Rev Soc Bras Med Trop Vol.:52:e20180226, 2019 monotherapy, but relapsing cases after successful treatment clinical manifestations of the disease as well as to highlight the as well as de novo cases have rarely been reported. Overall, importance of follow-up and treatment. treatment seems to influence the development of histoid leprosy To our knowledge, all of the present patient’s household 2,3 in most cases . Histoid leprosy cases serve as an important contacts were asymptomatic and disease-free based on clinical infection reservoir and as a source of incident cases; therefore, examination. However, subclinical leprosy cases might be it poses a threat to national leprosy eradication programs given overlooked due to a lack of resources and access to testing 10,11 the high bacillary load and the likely drug-resistant mutant . It has been reported modalities other than clinical examination 3,9 strains harbored . The disease is characterized by distinct that person-to-person transmission does not explain the patchy clinical, histopathological, immunological, and bacteriological epidemiology of the disease because incident cases are often 3,6,7, features . Clinical findings include painless, firm, cutaneous detected in persons with no known close contacts of leprosy . and/or subcutaneous papules, nodules or plaques with well- Periodic evaluation of household contacts for longer periods even defined edges and a smooth bright surface. The lesions are after treatment of the index case and use of laboratory testing usually located on the posterior and lateral aspects of the modalities (e.g., PCR or IgM anti-phenolic glycolipid [PGL-I]) 10,11 arms, dorsum of the hands, back, buttocks, thighs, legs, and may contribute to earlier detection of subclinical cases . over bony prominences such as the elbows and knees, with In conclusion, histoid leprosy is a rare presentation of LL a localized asymmetric pattern and arising from apparently that should be considered as a relevant differential diagnosis normal-looking skin. In contrast, classical LL presents with in endemic countries for patients presenting with skin nodules. generalized symmetric lesions that arise from infiltrated skin. Histopathological examination is needed for definitive Atypical skin lesions, such as tumor-like masses, molluscum diagnosis. Despite the low frequency of histoid leprosy among contagiosum-like lesions, xanthoma-like, mucosal, and genital all leprosy cases, early diagnosis and appropriate treatment are lesions, have been reported and may mislead the diagnosis of crucial to mitigate the impact of these important reservoirs of histoid leprosy . Nerve affectation, leading to anesthetic lesions resistant bacilli on the disease burden and spread worldwide. or sensory impairment to temperature, touch or pain, may be mild or absent. Earlobe infiltration could be present in some Conflict of interest: The authors declare that there is no conflict of interest. cases. The pathogenesis of histoid leprosy remains unclear but it is known that the cell-mediated and humoral immune responses against M. leprae in patients with histoid leprosy are enhanced REFERENCES compared with classical LL. This response includes increased 1. Britton WJ, Lockwood DNJ. Leprosy. Lancet 2004; 363(9416):1209-19. cluster of differentiation 36 (CD36) expression by keratinocytes, CD4 T lymphocytes, B lymphocytes, and immunoglobulin 2. Kaur I, Dogra S, De D, Saikia UN. Histoid leprosy: a retrospective study of 40 cases from India. Br J Dermatol. 2009;160(2):305-10. levels. However, macrophages seem to lack functionality to kill M. leprae bacteria present in high numbers in histoid lesions . 3. Gupta SK. Histoid leprosy: review of the literature. Int J Dermatol 2015;54(11):1283-8. The diagnosis of leprosy is initially clinical but must be 3,7 4. Mendiratta V, Jain A, Chander R, Khan A, Barara M. A nine-year supported by bacilloscopy and histopathology . Typical clinico-epidemiological study of Histoid Hansen in India. J Infect histopathological findings include epidermal atrophy with Dev Ctries. 2011;5(2):128-31. a subepidermal acellular band, known as the grenz zone. 5. World Health Organization. Global leprosy update, 2016: The leproma is contained within the dermis and is as a well- accelerating reduction of disease burden. Wkly Epidemiol Rec. circumscribed area consisting of spindle-shaped histiocytes 2017;92(35):501-20. arranged in an intertwining, whorled, or storiform pattern, 6. Ridley DS, Jopling WH. Classic fi ation of leprosy according to and surrounded by a pseudocapsule. An abundance of AFB, immunity. A v fi e-group system. Int J Lepr Other Mycobact Dis characteristically longer than ordinary lepra bacilli, can be 1966;34(3):255-73. seen within histiocytes, arranged singly, in clumps, or rarely 7. Wade H. The histoid variety of lepromatous leprosy. Int J Lepr. as globi. The identification of AFB on skin biopsy specimens 1963;31:129-42. stained by the Fite-Faraco stain helps to confirm the diagnosis. 