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Int J STD AIDS OnlineFirst, published on March 13, 2015 as doi:10.1177/0956462415577728
Case report International Journal of STD & AIDS 0(0) 1–3 ! The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0956462415577728 std.sagepub.com
An unusual perineal plaque Sumeet Hindocha, Shoma Banerjee and Fiona Lewis
Abstract We present a 60 year old male with a long-standing perineal and scrotal lesion. Investigations eventually revealed cutaneous tuberculosis, with complete resolution after appropriate treatment. It highlights the variable presentation of cutaneous tuberculosis and the importance of considering the diagnosis in chronic lesions.
Keywords Tuberculosis, perineal, ano-perineal, extrapulmonary Date received: 3 January 2015; accepted: 24 February 2015
Introduction Patients with genital lesions may present late in the course of their disease, when the features may have been modified by inappropriate treatment. We present a patient with a longstanding perineal and scrotal lesion, misdiagnosed for many years as psoriasis, but eventually shown to be cutaneous tuberculosis, which responded well to treatment. It illustrates the importance of clinico-pathological correlation and highlights that rare diseases can present at unusual sites.
Case report A 61-year-old man presented with a perineal lesion, which had been present for 20 years. It had been diagnosed at the time as psoriasis and treated with potent topical steroids intermittently since. He had no systemic or local symptoms and his only concern was that the lesion was increasing in size. He had a medical history of ischaemic heart disease and there was no relevant family history. He was a heterosexual married man who had relocated from India in the 1970s, and now worked in an office. On examination, a firm indurated plaque with superficial ulceration was noted on the perineum extending to the scrotum and right buttock (Figure 1a). There were no palpable inguinal lymph nodes. The clinical differential diagnoses included ano-genital Crohn’s disease, hypertrophic lichen planus, cutaneous lymphoma or a granulomatous disorder such as sarcoidosis or tuberculosis. Laboratory investigations, including haematological parameters, renal and liver function and serum angiotensin converting enzyme were
normal and his chest X-ray was clear. A TB-elispot test was positive. A punch biopsy was taken from the plaque which showed multiple non-caseating granulomata and a dense inflammatory infiltrate (Figure 2). No acid-fast bacilli were seen on Ziehl-Neelsen staining. Fungal stains were negative. A diagnosis of cutaneous perineal tuberculosis was made and he was commenced on anti-tuberculosis therapy (rifampicin, isoniazid, pyrazinamide and ethambutol for two months followed by a further four-month course of rifampicin and isoniazid). There was a dramatic response within just three weeks of starting treatment. He completed a six-month course of therapy and the plaque completely resolved with some post-inflammatory hypopigmentation (Figure 1b).
Discussion Extrapulmonary tuberculosis accounts for 15% of all cases and can affect any organ.1 Ano-perineal tuberculosis is a very rare form of extrapulmonary disease, accounting for less than 1% of cases. It presents in many ways2 making diagnosis a challenge. It can exist alone, or be associated with gastrointestinal tuberculosis, via direct extension or haematological or lymphatic spread. Anal fistula is the most common presentation but other
Department of Dermatology, Wexham Park Hospital, Slough, UK Corresponding author: Fiona Lewis, Department of Dermatology, Wexham Park Hospital, Slough, SL2 4HL, UK. Email: [email protected]
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International Journal of STD & AIDS 0(0)
Figure 1. (a) Ulcerated perineal plaque; (b) resolution of plaque after treatment.
