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Home , Anal dysplasia, Lymphogranuloma venereum, Proctocolitis

PRACTICE | CASES CPD

Proctocolitis caused by ­

Edward Weiss MD, Marie Sano BScN RN n Cite as: CMAJ 2018 March 19;190:E331-3. doi: 10.1503/cmaj.171292

47-year-old man was referred to our clinic for the evaluation of ongoing anorectal discomfort and an KEY POINTS anal mass. The patient had experienced severe rectal pain • Clinicians should suspect lymphogranuloma venereum andA cramping and incomplete defecation, and had passed stools when a patient presents with typical symptoms (rectal pain, containing pus and blood. He also had intermittent and dif- abdominal cramping, discharge and bleeding ) and risk factors ficulty starting and maintaining urine flow. These symptoms had for anal transmission, such as HIV infection, or a caused him considerable distress and disruption to his daily life history of anal intercourse or same-sex male intercourse. over the previous four months. • Nucleic acid testing is more sensitive than chlamydial culture The patient’s medical history included HIV infection, for which for detecting infection, but may not be available in all settings. he was receiving stable antiretroviral therapy. He also had a history • Urine nucleic acid amplification tests alone are not sufficient, because a substantial proportion of chlamydial infections in of coronary artery disease, with two previous myocardial infarc- men who have sex with men will be extragenital. tions requiring percutaneous intervention. The rest of his medical • Empiric treatment with should be considered if history was noncontributory. The patient was in a stable but non- the index of suspicion for lymphogranuloma venereum monogamous relationship with another man and practised anal proctitis is high. receptive intercourse with inconsistent condom use. The patient’s symptoms were initially attributed to external . When his condition did not improve with usual hem- anoscope, we saw florid proctitis with purulent discharge and fria- orrhoid treatment, he presented to the emergency department, ble anal and rectal mucosa (Figure 2). We took swabs from the most where he underwent computer tomography (CT) imaging of the purulent areas for chlamydial and gonorrheal culture. We opted to abdomen and pelvis (Figure 1) that showed moderate mucosal defer a high-resolution owing to the patient’s acutely involving the sigmoid colon. The interpreting radiologist abnormal mucosa and his discomfort. suggested that “it could represent an inflammatory process … or Based on the suggestive history and examination, we consid- infectious .” The patient was referred to an outpatient endos- ered a diagnosis of lymphogranuloma venereum to be the most copy clinic for further investigation where he underwent a colonos- likely cause of the patient’s symptoms, which was treated empir­ copy. The endoscopist noted the use of copious irrigation to ensure ically with a 21-day course of doxycycline (100 mg taken orally, visibility. The only finding of note was an “anal mass,” which was twice daily). Cultures for Chlamydia species showed growth, and biopsied. Histology from the lesion was reported as “acute inflam- molecular testing confirmed the presence of Chlamydia - matory ,” as well as “a small fragment of colonic mucosa tis serovar L2b, a known cause of lymphogranuloma venereum. showing low-grade dysplastic change.” The patient reported complete resolution of his symptoms after Before being seen at our clinic, the patient underwent urine completing the course of doxycycline. He has regular follow-up­ with nucleic acid testing for and chlamydia, the results of high resolution anoscopy for unrelated findings of anal dysplasia. which were negative. Screening for and C was also negative. A culture of the patient’s stool showed no bacterial infec- Discussion tion; Blastocystis hominis grew on culture, but the patient was not given treatment for it. Other laboratory investigations included an Lymphogranuloma venereum is a sexually transmitted infection undetectable HIV RNA target and a CD4 count of 1129 cells/mm3, associated with invasive serovars L1, L2 and L3 of C. trachomatis.1 with a CD4 percentage of 27.1 and a CD4:CD8 ratio of 0.7. The infection has historically been associated with painful inguinal On physical examination, the patient had diffuse abdominal (buboes) as its pathognomonic sign; however, in tenderness and no obvious inguinal lymphadenopathy. His peri- recent years, increasing attention has been given to rectal presenta- anal skin was normal. A digital rectal examination caused extreme tions of lymphogranuloma venereum.2 Inoculation of chlamydial pain and did not suggest any obvious mass. Upon insertion of an pathogens in the rectal mucosa, rather than the penile or vulvar

