Original article / research

Year : 2024 | Month : July | Volume : 18 | Issue : 7 | Page : WD01 - WD03

Full Version

An Unusual Presentation of Co-existence of Leprosy, Anetoderma and Abdominal Tuberculosis: A Case Report

Published: July 1, 2024 | DOI: https://doi.org/10.7860/JCDR/2024/69245.19577

Shreya Nirajkumar Gupta, Meenakshi Chandak, Adarshlata Singh, Bhushan Madke, Sugat Jawade

1. Junior Resident, Department of Dermatology, Datta Meghe Instiute of Higher Education and Research, Wardha, Maharashtra, India. 2. Senior Resident, Department of Dermatology, Datta Meghe Instiute of Higher Education and Research, Wardha, Maharashtra, India. 3. Professor, Department of Dermatology, Datta Meghe Instiute of Higher Education and Research, Wardha, Maharashtra, India. 4. Professor, Department of Dermatology, Datta Meghe Instiute of Higher Education and Research, Wardha, Maharashtra, India. 5. Professor, Department of Dermatology, Datta Meghe Instiute of Higher Education and Research, Wardha, Maharashtra, India.

Correspondence Address :
Dr. Shreya Nirajkumar Gupta,
Datta Meghe Instiute of Higher Education and Research, Sawangi, Wardha-442107, Maharashtra, India.
E-mail: sngsng2096@gmail.com

Abstract

Leprosy and Tuberculosis (TB), both significant mycobacterial infections, often affect populations with lowered specific immunity. TB is a widespread and life-threatening mycobacterial infection. Co-infection of these diseases is uncommon in routine clinical practice, adding complexity to diagnosis and treatment planning. However, instances of co-existence suggest a potential cross-immunity theory. Leprosy, known for its varied presentations, can manifest as an asymptomatic secondary presentation of anetoderma, characterised by elastinolysis-induced loose sac-like appearances. This case report presents a middle-aged man in his late 30s undergoing Anti-Tubercular Treatment (AKT) for abdominal tuberculosis, who presented with signs of Erythema Nodosum Leprosum (ENL) and small asymptomatic atrophied macules on his trunk and back in the Dermatology department. Through comprehensive history, clinical examination, slit skin smear, and biopsy, authors elucidated a rare case of co-infection of leprosy and atypical cutaneous manifestations as anetoderma, concomitant with abdominal tuberculosis.

Keywords

Atrophied macules, Cutaneous manifestations, Reinfection, Relapse, Tuberculoid leprosy

Case Report

A 37-year-old male visited the Dermatology Outpatient Department (OPD) with complaints of fever, tingling, and asymptomatic red lesions over both limbs, back, and trunk for 15 days (Table/Fig 1). In the past, the patient confirmed being a diagnosed case of Borderline Tuberculoid leprosy almost three years back and had completed a one-year treatment of Multibacillary Multidrug Treatment (MBMDT). Since then, he had not experienced any symptoms of Hansen’s disease.

The current macular lesions, which appeared on new sites, were not involved when he was previously diagnosed with Hansen’s disease. He was also a chronic alcoholic and had been diagnosed with abdominal tuberculosis, confirmed on Ultrasonography (USG) guided ascitic fluid cytology, showing raised Adenosine Deaminase (ADA) levels (86.77 IU/mL, cut-off titre: >40 IU/mL) (1),(2). He had been on Anti-Tubercular Drugs (AKT) for two months. A provisional diagnosis of lepra reaction was made, and he was prescribed oral steroids (prednisolone 20 mg daily) along with antacids (pantoprazole). The erythematous lesions resolved on follow-up after a week, but he still complained of tingling over limbs and sporadic fever episodes over the last two days. Upon general examination, new hypopigmented macules and papule lesions were observed over new sites (Table/Fig 2), along with old atrophied lesions on his back (Table/Fig 3). Gyanecomastia was also noted (Table/Fig 4). The clinical diagnosis was revised to Hansen’s disease with relapse, along with abdominal tuberculosis and anetoderma. Further investigations were advised for confirmation of anetoderma and relapse. Sensory tests for leprosy examination, including pinprick tests, cold-hot differentiation tests, and motor examinations like card, book, and pen tests, were not positive. There were no signs of nerve thickening or neuritis presentation upon palpation. Slit skin smear and biopsy from the hypopigmented macular and atrophied lesions were advised for histopathological examinations. The slit skin smear revealed +2 and +1 (as per Ridley’s logarithmic scale for Bacteriological index) (3) bacterial load from the earlobe and eyebrow, respectively. Histopathological findings from the atrophied lesion indicated fragmented elastic fibers with areas lacking elastic tissue, suggestive of anetoderma using Mason Trichrome stain (Table/Fig 5), and from the hypopigmented macular lesion, haematoxylin and eosin staining was consistent with Lepromatous leprosy (Table/Fig 6).

The patient was started on MBMDT drugs, including Clofazimine 300 mg, Ofloxacin 400 mg, and Dapsone 300 mg on the first day, followed by Clofazimine 50 mg and Dapsone 100 mg for 28 days, along with ongoing AKT treatment and 20 mg of prednisolone.