8. Rodríguez G, Henríquez R, Gallo S, Panqueva C. Histoid leprosy On the other hand, skin smear examination under the with giant lesions of fingers and toes. Biomedica. 2015;35(2):165-70. microscope (bacilloscopy) by the Ziehl-Neelsen stain is useful 9. Palit A, Inamadar AC. Histoid leprosy as reservoir of the disease; a to classify the disease; if the presence of M. leprae is detected challenge to leprosy elimination. Lepr Rev. 2007;78:47-9. (positive bacilloscopy), the case is classified as multibacillary 10. Cardona-Castro N, Beltrán-Alzate JC, Romero-Montoya M.. leprosy. Histoid leprosy can clinically mimic dermatofibroma, Clinical, bacteriological and immunological follow-up of household contacts of leprosy patients from a post-elimination area - Antioquia, neurofibromatosis, xanthoma, cutaneous lymphoma, keloids, Colombia. Mem Inst Oswaldo Cruz 2009; 104(6):935-6. molluscum contagiosum, sarcoidosis, lobomycosis, and 3 11. Romero-Montoya M, Beltran-Alzate JC, Cardona-Castro N. leishmaniasis, among others . The standard treatment of histoid Evaluation and monitoring of Mycobacterium leprae transmission leprosy is MB-MDT. As with other leprosy cases, early detection in household contacts of patients with Hansen’s disease in Colombia. and appropriate treatment are encouraged to prevent future PLoS Negl Trop Dis. 2017;11(1):e0005325. disabilities and to stop disease transmission . Strict clinical 12. Franco-Paredes C, Rodríguez-Morales AJ. Unsolved matters in follow-up must be ensured during and after treatment. Education leprosy: a descriptive review and call for further research. Ann Clin is important to alert patients and household contacts about the Microbiol Antimicrob 2016;15(1):33. OPEN ACCESS https://creativecommons.org/licenses/by/4.0/ 3/3 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Revista da Sociedade Brasileira de Medicina Tropical Unpaywall

De novo histoid leprosy in a Colombian patient with multiple skin nodules on the ears and extremities

Piedrahíta-Rojas, Lina Marcela; Díaz, Claudia Juliana; Escandón-Vargas, Kevin
Revista da Sociedade Brasileira de Medicina Tropical – Jan 1, 2019
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0037-8682
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10.1590/0037-8682-0502-2016
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Abstract

Histoid leprosy is an uncommon form of lepromatous leprosy with distinct clinical, histopathological, immunological, and bacteriological features. This variant usually occurs in multibacillary patients who have irregular or inadequate treatment. Herein, we report a case of de novo histoid leprosy diagnosed in a patient from Cali, Colombia. In endemic areas, histoid leprosy should be in the differential diagnosis of any patient presenting with skin nodules. Early diagnosis and appropriate treatment are recommended for mitigating the impact of histoid leprosy cases, which are important reservoirs of Mycobacterium leprae. Keywords: Leprosy. Histoid leprosy. Mycobacterium leprae. Physical examination revealed skin phototype VI, and multiple INTRODUCTION firm, keloid-like, skin-colored cutaneous nodules of various sizes Leprosy, also known as Hansen’s disease, is a chronic localized on the ears (Figure 1), arms, thighs, and legs (Figure 2). infectious disease caused by the non-cultivable, acid-fast, rod- The lesions were not tender, and there were no sensory deficits. shaped bacterium Mycobacterium leprae, which has affected Complete blood count and renal function tests were normal. 1, humans since ancient times . Leprosy mainly affects the skin Rapid plasma reagin and HIV serology results were non-reactive. and peripheral nerves in most cases, apart from some other De novo histoid leprosy was highly suspected on the basis of structures, depending on the disease subtype . Histoid leprosy clinical findings. Considerations in the differential diagnosis for is an uncommon variant of lepromatous leprosy that is generally this patient included spontaneous keloids, dermatofibroma, and reported in multibacillary patients who have irregular or 2-4, lobomycosis. An slit-skin smear for acid-fast bacilli (AFB) taken inadequate treatment . Herein, we describe a case of de novo histoid leprosy in a Colombian patient. from the earlobes and nodules revealed a bacteriological index of 1+. Examination of a skin biopsy specimen from one nodule CASE REPORT demonstrated a dense inflammatory infiltrate within the dermis consisting primarily of histiocytes, admixed with occasional A 29-year-old unemployed man from Cali, Colombia perivascular plasma cells. Histoid leprosy was confirmed by presented to our hospital with a 1-year history of non-pruritic nodular skin lesions, which started on the anterior aspect of his skin biopsy examination using the Fite-Faraco stain, which right leg and progressed to involve his left leg, thighs, arms, demonstrated the presence of abundant easily identifiable neck, and ears. The patient’s medical history was otherwise AFB predominantly arranged in clumps within histiocytes unremarkable. He did not recall any contact with animals or (Figure 3). The patient