Lymphoedema of the external genitalia is a symptom encountered in many acquired conditions include neoplastic, reactive, granulomatous and infectious disorders such as lymphogranuloma venereum (LGV). Since 2003, an ongoing LGV proctitis outbreak has been reported in industrialised countries with a new variant designated L2b. Only men who have sex with men (MSM) are affected and most are HIV co-infected; delayed or incorrect diagnoses are frequent. We report a rare clinical case of an LGV primary stage called “bubonulus” with secondary penile lymphoedema in an MSM seropositive man.

CLINICAL CASE

A 49-year-old MSM, HIV positive patient on HAART (highly active antiretroviral therapy), presented in September 2006 with a painless non-inflammatory brutal oedema of the penis with phimosis. He described three episodes of secondary syphilis in the past years (the last one treated a year ago with benzathine benzylpenicillin 2.4 MU) and gave an history of multiple sexual partners (40 per year) with “serosorting” and inconsistent condom use. The lesions appeared three days after a vigorous episode of “cock ring” use, used for a few hours with an anonymous partner.

Examination found three non-inflammatory centimetric subcutaneous nodular formations along the dorsal face of the penis with a soft, painless, circumferential oedema and several non-purulent ulcerations of the glans (fig 1). There was no scrotal oedema and no inguinal adenopathy. There were no rectal symptom and rectal examination was normal.

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Figure 1

 Appearance of penis showing subcutaneous nodular formations, oedema, and non-purulent ulcerations of the glans.

The biological assessment showed a CD4+ count at 50×10⁶/l and an undetectable HIV viral load. Haemophilus ducreyi, Treponema pallidum, and herpes simplex virus were not identified in ulcer swab using polymerase chain reaction (PCR) and immunofluorescence. Chlamydia trachomatis infection was demonstrated by PCR in the ulceration. Serovar L2b was typed by restriction fragment length polymorphism of omp1 gene amplified by polymerase chain reaction (PCR-RFLP genotyping) confirming LGV.

Syphilis serology showed a stable VDRL titre at 32 and C trachomatis serology showed high levels of IgG (more than 512) and positive IgA. A transcutaneous echography with Doppler ultrasound found three non-collected subcutaneous lymph nodes, 5–7 mm, of the dorsal face of the penis without other vascular, pelvic, or urinary tract abnormalities.

The patient was treated with oral doxycycline 200 mg/day for 3 weeks with complete disappearance of ulcerations and lymph nodes at day 15.

DISCUSSION

LGV is a systemic sexually transmitted disease caused by C trachomatis strains belonging to the L1, L2, or L3 genotype.

This lymphotropic organism leads to the development of thrombolymphangitis/perilymphangitis, and lymphadenitis, immunity of the host limiting the progression without eliminating it. Most of the damage is caused by the immune delayed cellular response, chronic oedema, and lymphatic fibrosis compromising the local vascularisation of the skin or the mucous membrane, leading to the traditional presentation: genital ulceration and buboes. The primary stage of the disease can rarely be associated with a lymphangitis of the dorsal penis, with formation of a large and tender lymphangial nodule classically called “bubonulus”.^1,2 Lymphangitis can be accompanied by local oedema, witch may produce phimosis. This is the case with our patient. Later, long standing oedema, fibrosis, and lymphogranulomatous infiltrate can result in elephantiasis with “esthiomene.”^3,4

A resurgence of LGV has been observed since 2004 in several industrialised countries, becoming an increasingly important public health issue.⁵

This outbreak concerns essentially MSM, with gastrointestinal symptoms—bloody proctitis, purulent discharge, and buboes, with a high rate of HIV seropositivity and other sexually transmitted diseases. Serovar L2b is generally found.^6,7

Our patient is part of this new outbreak population but presents with this rare “bubonulus” appearance. High rates of C trachomatis Ig G levels suggest an old infection. Can modified lymphatic drainage of the infected site after use of the cock ring (with or without HIV modified immune response) be an explanation for these acute localised buboes?

For instance, it is still unclear why rectal symptoms are found more frequently in the new LGV MSM cases. Is the appearance of LGV a new outbreak or has it been always present undiagnosed? Some studies suggest that LGV has always been present but was unreported,^8,9 and the paucity of penile lesions is consistent with failing to recognise cases in insertive partners.

Ward et al recently described 19% prevalence of sex toy use and 32% of fisting in the LGV population but with missing data⁷ and some authors showed the possibility of a high rate of asymptomatic ⁶ and urethral cases.¹⁰ Our patient provides an additional argument to suggest that host factors (HIV infection, behavioural factors such as sex toys) can influence clinical presentation, mimicking a new rectal outbreak in a slow epidemic.