The appearance of cutaneous lesions in the ano-genital area can be significantly altered due to the moist environment and this renders clinical assessment alone challenging in making a final diagnosis. In this case, there were no deep fissures or other clinical signs to suggest cutaneous Crohn’s disease and he had no gastro-intestinal complaints. If bowel symptoms are present, it is important to exclude gastrointestinal tuberculosis as this can mimic Crohn’s disease.8 He was otherwise well with no other symptoms to suggest sarcoid or systemic disease. Occasionally, sarcoid-like histological patterns are seen in patients with cutaneous tuberculosis and positive immunoassays are helpful.9 A thorough history and examination is the starting point in diagnosis and helps to identify extra-cutaneous foci of infection. Tuberculin-skin-testing can be negative in a quarter of cases and acid-fast bacilli may not be present. Biopsy is important but acid-fast bacilli may only be seen in 8% cases.10 Although culture is perhaps the most reliable method to detect the presence of mycobacteria, it takes a long time and the yield is often low, and has been estimated at 53% in one study.10 Polymerase chain reaction can also be used. However, the enzyme-linked immunospot assay for interferon-gamma (TB Elispot) has been shown to be very helpful in the diagnosis of cutaneous tuberculosis11 with a sensitivity of 91.6%.12 A negative test therefore makes the diagnosis of tuberculosis unlikely. Treatment is with standard anti-TB therapy13 and the diagnosis is further supported by the prompt resolution of lesions. This case highlights the variety of presentations of cutaneous tuberculosis, and even at unusual sites it should be included in the differential diagnosis of chronic lesions. Acknowledgements We would like to thank Dr Wassim Al-Salti, Consultant Histopathologist for help with the histology and photomicrographs, and Dr Angela Jones, Consultant Respiratory Physician for advice on anti-tuberculous therapy.
Declaration of conflicting interests The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article. Figure 2. Skin biopsy showing non-caseating granulomata and inflammatory infiltrate.
lesions include ulceration (as in this case), abscess, anal stricture, discharge and haemorrhoidal thrombosis with or without other systemic features.3–6 It is important to recognise this rare presentation as it requires specific anti-tuberculous treatment.7
Funding The authors received no financial support for the research, authorship, and/or publication of this article.
References 1. Mehta JB, Dutt A, Harvill L, et al. Epidemiology of extrapulmonary tuberculosis: a comparative analysis within the pre-AIDS era. Chest 1991; 99(5): 1134–1138.
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2. Alvarez Conde JL, Gutierrez Alonso VM, Del Riego Tomas J, et al. Perianal ulcers of tubercular origin: a report of 3 new cases. Rev Esp Enferm Dig 1992; 81: 46–48. 3. Tai WC, Hu TH, Lee CH, et al. Ano-perianal tuberculosis: 15 years of clinical experiences in Southern Taiwan. Colorect Dis 2010; 12: 114–120. 4. Gupta PJ. Ano-perianal tuberculosis – solving a clinical dilemma. Afr Health Sci 2005; 5: 345–347. 5. Mathew S. Anal tuberculosis: report of a case and review of the literature. Int J Surg 2008; 6: e36–39. 6. Ibn Majdoub Hassani K, Ait Laalim S, Toughrai I, et al. Perianal tuberculosis: a case report and a review of the literature. Case Reports Infect Dis 2012. DOI: 10.1155/ 2012/852763. 7. Sammain A, Jocher A, Bruckner-Tuderman L, et al. Lupus vulgaris – a case diagnosed more than 20 years after onset. J Dtsch Dermatol Ges 2006; 4: 958–960. 8. Yaghoobi R, Khazanee A, Bagherani N, et al. Gastrointestinal tuberculosis with anal and perianal involvement misdiagnosed as Crohn’s disease for 15 years. Acta Derm Venereol 2011; 91: 348–349.
9. Chokoeva AA, Tchernev G, Tana C, et al. Sarcoid-like pattern in a patient with tuberculosis. J Biol Regul Homeost Agents 2014; 28: 783–788. 10. Marcoval J and Alcaide F. Evolution of cutaneous tuberculosis over the past 30 years in a tertiary hospital on the Europena Mediterranean coast. Clin Exp Dermatol 2013; 38: 131–136. 11. Koh HY, Tay LK, Pang SM, et al. Changing the way we diagnose tuberculids with interferon gamma release assays. Australas J Dermatol 2012; 53: 73–75. 12. Lai CC, Tan CK, Lin SH, et al. Diagnostic value of an enzyme-linked immunospot assay for interferon-gamma in cutaneous tuberculosis. Diagn Microbiol Infect Dis 2011; 70: 60–64. 13. WHO guidelines for the treatment of tuberculosis, http:// www.who.int/tb/publications/2010/9789241547833/en/
Downloaded from std.sagepub.com at University of British Columbia Library on June 18, 2015
(STD)
[1–3] [PREPRINTER stage]
Int J STD AIDS OnlineFirst, published on March 13, 2015 as doi:10.1177/0956462415577728
Case report International Journal of STD & AIDS 0(0) 1–3 ! The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0956462415577728 std.sagepub.com
An unusual perineal plaque Sumeet Hindocha, Shoma Banerjee and Fiona Lewis
Abstract We present a 60 year old male with a long-standing perineal and scrotal lesion. Investigations eventually revealed cutaneous tuberculosis, with complete resolution after appropriate treatment. It highlights the variable presentation of cutaneous tuberculosis and the importance of considering the diagnosis in chronic lesions.