© 2018 Joule Inc. or its licensors CMAJ | MARCH 19, 2018 | VOLUME 190 | ISSUE 11 E331 PRACTICE tion among populations of men who have sex with men in northern tion amongpopulationsofmenwhohavesexwithinnorthern tries. However, starting in the early 2000s, case reports of the infec- tion not commonly encountered or transmitted in developed coun- E332 arrow) andanenlarged obturatorlymphnode(B,arrow). Figure 2: Contrast-enhanced computed tomography image of the patient’s abdomen and pelvis showing thickened mucosa of the sigmoid colon (A, months later(B)ina47-year-oldmanreferredtoananaldysplasiaclinicfortheevaluationofongoinganorectaldiscomfortandmass. Figure 1:Anoscopicimageoftheanorectaljunctionshowingdiffusepurulentproctitisbeforetreatment(A)andrestorationnormalana tomy six patient’s sexualpracticesorHIVstatus. response totreatmentorfurtherinquiriesaremadeintothe disease, andtheinfectionmaynotbesuspectedunlessthereisno The clinicalappearancecanoftenmimicthatofinflammatorybowel without thedevelopmentofclinicallyapparentlymphadenopathy. ­tissue, can result in a severe hemorrhagic proctitis or Lymphogranuloma venereum has been portrayed as an infec CMAJ | MARCH 19,2018 - | VOLUME 190 and westernEuropegrewinfrequency. ada; tinued, 170 cases were reported to the Public Health Agency of Can- national surveillanceforlymphogranulomavenereumwasdiscon- venereum in North America. Between 2004 and 2012, before shown asubstantialincreaseintheincidenceoflymphogranuloma generally divided into three stages. Patients may initially have a generally dividedintothreestages.Patientsmayinitiallyhavea men, and48%involvedHIVcoinfection. months oftheyear. The natural history of lymphogranuloma venereum infection is The naturalhistoryoflymphogranulomavenereuminfectionis 3 in 2016, 39 cases were reported in Toronto alone in the first six in2016,39caseswerereportedTorontoalonethefirstsix | ISSUE 11 4 Of these latter cases, all of the patients were Oftheselattercases,allofthepatientswere 1 Recentstatisticshave PRACTICE ​ ​ ​ ​ www. E333 20. 9 accessed 74. 41-02: 67- - Sexual part 41: 1 Available: 2015; 2005; 2014. Recommendations for the Recommendations for the Clin Infect Dis www.toronto.ca/community-people/ Can Commun Dis Rep ; empiric treatment for gonorrhea; empiric treatment for Chlamydia and lymphogranuloma venereum 1 Prevalence of rectal, urethral, and pharyngealrectal, urethral, Prevalence of Available: et al. , Lymphogranuloma venereum: an emerging cause Lymphogranuloma venereum: et al. et E , D. W The authors thank Acknowledgements: The authors thank Dr. Irving Salit for his guidance and support, assistance his for Magnuson William Dr. and with the computed tomography images. Correspondence to: Edward Weiss, [email protected] Payne Lymphogranuloma venereum: diagnostic and treatment treatment and diagnostic venereum: Lymphogranuloma , . Wong R www.phac-aspc.gc.ca/publicat/lgv/lgv-rdt1-eng.php ( SJ JK, Goldmeier ISSUE 11 ISSUE D, | Gulin MacLean Chaw , , Available: Lymphogranuloma venereum in the differential diagnosis of proctitis. Lymphogranuloma venereum in the differential diagnosis R S

CK, E. Testing for common coinfections, including HIV, gonorrhea, Testing for common 1 resurgence/ (accessed 2017 Apr. 24). Supplementary statement concerning the laboratory diagnosis of lympho- venereum (LGV): Canadian guidelines on sexually transmitted infections. Ottawa: Public Health Agency of Canada; chlamydia and gonorrhea detected in 2 clinical settings among men who havechlamydia and gonorrhea detected in 2 clinical settings sex with men: San Francisco, California, 2003. canada.ca/content/dam/phac-aspc/migration/phac-aspc/std-mts/sti-its/cgsti -ldcits/assets/pdf/-supp-lgv-eng.pdf (accessed 2017 Nov. 16). Kent 2007;18:11-4, quiz 15. of proctitis in men who have sex with men. Int J STD AIDS Centers for Disease Control and Prevention. and Neisseria gonor- ­laboratory-based detection of rhoeae — 2014. MMWR Recomm Rep 2014;63:1-19. Ceovic challenges. Infect Drug Resist 2015;8:39-47. -info-for-health-professionals/syphilis-and-lymphogranuloma-venereum - Richardson health-wellness-care/information-for-healthcare-professionals/sexual-health Syphilis and lymphogranuloma venereum resurgence. Toronto: City of Toronto: City of Syphilis and lymphogranuloma venereum resurgence. Toronto; updated 2017 Aug. 3. in Canada: 2003–2012 — summary report. 2017 Apr. 24). Weir CMAJ 2005;172:185. Totten Interim statement on the diagnosis, treatment and reporting of lymphogranulomaInterim statement on the diagnosis, treatment and reporting of Canada; modified 2005venereum (LGV) in Canada. Ottawa: Public Health Agency June 6. Treatment is traditionally 100 mg doxycycline twice daily for 21 doxycycline twice daily is traditionally 100 mg Treatment . . . 9. 6. 7. 8 5. 4. species and difficulties in interpreting variations in titres — specif­ in titres variations interpreting in and difficulties species venereum. do not rule out lymphogranuloma ically, low titres 2. 3 with ceftriaxone (250 mg intramuscularly) should be considered at mg intramuscularly) should be considered with ceftriaxone (250 if exudate is present but other symptomsthe time of presentation venereum. are not typical for lymphogranuloma days. is an alternative, and some authorities suggest authorities some and alternative, an is Erythromycin days. may be effective. for three weeks weekly azithromycin ners should undergo treatment as per usual recommendations, with per usual recommendations, undergo treatment as ners should - doxycycline, even in the absence of symp either azithromycin or toms. References 1 Conclusion entity clinical is a serious proctitis venereum Lymphogranuloma incidence. Complications and prolonged that is increasing in and early collec- symptoms can be avoided through the prudent reviewing a thorough tion of targeted rectal specimens, and by with typical symp- sexual health history of patients presenting is necessary, and toms. Maintaining a high index of suspicion waiting for the results empiric treatment may be warranted while of investigations to avoid unnecessary morbidity. syphilis and , is recommended, particularly for patientssyphilis and hepatitis known whose HIV status is not VOLUME 190 VOLUME |

