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Reactive arthritis associated with L2b lymphogranuloma venereum proctitis
  1. K El Karoui1,2,
  2. F Méchaï1,2,
  3. F Ribadeau-Dumas1,2,
  4. J-P Viard1,2,
  5. M Lecuit1,2,
  6. B de Barbeyrac3,
  7. O Lortholary1,2
  1. 1
    Centre d’Infectiologie Necker Pasteur, Paris, France
  2. 2
    Université Paris Descartes, Service des Maladies Infectieuses et Tropicales, Hôpital Necker Enfants-Malades, Paris, France
  3. 3
    Centre National de Référence des Infections à Chlamydiae, Laboratoire de Bactériologie, Université Victor Segalen, Bordeaux, France
  1. Professor O Lortholary, Service des Maladies Infectieuses et Tropicales, Hôpital Necker Enfants-Malades, 149 rue de Sèvres, 75015 Paris, France; olivier.lortholary{at}nck.aphp.fr

Abstract

An ongoing outbreak of lymphogranuloma venereum (LGV) L2b proctitis, predominantly in HIV-positive men who have sex with men (MSM), has been reported in industrialised countries. A case of reactive arthritis after L2b proctitis is described. This case expands the spectrum of severe complications related to LGV L2b proctitis. Since this infection may be asymptomatic, this organism should be screened for in HIV-positive MSM with symptoms consistent with reactive arthritis.

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Chlamydia trachomatis is the aetiological agent of major human infections including trachoma (classically serovars A–C), urogenital infections (serovars D–K) and lymphogranuloma venereum (LGV) (serovars L1–L3).

Since 2003, an ongoing LGV proctitis outbreak has been reported in industrialised countries, including France (mainly in Paris).1 To date, most LGV isolates of the recent outbreak have been identified as L2b.2 We report a case of sexually acquired reactive arthritis (SARA) associated with C trachomatis serovar L2b proctitis in an HIV-infected man.

CASE REPORT

A 41-year-old white homosexual man was admitted to our infectious diseases department for oligoarthritis. HIV-1 infection had been diagnosed 4 years earlier, when he presented with cutaneous secondary syphilis. Antiretroviral therapy had not yet been commenced as he had remained well.

One month after an episode of unprotected receptive anal intercourse, he presented with weight loss, fever, purulent rectal discharge and tenesmus. Proctitis was clinically diagnosed and he was successfully treated with oral doxycycline (100 mg twice/day). PCR for C trachomatis was positive on the blind anal sample taken. The LGV variant L2b was detected by genotyping (PCR restriction fragment-length polymorphism) of the omp1 gene encoding the major outer membrane protein and sequencing the variable domain 2 of the omp1 gene.3 Seven days after the diagnosis of proctitis, he presented with bilateral serous conjunctivitis. A few days later, the patient was hospitalised for an investigation of oligoarthritis of the right wrist, right knee and both ankles. Physical examination revealed pain and swelling of the right wrist, right knee, both ankles and left metatarsophalangeal joints, with sausaging of the second and third left toes. Laboratory tests were as follows: CD4 lymphocyte count 252 cells/μl (21%), HIV-1 viral load 6200 genome copies/ml (log 3.8), negative serological tests for yersinia, salmonella and campylobacter, negative stool culture. HLA-B27 testing was positive, whereas a search for rheumatoid factor, antinuclear antibodies and anticyclic citrullinated peptide antibodies was negative. Analysis of right knee synovial fluid revealed a raised white blood cell count of 4900 cells/ml (90% neutrophils) and negative bacterial culture. 16S ribosomal RNA gene PCR amplification and C trachomatis-specific PCR on synovial fluid were both negative. Cultures for N gonorrhoeae, and mycoplasma species from a urethral swab were negative, as well as C trachomatis-specific PCR on a first void urine sample. x Ray examinations of all involved joints revealed no abnormality.

A diagnosis of SARA complicating C trachomatis L2b proctitis was proposed. Oral doxycycline was continued for a total of 30 days (200 mg/day). Oral ketoprofen (150 mg/day) allowed complete clinical recovery after 2 months and no relapse occurred after one year of follow-up.

Key messages

  • LGV L2b, as well as other C trachomatis serovars, may trigger SARA in HLAB27-positive HIV-infected men who have sex with men (MSM).

  • Given the recent outbreak, LGV L2b infection should be systematically screened for in MSM with reactive arthritis, even though no proctitis symptoms are present.

  • This case adds reactive arthritis to the severe complications of LGV L2b proctitis and underscores the importance of rapidly diagnosing and treating this infection.

DISCUSSION

Our patient presented with SARA, 2 weeks after C trachomatis serovar L2b infectious proctitis. Of note, conjunctivitis was bilateral and resolved spontaneously, which argues against a direct conjunctive C trachomatis infection. Other classic features of SARA, such as balanitis or sacro-ileitis, were absent. However, our patient fulfilled the currently proposed criteria for reactive arthritis.4

The association of urogenital C trachomatis (serovars D–K) with SARA is well described.5 6 This association, and the strong temporal relationship between SARA and proctitis in our patient, argues for SARA related to C trachomatis L2b proctitis. Cases of reactive arthritis-associated proctitis and C trachomatis infection have already been published, but direct evidence of C trachomatis infection was not available, co-infection with other reactive arthritis-associated microorganisms was not ruled out and the serovar was not determined.7 8

Differential diagnoses, including infection with other reactive arthritis-triggering microorganisms and gonococcal arthritis, were ruled out with serological tests, cultures of urethral swab and 16S rRNA gene PCR on synovial fluid. Reactive arthritis has been reported to occur in up to 10% of patients with HIV infection. However, there is no direct correlation between reactive arthritis and HIV infection per se, and this association is thought to be related to the co-infections occurring in HIV-infected patients, as was shown in our case.9

In the recent outbreak, LGV predominantly caused proctitis or proctocolitis, primarily through unprotected receptive or insertive anal intercourse,1 a substantial proportion of patients even being asymptomatic.10 Diagnosis is essential as prolonged treatment (3 weeks) with doxycycline or macrolides is required in LGV (C trachomatis serovar L1–3) proctitis, in contrast to infection with other serovars for which short-term treatment is possible.10

Given the recent outbreak, C trachomatis L2b proctitis-associated SARA appears to be rare, and is less frequent than SARA related to other C trachomatis serovars (incidence estimated at approximately 1–3% in C trachomatis serovars D–K).4 A strong local immune response in proctitis could prevent the dissemination of infection and subsequent reactive arthritis.4

In conclusion, SARA may be triggered by the epidemic LGV L2b isolate responsible for proctitis in HIV-infected MSM.

Acknowledgments

The authors would like to thank Maithe Clerc for the genotyping analysis and Nicolas Dupin for his careful review of the manuscript.

REFERENCES

Footnotes

  • See Editorial, p 157

  • Competing interests: None.

  • Patient consent: Obtained.

  • Contributors: KEK, FM and OL were in charge of the patient and wrote the manuscript. FRD, JPV and ML were in charge of the patient and carefully revised the manuscript. BdB performed the strain serovar identification and carefully revised the manuscript.

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