Keywords Tuberculosis, perineal, ano-perineal, extrapulmonary Date received: 3 January 2015; accepted: 24 February 2015
Introduction Patients with genital lesions may present late in the course of their disease, when the features may have been modified by inappropriate treatment. We present a patient with a longstanding perineal and scrotal lesion, misdiagnosed for many years as psoriasis, but eventually shown to be cutaneous tuberculosis, which responded well to treatment. It illustrates the importance of clinico-pathological correlation and highlights that rare diseases can present at unusual sites.
Case report A 61-year-old man presented with a perineal lesion, which had been present for 20 years. It had been diagnosed at the time as psoriasis and treated with potent topical steroids intermittently since. He had no systemic or local symptoms and his only concern was that the lesion was increasing in size. He had a medical history of ischaemic heart disease and there was no relevant family history. He was a heterosexual married man who had relocated from India in the 1970s, and now worked in an office. On examination, a firm indurated plaque with superficial ulceration was noted on the perineum extending to the scrotum and right buttock (Figure 1a). There were no palpable inguinal lymph nodes. The clinical differential diagnoses included ano-genital Crohn’s disease, hypertrophic lichen planus, cutaneous lymphoma or a granulomatous disorder such as sarcoidosis or tuberculosis. Laboratory investigations, including haematological parameters, renal and liver function and serum angiotensin converting enzyme were
normal and his chest X-ray was clear. A TB-elispot test was positive. A punch biopsy was taken from the plaque which showed multiple non-caseating granulomata and a dense inflammatory infiltrate (Figure 2). No acid-fast bacilli were seen on Ziehl-Neelsen staining. Fungal stains were negative. A diagnosis of cutaneous perineal tuberculosis was made and he was commenced on anti-tuberculosis therapy (rifampicin, isoniazid, pyrazinamide and ethambutol for two months followed by a further four-month course of rifampicin and isoniazid). There was a dramatic response within just three weeks of starting treatment. He completed a six-month course of therapy and the plaque completely resolved with some post-inflammatory hypopigmentation (Figure 1b).
Discussion Extrapulmonary tuberculosis accounts for 15% of all cases and can affect any organ.1 Ano-perineal tuberculosis is a very rare form of extrapulmonary disease, accounting for less than 1% of cases. It presents in many ways2 making diagnosis a challenge. It can exist alone, or be associated with gastrointestinal tuberculosis, via direct extension or haematological or lymphatic spread. Anal fistula is the most common presentation but other
Department of Dermatology, Wexham Park Hospital, Slough, UK Corresponding author: Fiona Lewis, Department of Dermatology, Wexham Park Hospital, Slough, SL2 4HL, UK. Email: [email protected]
Downloaded from std.sagepub.com at University of British Columbia Library on June 18, 2015
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(STD)
[1–3] [PREPRINTER stage]
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International Journal of STD & AIDS 0(0)
Figure 1. (a) Ulcerated perineal plaque; (b) resolution of plaque after treatment.