was started on multibacillary-multidrug sick people. He had not received any treatment for his lesions. therapy (MB-MDT) consisting of dapsone 100mg/d, clofazimine 50mg/d, and a monthly course of clofazimine 300mg and rifampin 600mg for 1 year. The patient had a good response Corresponding author: Dr. Claudia J. Díaz to antimicrobials with no leprosy reactions or relapses. The Orcid: 0000-0003-3582-7748 Leprosy Control Program of the Secretaría de Salud Municipal e-mail: clajudiaz@yahoo.com of Cali performed monthly follow-up of the patient. Extensive Received 8 February 2017 Accepted 27 December 2017 education was provided by both the healthcare providers that 1/3 Piedrahíta-Rojas LM et al. - De novo histoid leprosy in a Colombian patient FIGURE 1: Nodular lesions involving the pinna of both ears (arrows). Earlobe infi ltration was a remarkable fi nding. FIGURE 3: Skin biopsy sample showing many acid-fast bacilli singly and in clumps inside histiocytes in the dermis (Fite-Faraco stain; original magnifi cation, ×1,000). immigration from endemic areas. According to offi cial reports from 143 countries and territories, the global registered prevalence of leprosy by the r fi st quarter of 2017 was 0.23 cases per 10,000 population (171,948 cases) . The clinical-histopathological spectrum based on the Ridley- Jopling classifi cation refl ects the interplay between M. leprae and the host’s cell-mediated immunity, and includes the five presentations: polar tuberculoid (TT), borderline tuberculoid (BT), borderline-borderline (BB), borderline lepromatous (BL), and polar lepromatous (LL); indeterminate leprosy (I) and pure neuritic leprosy are not considered in this spectrum 6, classifi cation . A simplifi ed classifi cation of leprosy based on the number of skin lesions and the bacillary load is used for the purpose of treatment. Multibacillary disease (BB, BL, and LL FIGURE 2: Multiple nodular lesions on the anterior aspect of the legs. forms), which typically correlates with having more than five skin lesions, represents the most infectious form of the disease first evaluated the case and the medical staff from the local and needs to be treated with MB-MDT, which is a combination program following the patient. of dapsone, rifampin, and clofazimine, for at least 12 months. Conversely, paucibacillary disease (TT, BT, and I forms) There were no other leprosy cases among the patient’s family 1, requires treatment with dapsone and rifampin for 6 months . members and close contacts based on signs and symptoms; nevertheless, Bacillus Calmette-Guérin (BCG) vaccination Histoid leprosy, first described by Dr. Herbert Windsor Wade was recommended. in the 1960s , is an uncommon variant of LL, affecting 1.1-3.6% 2- of all patients with leprosy and occurring predominantly in men DISCUSSION 4, . To date, there are no epidemiological data regarding histoid leprosy in Colombia, but a few cases have been described . Leprosy continues to be an important health problem in 1,5 Histoid leprosy usually occurs in multibacillary patients who several countries of southeast Asia, Africa, and the Americas . have irregular or inadequate treatment, particularly dapsone Cases in countries elsewhere are rare and usually linked to 2/3 Rev Soc Bras Med Trop Vol.:52:e20180226, 2019 monotherapy, but relapsing cases after successful treatment clinical manifestations of the disease as well as to highlight the as well as de novo cases have rarely been reported. Overall, importance of follow-up and treatment. treatment seems to influence the development of histoid leprosy To our knowledge, all of the present patient’s household 2,3 in most cases . Histoid leprosy cases serve as an important contacts were asymptomatic and disease-free based on clinical infection reservoir and as a source of incident cases; therefore, examination. However, subclinical leprosy cases might be it poses a threat to national leprosy eradication programs given overlooked due to a lack of resources and access to testing 10,11 the high bacillary load and the likely drug-resistant mutant . It has been reported modalities other than clinical examination 3,9 strains harbored . The disease is characterized by distinct that person-to-person transmission does not explain the patchy clinical, histopathological, immunological, and bacteriological epidemiology of the disease because incident cases are often 3,6,7, features . Clinical findings include painless, firm, cutaneous detected in persons with no known close contacts of leprosy . and/or subcutaneous papules, nodules or plaques with well- Periodic evaluation of household contacts for longer periods even defined edges and a smooth bright surface. The lesions are after treatment of the index case and use of laboratory testing usually located on the posterior and lateral aspects of the modalities (e.g., PCR or IgM anti-phenolic glycolipid [PGL-I]) 10,11 arms, dorsum of the hands, back, buttocks, thighs, legs, and may contribute to earlier detection of subclinical cases . over bony prominences such as the elbows and knees, with In