The ongoing AKT for the past two months, targeting abdominal tuberculosis, consisted of the HRZE regimen (Isoniazid 75 mg, Rifampicin 150 mg, Pyrazinamide 400 mg, and Ethambutol 275 mg), with 4 tablets a day prescribed based on the weight range.

Based on the history, cutaneous examination, and guidelines provided by the National Leprosy Eradication Program (NLEP), which states that “Recurrence of disease at any time after completion of full treatment of MBMDT, irrespective of their clinical status (4),(5), is a case of relapse,” this case was diagnosed as a relapse of Hansen’s disease and anetoderma co-existing with abdominal TB. However, whole gene sequencing is considered the gold standard for distinguishing reinfection from relapse of leprosy (6). Due to the unavailability of molecular testing at the present institution, further subtype differentiation couldn’t be obtained.

Discussion

Leprosy represents one of the oldest mycobacterial infections, impacting large populations. It manifests as an immune-mediated condition, presenting with diverse neurocutaneous involvement. Another disease caused by the mycobacteria genus is TB, which is the most prevalent infection, posing a threat to nearly 10 million lives (7),(8).

TB spreads through inhalation, causing both pulmonary and extrapulmonary manifestations. Research indicates potential cross-infections between Leprosy and TB, especially among populations of low socio-economic status or with limited access to healthcare services (9). While encountering both TB and Leprosy in the same endemic setting is rare, evidence of their co-existence is documented in the literature. Leprosy, known as a great imitator, can mimic various diseases and also serves as a secondary cause for a specific loose skin condition termed anetoderma (9),(10).

Anetoderma (relaxed skin in Greek) is an atrophic idiopathic condition mainly presenting as a loose wrinkled outpouching of skin. The reason for this is connective tissue weakening in the dermis and mid-dermal elastic tissue degeneration. It is classified into primary type, which is idiopathic, while secondary anetoderma is associated with an underlying pathology (11). Primary anetoderma exhibits macules on normal skin, while secondary anetoderma involves an inflammatory and infiltrative dermatosis. Secondary anetoderma is further categorised based on the presence or absence of inflammation. a) Schweninger-Buzzi anetoderma lacks preceding inflammation, whereas b) Jadassohn-Pellizzari anetoderma shows atrophic lesions after inflammation, appearing as small round to oval lesions of 0.5 to 1 cm, typically seen in individuals aged 20 to 40 years (12). Secondary anetoderma linked with dermatoses includes lupus erythematosus, acne vulgaris, tuberculosis, leprosy, and varicella. Though the exact cause remains unclear, evidence suggests elastin tissue breakdown. Diagnosis relies on clinical assessment, confirmed by histopathology, with no established treatment available.

The co-existence with active leprosy cases or misdiagnosis as leprosy necessitates histopathological confirmation (12). The coexistence of leprosy and TB can influence the clinical presentation of both, posing challenges in treatment regimens (13). Mangum L et al., discussed co-infections, noting TB as a common finding in borderline and lepromatous leprosy cases. Impaired cell-mediated immunity in multibacillary leprosy patients may predispose them to TB co-infection (14). A study proposed a cross-immunity link between TB and leprosy, emphasising the role of innate immunity in the broad-spectrum presentation of leprosy. Elevated proinflammatory cytokine responses like Interferon (IFN) gamma and Tumour Necrosis Factor (TNF) alpha are crucial in leprosy, also detected in TB cases. Different mycobacterial strains can trigger an inflammatory response on Interleukin mediators, establishing a potential link between them (15). Individuals with immune TB might display some immunity to leprosy (13). Stefani MMA et al., stated the use of Whole Genome Sequencing (WGS) distinguishes between relapse and reinfection in recurrent leprosy cases (6). They stated that relapse and reinfection cannot be differentiated clinically, and molecular genotyping of a predefined set of loci has limited resolution due to exceptional Mycobacterium leprae genome conservation and low sequence diversity between strains from the same geographical area. The clear evidence for reinfection is an unrelated strain of bacilli in the first and second diagnosis whereas the true relapses are due to minor strain difference (14). (Table/Fig 7) gives an overview of a comparison between the findings of the present case and cases reported in the past literature (16),(17). As per the NLEP, Disability Prevention, and Medical Rehabilitation Guidelines for Primary, Secondary, and Tertiary Care issued by the Central Leprosy Division (2023) (5), the criteria for relapse is that the time since completion of treatment is usually more than three years and for a reaction, it is less than three years. The presented case had lesions over new places after approximately three years of initial MDMDT completion. Thus, on comparative analysis between the published reports by various authors and findings of the presented case, it is a relapse case of lepromatous leprosy with atrophied lesions of anetoderma co-existing with abdominal tuberculosis.

Conclusion

The significance of reporting this case is due to the rare co-existence of two mycobacterial diseases coupled with cutaneous degeneration of elastin fibers as a relapse of lepromatous leprosy, abdominal tuberculosis, along with anetoderma. This reporting will help treating physicians in diagnosing and planning the treatment for the same.

