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Lymphogranuloma venereum presenting as perianal ulceration: an emerging clinical presentation?
  1. Tajinder Singhrao,
  2. Elizabeth Higham,
  3. Patrick French
  1. The Mortimer Market Centre, Camden Primary Care Provider Services, London, UK
  1. Correspondence to Dr Patrick French, The Mortimer Market Centre, Camden Primary Care Provider Services, off Tottenham Court Road, London WC1E 6JB, UK; patrick.french{at}camdenpct.nhs.uk

Abstract

An outbreak of lymphogranuloma venereum (LGV) infection has been recognised in the UK since 2004, predominantly affecting HIV-positive men who have sex with men (MSM). Patients typically present with proctitis symptoms. The prevalence of rectal LGV in MSM attending sexually transmitted infection clinics in London is estimated at 1%. Health Protection Agency surveillance has shown a decrease in anorectal manifestations despite little demographic change. Two cases of HIV-infected patients presenting with isolated perianal ulcer disease are reported here. Both cases were confirmed to have rectal Chlamydia trachomatis-specific DNA of an LGV associated serovar. As presentations of LGV diversify, further education and surveillance are needed in order to reduce transmission and prevent long-term complications. A strong argument already exists for the incorporation of chlamydia nucleic acid amplification tests in the management of MSM with proctitis; this paper provides evidence that this should be extended to MSM with perianal ulcer disease.

  • Chlamydia trachomatis
  • gay men
  • genital ulcers
  • homosexuality
  • lymphogranuloma venereum
  • male
  • ulcer

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Lymphogranuloma venereum (LGV), first identified in the UK in 2004, has become endemic among men who have sex with men (MSM). The prevalence of LGV among MSM (symptomatic and asymptomatic) attending London genitourinary clinics in 2009 was estimated at 0.9% rectal and 0.04% urethral.1 Health Protection Agency (HPA) surveillance from 2003 to 2008 found 849 cases of LGV, the majority in London (72%). All cases were male, predominantly white MSM (99%), co-infected with HIV (75%).2 Risk factors for acquisition include co-infection with a sexually transmitted infection, unprotected receptive anal intercourse, fisting, group and anonymous sex.

Historically, LGV infection was an inguinal syndrome,3 including a transient primary ulcerative lesion with thrombolymphangitis and perilymphangitis resulting in tender inguinal lymphadenopathy. However, an anorectal LGV syndrome among MSM is well recognised,4 and is the commonest presentation of LGV in the UK,5 with genital ulcers and inguinal symptoms rarely encountered.6 HPA data since 2004 have consistently shown over 75% of patients identified with LGV infection presented with proctitis.7

Case report

A 28-year-old white British man with HIV presented with a 2-week history of a single painful ulcer at the anal margin without rectal or inguinal symptoms and no proctitis on proctoscopy. This patient had been diagnosed with LGV and rectal gonorrhoea 4 months previously, presenting with proctitis, inguinal lymphadenopathy and an indurated perianal ulcer. LGV test of cure was negative 2 months following previous treatment.

A 45-year-old white British man with HIV presented with a 3-day history of a single painful 0.5 cm indurated perianal ulcer associated with tender unilateral inguinal lymphadenopathy without proctitis on proctoscopy. He subsequently developed fevers.

Chlamydia trachomatis-specific DNA of an LGV associated serovar was identified on rectal (not ulcer) swabs taken from both patients. PCR tests for herpes simplex virus (HSV) types 1 and 2/Treponema pallidum taken from the ulcers in both patients were negative. At presentation patient 1 was rapid plasma reagin negative (having been treated for primary syphilis 1 year previously) and patient 2 had negative T pallidum serology. Urethral chlamydia nucleic acid amplification tests (NAAT) were negative in both patients. Patient 1 was diagnosed with hepatitis C at presentation.

Both patients reported unprotected receptive and insertive anal intercourse in the past 3 months. At presentation patient 1 was treated for an anal fissure and patient 2 for presumed HSV infection. Both patients received a 3-week course of doxycycline once they were found to be chlamydia positive. Their symptoms resolved completely on treatment and both had a negative test of cure at 6 weeks.

Discussion

From 2004 to March 2010 we have seen 146 presentations of LGV at our centre, accounting for 17% of cases reported to the HPA (unpublished data). While largely among the HIV-positive population, 22/146 (15%) presentations affected HIV-negative patients. Only 11/146 (7.5%) did not have proctitis symptoms, of these two were asymptomatic, two had isolated ulcer disease (the patients we report here) and seven had genital symptoms, four with inguinal syndrome, some with systemic symptoms. We have seen two other patients with LGV and perianal ulcers who had concurrent proctitis. Both had negative PCR tests for HSV from the ulcer and T pallidum serological testing was negative (T pallidum PCR testing was not undertaken). Both had ulcer resolution with doxycycline. A case–control study showed an association between anorectal ulceration and LGV but it is not clear whether these patients had perianal, anal canal disease or concomitant proctitis.8 We are not aware of any other published reports of patients with isolated perianal ulceration from LGV infection in which other causes of ulceration were excluded.

LGV-associated genital ulcer disease is rare in the UK, explaining why our patients were not initially treated for LGV infection. A cohort of patients presenting to London genitourinary clinics with inguinal syndrome and ulceration described four patients with genital ulceration, one with multiple genital and perianal ulcers.9 Primary LGV erosions typically affect the genitalia and are classically painless.6

Neither of our patients had genital ulceration or proctitis. Both had positive rectal LGV chlamydia NAAT (HSV and syphilis were excluded on PCR testing). The validity of ulcer chlamydia NAAT is uncertain and this paper suggests it may be a useful test. It raises questions regarding the biology and transmission of LGV, including possibilities of subclinical proctitis and ulceration.

Health promotion and behavioural intervention are paramount in preventing and identifying cases of LGV infection and addressing sexual health needs of at-risk groups.5 10 The HPA recommend LGV testing for all symptomatic HIV-positive MSM.2 There is a strong argument for rectal chlamydia NAAT to be used in MSM with proctitis, and this report provides evidence that rectal LGV testing should be extended to patients with anogenital ulcers.

Key messages

  • These cases, to our knowledge, are the first described LGV infections presenting with isolated perianal ulcer disease with normal proctoscopy and other causes excluded.

  • LGV typically presents with proctitis symptoms among HIV-positive MSM within the UK.

  • We recommend testing for LGV infection in HIV-positive MSM presenting with isolated perianal ulcer disease using chlamydia NAAT.

  • A positive rectal LGV chlamydia NAAT with isolated perianal ulcer disease may provide insight into the biology and transmission of LGV, including subclinical proctitis and unrecognised ulceration.

Acknowledgments

The authors would like to thank colleagues at the University College London Hospital Medical Microbiology Laboratory and the Sexually Transmitted Bacteria Reference Laboratory at the HPA.

References

Footnotes

  • Competing interests None.

  • Patient consent Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.

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