conclusion, histoid leprosy is a rare presentation of LL a localized asymmetric pattern and arising from apparently that should be considered as a relevant differential diagnosis normal-looking skin. In contrast, classical LL presents with in endemic countries for patients presenting with skin nodules. generalized symmetric lesions that arise from infiltrated skin. Histopathological examination is needed for definitive Atypical skin lesions, such as tumor-like masses, molluscum diagnosis. Despite the low frequency of histoid leprosy among contagiosum-like lesions, xanthoma-like, mucosal, and genital all leprosy cases, early diagnosis and appropriate treatment are lesions, have been reported and may mislead the diagnosis of crucial to mitigate the impact of these important reservoirs of histoid leprosy . Nerve affectation, leading to anesthetic lesions resistant bacilli on the disease burden and spread worldwide. or sensory impairment to temperature, touch or pain, may be mild or absent. Earlobe infiltration could be present in some Conflict of interest: The authors declare that there is no conflict of interest. cases. The pathogenesis of histoid leprosy remains unclear but it is known that the cell-mediated and humoral immune responses against M. leprae in patients with histoid leprosy are enhanced REFERENCES compared with classical LL. This response includes increased 1. Britton WJ, Lockwood DNJ. Leprosy. Lancet 2004; 363(9416):1209-19. cluster of differentiation 36 (CD36) expression by keratinocytes, CD4 T lymphocytes, B lymphocytes, and immunoglobulin 2. Kaur I, Dogra S, De D, Saikia UN. Histoid leprosy: a retrospective study of 40 cases from India. Br J Dermatol. 2009;160(2):305-10. levels. However, macrophages seem to lack functionality to kill M. leprae bacteria present in high numbers in histoid lesions . 3. Gupta SK. Histoid leprosy: review of the literature. Int J Dermatol 2015;54(11):1283-8. The diagnosis of leprosy is initially clinical but must be 3,7 4. Mendiratta V, Jain A, Chander R, Khan A, Barara M. A nine-year supported by bacilloscopy and histopathology . Typical clinico-epidemiological study of Histoid Hansen in India. J Infect histopathological findings include epidermal atrophy with Dev Ctries. 2011;5(2):128-31. a subepidermal acellular band, known as the grenz zone. 5. World Health Organization. Global leprosy update, 2016: The leproma is contained within the dermis and is as a well- accelerating reduction of disease burden. Wkly Epidemiol Rec. circumscribed area consisting of spindle-shaped histiocytes 2017;92(35):501-20. arranged in an intertwining, whorled, or storiform pattern, 6. Ridley DS, Jopling WH. Classic fi ation of leprosy according to and surrounded by a pseudocapsule. An abundance of AFB, immunity. A v fi e-group system. Int J Lepr Other Mycobact Dis characteristically longer than ordinary lepra bacilli, can be 1966;34(3):255-73. seen within histiocytes, arranged singly, in clumps, or rarely 7. Wade H. The histoid variety of lepromatous leprosy. Int J Lepr. as globi. The identification of AFB on skin biopsy specimens 1963;31:129-42. stained by the Fite-Faraco stain helps to confirm the diagnosis. 8. Rodríguez G, Henríquez R, Gallo S, Panqueva C. Histoid leprosy On the other hand, skin smear examination under the with giant lesions of fingers and toes. Biomedica. 2015;35(2):165-70. microscope (bacilloscopy) by the Ziehl-Neelsen stain is useful 9. Palit A, Inamadar AC. Histoid leprosy as reservoir of the disease; a to classify the disease; if the presence of M. leprae is detected challenge to leprosy elimination. Lepr Rev. 2007;78:47-9. (positive bacilloscopy), the case is classified as multibacillary 10. Cardona-Castro N, Beltrán-Alzate JC, Romero-Montoya M.. leprosy. Histoid leprosy can clinically mimic dermatofibroma, Clinical, bacteriological and immunological follow-up of household contacts of leprosy patients from a post-elimination area - Antioquia, neurofibromatosis, xanthoma, cutaneous lymphoma, keloids, Colombia. Mem Inst Oswaldo Cruz 2009; 104(6):935-6. molluscum contagiosum, sarcoidosis, lobomycosis, and 3 11. Romero-Montoya M, Beltran-Alzate JC, Cardona-Castro N. leishmaniasis, among others . The standard treatment of histoid Evaluation and monitoring of Mycobacterium leprae transmission leprosy is MB-MDT. As with other leprosy cases, early detection in household contacts of patients with Hansen’s disease in Colombia. and appropriate treatment are encouraged to prevent future PLoS Negl Trop Dis. 2017;11(1):e0005325. disabilities and to stop disease transmission . Strict clinical 12. Franco-Paredes C, Rodríguez-Morales AJ. Unsolved matters in follow-up must be ensured during and after treatment. Education leprosy: a descriptive review and call for further research. Ann Clin is important to alert patients and household contacts about the Microbiol Antimicrob 2016;15(1):33. OPEN ACCESS https://creativecommons.org/licenses/by/4.0/ 3/3

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Published: Jan 1, 2019

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