Acknowledgement

Dr. Sunil Kumar, Dr. Kishor Hiwale, Dr. Samarth Shukla, Dr. Preeti Mishra for their expert opinion on this case.

Reference


1.	Rao SD, Adusumalli S, Mazurek JA. Pulmonary hypertension in heart failure patients. Card Fail Rev. 2020r;6:e05. [CrossRef] [PubMed]
2.	Koff A, Azar MM. Diagnosing peritoneal tuberculosis. BMJ Case Rep. 2020;13(2):e233131. [CrossRef] [PubMed]
3.	Naik SM, More SA, Joshi SR. Correlation of histomorphological findings with bacteriological index in leprosy patients. Iranian J Pathol. 2022;17(1):48. [CrossRef] [PubMed]
4.	Kaimal S, Thappa DM. Relapse in leprosy. Indian J Dermatol Venereol Leprol. 2009;75(2):126-35. [CrossRef] [PubMed]
5.	National Leprosy Eradication Program. Disability Prevention & Medical Rehabilitation. Guidelines for Primary, Secondary and Tertiary Level Care. Nirman Bhavan: New Delhi Government of India: 2012.
6.	Stefani MMA, Avanzi C, Bührer-Sékula S, Benjak A, Loiseau C, Singh P, et al. Whole genome sequencing distinguishes between relapse and reinfection in recurrent leprosy cases. PLoS Negl Trop Dis. 2017;11(6):e0005598. [CrossRef] [PubMed]
7.	Shetty S, Umakanth S, Manandhar B, Nepali PB. Coinfection of leprosy and tuberculosis. BMJ Case Rep. 2018;2018:bcr2017222352. [CrossRef] [PubMed]
8.	Keragala BSDP, Herath HMMTB, Janapriya GHDC, Vanitha S, Balendran T, Janani T, et al. Coexistence of mycobacterial infections-Mycobacterium tuberculosis and Mycobacterium leprae-in Sri Lanka: A case series. J Med Case Reports. 2020;14(1):101. [CrossRef] [PubMed]
9.	Garg G, Gogia A, Kakar A. Leprosy: A great mimicking disease. Indian J Lepr. 2016;88(2):123-27.
10.	Parajuli N, Shrestha S, Lama L, Shrestha R, Tiwari S, Karki A. Multibacillary leprosy presenting as anetoderma in a young teenager. Case Rep Dermatol Med. 2020;2020:01-03. [CrossRef] [PubMed]
11.	Paller AS, Mancini AJ. Collagen Vascular Disorders. In: Hurwitz Clinical Pediatric Dermatology [Internet]. Elsevier; 2011. p. 497-527. Available from: https:// linkinghub.elsevier.com/retrieve/pii/B9781437704129000228. [CrossRef]
12.	Razmi TM, Narang T, Chatterjee D, Dogra S, Saikia UN. Lepromatous leprosy masquerading as anetoderma in a boy. Indian J Dermatol Venereol Leprol. 2020;86(2):176-77. [CrossRef] [PubMed][Internet]. International textbook of leprosy 1
13.	st ed. American Leprosy Missions; 2016. [Cited 2023 Dec 20]. Available from: https://internationaltextbookofleprosy.org/.
14.	Mangum L, Kilpatrick D, Stryjewska B, Sampath R. Tuberculosis and leprosy coinfection: A perspective on diagnosis and treatment. Open Forum Infect Dis. 2018;5(7):ofy133. [CrossRef] [PubMed]
15.	Crespo F, White J, Roberts C. Revisiting the tuberculosis and leprosy cross-immunity hypothesis: Expanding the dialogue between immunology and paleopathology. Int J Paleopathol. 2019;26:37-47. [CrossRef] [PubMed]
16.	Shen J, Yan L, Sun P. Clinical features of relapse after multidrug therapy for leprosy in China. Lepr Rev. 2015;86(2):165-69. [CrossRef] [PubMed]
17.	Chagas DF, Diniz LM, Lucas EA, Moraes MO de. Relapse in leprosy and drug resistance assessment in a tertiary hospital of the state of Espírito Santo, Brazil. Rev Soc Bras Med Trop. 2021;54:e0375-2020, S0037-86822021000100626. [CrossRef] [PubMed]

DOI and Others

DOI: 10.7860/JCDR/2024/69245.19577

Date of Submission: Dec 23, 2023
Date of Peer Review: Mar 09, 2024
Date of Acceptance: Apr 12, 2024
Date of Publishing: Jul 01, 2024

AUTHOR DECLARATION:
• Financial or Other Competing Interests: None
• Was informed consent obtained from the subjects involved in the study? No
• For any images presented appropriate consent has been obtained from the subjects. No

PLAGIARISM CHECKING METHODS:
• Plagiarism X-checker: Dec 23, 2023
• Manual Googling: Mar 19, 2024
• iThenticate Software: Apr 10, 2024 (5%)

ETYMOLOGY: Author Origin

EMENDATIONS: 5

Tables and Figures

[Table/Fig-1]

[Table/Fig-2]

[Table/Fig-3]

[Table/Fig-4]

[Table/Fig-5]

[Table/Fig-6]

[Table/Fig-7]