Two Lymphogranuloma Venereum Cases in a Heterosexual Couple in Bilbao (Spain)

LYMPHOGRANULOMA VENEREUM (LGV) WAS AN unusual disease in industrialized countries until 4 years ago; before this when it did appear, it had generally been acquired in some endemic area (Africa, Asia, South America, or the Caribbean Islands). In December 2003, an outbreak of LGV was detected in Rotterdam, Netherlands.1 Since then, more than 1000 cases have been reported in the Netherlands, France, the United Kingdom, Germany, Belgium, Switzerland, Sweden, Portugal, Denmark, and Spain.2–10 In the United States and Canada, the first cases were recorded in 2004 and between then and November 2006, 85 cases were reported in Canada.1,11 All these cases had remarkably uniform characteristics: men who have sex with men (MSM), a high rate of HIV seropositivity and frequent concurrent infection with other STIs. Most of them had rectal symptoms and only a few inguinal lymphadenopathy or classic genital ulcers.12 All confirmed cases were of the genotype L2. There was no evidence that this infection had extended beyond these nuclear groups, until 3 LGV cases were reported among Portuguese women during 2007.9 The aim of this article is to describe the first LGV cases diagnosed in a heterosexual couple in Bilbao (Spain). We directly documented sexual transmission by identifying genital Chlamydia trachomatis type L2 infection in both patients. The results also suggest that primary classic LGV can take the form of urethritis and cervicitis, without genital ulceration. A further 2 cases of LGV had previously been recorded in Spain, but both of them were in MSM.10 In June 2006, a 33-year-old Spanish man came to see us complaining of 2 months of gradually progressive and painful right inguinal lymphadenopathy (swelling). The onset of swelling was accompanied by radiating low back pain, myalgia in the lower extremities, and dysuria. He denied any history of previous genital ulceration, inguinal buboes, fever, malaise, proctitis, or urethral discharge. His general practitioner had treated him with an antibiotic and antiinflammatory drug whose name he did not know. Dysuria remitted with treatment but the swelling did not. The patient reported multiple unprotected heterosexual contacts during the previous year but he had not traveled abroad. He also denied having had sexual intercourse with men or immigrants. Physical examination revealed bilateral inguinal lymph nodes of firm consistency, those on the right being bigger. Urethral, rectal, and pharyngeal samples were taken for microbiologic diagnosis. Samples for direct fluorescent antibody and culture for C. trachomatis were negative. So were culture for Neisseria gonorrhoeae and herpes simplex virus. There was no serological evidence of syphilis or HIV infection. The laboratory extracted DNA from all samples using the Amplicor CT/NG extraction kit and tested them for C. trachomatis using an in-house TaqMan real-time PCR assay, which targeted the cryptic plasmid. C. trachomatis PCR was positive in the urethral sample and negative in other sites. Ultrasound-guided fine needle aspiration of the right lymph node was performed during a second visit in July 2006. The material obtained was processed in the same way as described above and C. trachomatis PCR was positive. Urethral and lymph node DNA samples were tested for LGV using a TaqMan based real-time PCR that used the polymorphic membrane protein H gene as a PCR target.13 The results of this assay were positive. Both samples were identified as LGV serovar 2 and not L2b genovar, by sequencing an ompA segment spanning variable segments 1 and 2 using an ABI Genetic Analyzer 3130 and Big Dye Terminator kit version 3.1 according to the manufacturer’s protocol. The man’s partner, a 29 year-old Spanish woman, described a history of painful bilateral inguinal swellings. She was 8-weeks pregnant and denied any sexual contact other than with her partner over the previous 2 years. Examination found painless bilateral inguinal nodes of firm consistency. Vaginal, endocervical, and pharyngeal samples were collected for routine STI microbiologic testing. Direct fluorescent antibody for C. trachomatis and samples cultured for N. gonorrhoeae and herpes simplex virus were negative. All serological tests for syphilis, hepatitis B, hepatitis C, and HIV infection were negative. The laboratory extracted DNA from all samples and processed them in the same way described for her partner’s samples. C. trachomatis PCR and culture was positive for the endocervical sample and negative for other sites. The C.trachomatis strain was determined as L2 genotype and was confirmed using the same method as for her partner’s samples. The man was treated with doxycycline 100 mg twice daily for 21 days and the woman with erythromycin 500 mg 4 times daily for the same period.14 Both partners improved clinically after treatment. Repeated PCR samples that were collected in a follow-up visit 4 weeks after treatment were negative in both individuals. This report on LGV in both members of a heterosexual couple is interesting for several reasons. First, we have not found any documented cases of LGV in heterosexual couples during the past 2 decades. Second, although primary rectal infection in MSM is the dominant clinical presentation of LGV in Europe, our results show that classic genital-inguinal LGV continues to present from time to time. It is also important to notice that C. trachomatis L2 and not the L2b genovar was detected in both partners. The C. trachomatis L2b genovar is far more common in MSM and it is the predominant genovar isolated in the outbreaks described in Europe.1,2,5,6,15–17 Third, C. trachomatis L2 serovar was documented in the genital tracts of both partners and the male’s lymph node specimen but no genital ulceration was found. LGV is historically described as having a genital ulcer component, but the genital ulcer is often not documented.18,19 It may be possible that the urethra and cervix are common, even the usual, primary infection sites of LGV in heterosexual men and women, just as the primary infection in MSM is primarily rectal. These 2 reported cases are not enough to answer this question, but they contribute to emphasize the uncertainties about the primary infection site in LGV cases. Clinicians should be alert to detecting new cases of LGV regardless of clinical presentation or sexual orientation of the patient. LGV cases may be missed if this diagnosis is not considered or if appropriate diagnostic tools are not available and patients may remain undiagnosed and develop severe complications apart from continued dissemination of C. trachomatis infection. With modern DNA amplification technology, it may be less necessary to rely on serological diagnosis. Serology has always been imprecise, and it is likely that nowadays, more laboratories have good DNA amplification experience than C. trachomatis serology experience. It therefore seems that we should focus on the problems LGV presents and provide new tools to improve diagnosis and epidemiologic surveillance of LGV infections.

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