Edward Weiss participated in in participated Weiss Edward 1 MARCH 19, 2018 | direct clinical care of the patient and drafted the article. Marie Sano provided direct clinical care of the patient and helped revise the arti- cle. Both authors approved the final version of the article to be published and agreed to be responsible for its integrity. Contributors: 8 Delays in treatment Delays in treatment 5 CMAJ However, nucleic acid amplifi- 6 - Depart Clinic, Immunodeficiency Although none of the commercial test kits available in Although none of the commercial test kits 7 If lymphogranuloma venereum is suspected based on risk fac- Untreated lymphogranuloma venereum proctitis can lead to venereum proctitis can lead to Untreated lymphogranuloma The diagnosis of lymphogranuloma venereum proctitis has pre- The diagnosis of lymphogranuloma venereum Competing interests: None declared. This article has been peer reviewed. The authors have obtained patient consent. Affiliation: ment of Medicine, University Health Network, Toronto, Ont. tors and clinical presentation, specimens that test positive for tors and clinical presentation, specimens that test positive for Chlamydia should be sent to the National Microbiology Laboratory (Winnipeg, Manitoba) for confirmatory serotyping by DNA sequencing. Serologic testing for lymphogranuloma venereum is not recommended owing to cross-reactions with other Chlamydia complications such as anorectal strictures and fistulae, which as anorectal strictures and fistulae, which complications such morbidity even after treatment has been can cause substantial a is uncommon, but is completed. Complete death in severe cases. recognized cause of self-limited painless papule in the genital mucosa or in the , or in the mucosa in the genital papule painless self-limited initial lesion,Within six weeks of the often go unnoticed. which can with either a manifests lymphogranuloma venereum secondary or an lymphadenopathy, painful inguinal or femoral florid and discharge or may have rectal proctitis. Patients acute hemorrhagic or have rec- defecation, urge to defecate or incomplete pain, feel an fevers, as such symptoms Systemic itching. anal and bleeding tal lymphogranulomacommon. Finally, tertiary malaise are chills or with chronic inflammatory changes, suchvenereum may present causing genital . as lymphatic obstruction cation tests are much more sensitive in their ability to detect C. tra- cation tests are much more sensitive in their chomatis. are common, and multiple investigations are often undertaken investigations are often undertaken are common, and multiple diagnosis is made. We have seen several before the correct multi- by assessment undergone have who clinic our in patients and cross-sectional ple specialists with unnecessary endoscopy lymphogranuloma CT or magnetic resonance imaging before symptoms. venereum was considered as a cause for their testing sensitive suitably of lack the by hampered been viously have limitedmethods. Traditional chlamydial culture techniques when performedsensitivity (in the range of 30%–50%), especially blindly, without anoscopic guidance. Canada are approved for extragenital use, many laboratories haveCanada are approved for extragenital use, acid amplification conducted independent validation of nucleic to check with localtesting for extragenital sites. Thus, it is important determine availability ofpublic health and laboratory authorities to spp. (Alberto Sever- nucleic acid amplification testing for Chlamydia Lab­ Microbiology National Section, STD and Exanthemata Viral ini, Ottawa: personal com­ oratory, Public Health Agency of Canada, presentation, ourmunication, 2017). At the time of our patient’s Clinicians should behospital had not yet validated a suitable assay. tests alone are not suffi- aware that urine nucleic acid amplification cient, because a substantial proportion of chlamydial infections in men who have sex with men will be extragenital.