Author(s) : Ricardo Coelho, Candida Fernandes, Joao Machado, Fernanda Correia, Teresa Martins, Fernando Maltez, Jorge Cardoso , Faro Hospital Dermatology Department, Hospital Central de Faro, Rua Leão, Penedo 8000 Faro, Portugal, Dermatovenereology Department, Lisbon, Portugal, Infectious Disease Department, Lisbon, Portugal. |
ARTICLE
Auteur(s) : Ricardo Coelho1,
Candida Fernandes2, Joao Machado3, Fernanda
Correia3, Teresa Martins3, Fernando
Maltez3, Jorge Cardoso2
1Faro Hospital Dermatology Department, Hospital
Central de Faro, Rua Leão, Penedo 8000 Faro, Portugal
2Dermatovenereology Department, Lisbon, Portugal
3Infectious Disease Department, Lisbon, Portugal
In June 2007, a 54-year-old HIV1 positive man from Lisbon
presented with a 3-month history of rectal pain, mucopurulent
rectal discharge, rectal bleeding, tenesmus, weight loss (8 kg) and
generalized weakness. He was on antiretroviral therapy
(Combivir® + Telzir® + Norvir®)
with a CD4 count of 1158 cell/mm3 and a viral load of
< 50 copies/mL. He had several stool cultures that showed no
enteric pathogens. A sigmoidoscopy and later, a colonoscopy
(figure 1),
showed multiple rectal ulcers; histological examination revealed
ulceration with mixed acute and chronic inflammatory cells and a
lymphoid infiltrate. He was treated with metronidazol (several
courses) but his symptoms failed to resolve. He was then referred
to our dermatovenereology department for screening of enteric
sexually transmitted diseases.
The patient had had at least 4 anonymous sex partners in the
previous 6 months and regular anal-receptive intercourse without
condoms. VDRL, TPHA, VHB and VHC serology were performed. Serology
for Chlamydia trachomatis showed a titer of 1:1024 (IgG), IgM <
16 and IgA 1:32. VDRL was reactive (1/32) and TPHA was positive. We
performed a rectal swab that was tested for Neisseria gonorrhoeae,
Chlamydia trachomatis, herpes simplex virus 1 and 2 and Treponema
pallidum, through commercial nucleic acid amplification tests
(Cobas-Amplicor and Sacace), performed according to manufacturers’
instructions. Chlamydia trachomatis and Treponema pallidum were
both detected and further molecular biological studies of the
Chlamydia trachomatis isolate (ompA genotyping) confirmed the
infection with a new lymphogranuloma venereum (LGV) variant
(different from all the described to date) associated with recent
syphilis. In the C. trachomatis ompA gene, our strain presents 3
nucleotide changes to L2-434 (which is the L2
prototype strain), 1 nucleotide change to L2b-144276 and
2 nucleotide changes to the recently described L2c.
Treatment with 100 mg oral doxycycline twice daily for 3 weeks and
a single dose of benzatinic penicillin (2.400.000 U) was
effective. Test of cure was performed 5 weeks after treatment and
was negative. LGV is caused by the L1, L2 and L3 serovars of
Chlamydia trachomatis and was previously confined to endemic areas
in tropical regions. However, since 2003, a cluster of LGV cases
presenting with anorectal symptoms in MSM has been reported in
Rotterdam. This was followed by a series of case reports in various
cities in Western Europe and the United States. Most of these
patients presented with rectal symptoms and only a few with
inguinal lesions. The majority of patients were co-infected with
HIV, and high levels of co-infection with sexually transmitted
bacterial infections and hepatitis C were also reported [1-3]. The
majority reported large numbers of sexual partners and unprotected
anal intercourse. Therefore, as in our patient, contact tracing has
been of limited use so far. This is the first rectal LGV case
reported in Portugal; Chlamydia trachomatis (L2 serovar) has
previously been isolated from urogenital samples in another 5
patients.
As in the case of our patient, a diagnostic delay has been
common as many doctors are unfamiliar with this condition.
Endoscopic features are non-specific, with a wide range of
differential diagnoses including Crohn’s disease, anorectal
carcinoma and other non-sexually and sexually transmitted
infections. Most guidelines [4, 5] have suggested that first-line
treatment is doxycycline 100 mg twice daily for 21 days
(erythromycin 500 mg, 4 times daily, 21 days is an alternative). We
treated our patient first with penicillin and introduced
doxycycline when we received nucleic acid amplification tests.
A diagnostic delay is common in this condition and LGV cases may
easily be missed if LGV is not considered during diagnosis or if
appropriate diagnostic tools are not available. This condition is
an important sentinel event with serious implications for the
patient and for public health. For these reasons, it is important
to consider this diagnosis when seeing MSM with bowel symptoms.
Acknowledgements
We thank the Chlamydia-Neisseria unit of the National Institute of
Health, Lisbon, Portugal for the molecular diagnosis support and
molecular characterization strains. Financial support: none.
Conflict of interest: none.
References
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et al. Resurgence of lymphogranuloma venereum in Western
Europe: an outbreak of Chlamydia trachomatis serovar L2 proctitis
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2004; 39: 996-1003.
2 Van der Bij AK, Spaargaren J, Morre SA,
et al. Diagnostic and clinical implications of anorectal
lymphogranuloma venereum in men who have sex with men: a
retrospective case-control study. Clin Infect Dis 2006; 42:
186-94.
3 Ranki A. HIV infection. Eur J Dermatol 2008; 18:
217-9.
4 CDC guidelines for LGV (www.cdc.gov/STD/treatment/).
5 McMillan A, van Voorst Vader PC, de Vries HJ.
The 2007 European guideline (IUSTI/WHO) on the management of
proctitis, proctocolitis and enteritis caused by sexually
transmissible pathogens. Int J STD AIDS 2007; 18: 514-20.
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