The appearance of cutaneous lesions in the ano-genital area can be significantly altered due to the moist environment and this renders clinical assessment alone challenging in making a final diagnosis. In this case, there were no deep fissures or other clinical signs to suggest cutaneous Crohn’s disease and he had no gastro-intestinal complaints. If bowel symptoms are present, it is important to exclude gastrointestinal tuberculosis as this can mimic Crohn’s disease.8 He was otherwise well with no other symptoms to suggest sarcoid or systemic disease. Occasionally, sarcoid-like histological patterns are seen in patients with cutaneous tuberculosis and positive immunoassays are helpful.9 A thorough history and examination is the starting point in diagnosis and helps to identify extra-cutaneous foci of infection. Tuberculin-skin-testing can be negative in a quarter of cases and acid-fast bacilli may not be present. Biopsy is important but acid-fast bacilli may only be seen in 8% cases.10 Although culture is perhaps the most reliable method to detect the presence of mycobacteria, it takes a long time and the yield is often low, and has been estimated at 53% in one study.10 Polymerase chain reaction can also be used. However, the enzyme-linked immunospot assay for interferon-gamma (TB Elispot) has been shown to be very helpful in the diagnosis of cutaneous tuberculosis11 with a sensitivity of 91.6%.12 A negative test therefore makes the diagnosis of tuberculosis unlikely. Treatment is with standard anti-TB therapy13 and the diagnosis is further supported by the prompt resolution of lesions. This case highlights the variety of presentations of cutaneous tuberculosis, and even at unusual sites it should be included in the differential diagnosis of chronic lesions. Acknowledgements We would like to thank Dr Wassim Al-Salti, Consultant Histopathologist for help with the histology and photomicrographs, and Dr Angela Jones, Consultant Respiratory Physician for advice on anti-tuberculous therapy.
Declaration of conflicting interests The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article. Figure 2. Skin biopsy showing non-caseating granulomata and inflammatory infiltrate.
lesions include ulceration (as in this case), abscess, anal stricture, discharge and haemorrhoidal thrombosis with or without other systemic features.3–6 It is important to recognise this rare presentation as it requires specific anti-tuberculous treatment.7
Funding The authors received no financial support for the research, authorship, and/or publication of this article.
References 1. Mehta JB, Dutt A, Harvill L, et al. Epidemiology of extrapulmonary tuberculosis: a comparative analysis within the pre-AIDS era. Chest 1991; 99(5): 1134–1138.
Downloaded from std.sagepub.com at University of British Columbia Library on June 18, 2015
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Hindocha et al.
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2. Alvarez Conde JL, Gutierrez Alonso VM, Del Riego Tomas J, et al. Perianal ulcers of tubercular origin: a report of 3 new cases. Rev Esp Enferm Dig 1992; 81: 46–48. 3. Tai WC, Hu TH, Lee CH, et al. Ano-perianal tuberculosis: 15 years of clinical experiences in Southern Taiwan. Colorect Dis 2010; 12: 114–120. 4. Gupta PJ. Ano-perianal tuberculosis – solving a clinical dilemma. Afr Health Sci 2005; 5: 345–347. 5. Mathew S. Anal tuberculosis: report of a case and review of the literature. Int J Surg 2008; 6: e36–39. 6. Ibn Majdoub Hassani K, Ait Laalim S, Toughrai I, et al. Perianal tuberculosis: a case report and a review of the literature. Case Reports Infect Dis 2012. DOI: 10.1155/ 2012/852763. 7. Sammain A, Jocher A, Bruckner-Tuderman L, et al. Lupus vulgaris – a case diagnosed more than 20 years after onset. J Dtsch Dermatol Ges 2006; 4: 958–960. 8. Yaghoobi R, Khazanee A, Bagherani N, et al. Gastrointestinal tuberculosis with anal and perianal involvement misdiagnosed as Crohn’s disease for 15 years. Acta Derm Venereol 2011; 91: 348–349.
9. Chokoeva AA, Tchernev G, Tana C, et al. Sarcoid-like pattern in a patient with tuberculosis. J Biol Regul Homeost Agents 2014; 28: 783–788. 10. Marcoval J and Alcaide F. Evolution of cutaneous tuberculosis over the past 30 years in a tertiary hospital on the Europena Mediterranean coast. Clin Exp Dermatol 2013; 38: 131–136. 11. Koh HY, Tay LK, Pang SM, et al. Changing the way we diagnose tuberculids with interferon gamma release assays. Australas J Dermatol 2012; 53: 73–75. 12. Lai CC, Tan CK, Lin SH, et al. Diagnostic value of an enzyme-linked immunospot assay for interferon-gamma in cutaneous tuberculosis. Diagn Microbiol Infect Dis 2011; 70: 60–64. 13. WHO guidelines for the treatment of tuberculosis, http:// www.who.int/tb/publications/2010/9789241547833/en/
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