Sexually Transmitted Diseases:
Hajdarbegovic, Enes MD; van der Snoek, Eric M. PHD, MD; Ossewaarde, Jacobus M. PHD, MD; van der Meijden, Willem I. PHD, MD; Thio, H. Bing PHD, MD
From the Department of Dermatology, Erasmus Medical Centre Rotterdam, Rotterdam, The Netherlands
Consent form was signed by the patient before publication of the image and the case history.
Correspondence: Enes Hajdarbegovic, MD, Burg. S' Jacobplein 51, 3015 CA, Rotterdam, The Netherlands. E-mail: email@example.com.
Received for publication August 3, 2009, and accepted September 13, 2009.
In this paper we present a case history of a homosexual HIV-positive male with a painless nodule on the penis. Screening for sexually transmitted diseases did not detect any infection until the node perforated spontaneously. A diagnosis of lymphogranuloma venereum was made when chlamydia trachomatis type L2 DNA was extracted from the lesion. This case illustrates that standard screening may not be sufficient for making the diagnosis of lymphogranuloma venereum.
A 48-year-old HIV-positive homosexual male presented to our outpatient clinic for sexually transmitted diseases with a nodule on his penis. Besides the obvious mechanical problem the node was asymptomatic. The lesion had been present for a week now. His CD4 count was 0.21 × 109/L and the viral load was 4.56 × 102. He had been on HAART for 6 months. Five years earlier he had been treated for secondary syphilis with maculopapular rash. He engaged in unprotected active anal intercourse and both passive and active oral sex. In the past 6 months he had had 6 casual sexual partners whose HIV-status was unknown. He denied visiting saunas, sex parties, and using toys. He had not noticed a fever and had lost no weight. He denied having travelled outside the European Union.
On examination we palpated a firm node at the distal end of the prepuce, adjacent to the coronal sulcus. The node could be moved freely. It was ovoid in shape, not painful and approximately 1 cm in diameter. There was a nontender bilateral inguinal lymphadenopathy about 2 cm in size. There was a small genital wart close to the nodule which according to the patient had been present for months. He did not wish to be treated for this wart. Full sexually transmitted diseases screening was performed including nucleic acid amplification test (NAAT) for chlamydia and gonorrhoea in urine, rectal and pharyngeal swabs, and serology for syphilis and HIV. The NAATs (ProbeTec3, BD Dignostic System, Aalst, Belgium) were negative for Chlamydia trachomatis and Neisseria gonorrhoeae on all samples. The VDRL titre was repeatedly undetectable, consistent with treated syphilis. A provisional diagnosis of penile lymphadenopathy of unknown cause was made.
One week later the node had perforated spontaneously and pus emerged from the opening. The lesion was painful now (Fig. 1). Inguinal nodes were bilaterally enlarged to 4 cm and were tender on palpation. They were nonfluctuant and no overlying skin changes could be observed. The sinus was probed and had a length of approximately 2 cm. A darkground specimen showed no spirochetes. C. trachomatis DNA was detected by NAAT in a swab taken from the sinus. Further typing showed serovar L2. The technique and systems we have used are described elsewhere.1 Screening for hepatitis C infection proved negative.
The patient was treated with doxycycline 100 mg bid for 3 weeks and the lesion healed with minimal scarring.
Lymphogranuloma venereum (LGV) is a rare disease in industrialized countries but is now becoming relatively common among HIV+ men who have sex with men (MSM). However, penile lesions remain rare even in this population, though they are now becoming recognized more frequently.2 LGV infection is caused by Chlamydia trachomatis serovars type L1–L3 and is mostly found in MSM. The identifying technique we have used does not allow further sub typing, however, we believe that in our case type L2b was responsible for the infection as in other outbreaks in Europe. Being HIV-positive is one of the most reported risk factors but fisting, rectal drug use, sex toys, and enemas have also been linked with LGV infections.3,4 The organism enters the host through breaches in skin and mucosa where it can evoke a local inflammatory response, which is clinically visible as an ulcer or papule. The site of inoculation in Western European patients is usually located in the rectum. Here chlamydia can cause a range of clinical appearances, from fulminant proctitis to asymptomatic infection. In the second stage the organism resides in the lymphatics and causes inflammation.3 The nodes become tender and fluctuant and eventually rupture. Rupturing nodes are called buboes and are not seen in cases presenting with procitits as draining lymphatics of these regions are found perirectally. In our case, the LGV infection was probably acquired at a site distal of the nodule which may have been in the urethral meatus or a point of entry on the glans or the prepuce. It is highly likely that it was contracted through anal sex with an infected partner. The nodule we found probably represents a pathogenetically enlarged lymph node of the penis. Here it is called a “bubonulus.” Recently, another case report of a bubonulus was published but most cases of LGV present as proctitis rather than a genital lesion.5 Any lymphatic tissue can become infected and reports of tonsillar and cervical buboes have also been published.6
The third stage is characterized by formation of sinuses, abscesses and strictures, or genital elephantiasis due to lymphatic dysfunction.
Standard testing for chlamydial infection in MSM is usually limited to NAAT on samples from mucosal surfaces of urethra, pharynx, and rectum. This case illustrates that some LGV infections may be missed with standard mucosal sampling as the organism is present within the lymphoid tissue. Lymphadenopathy with abscess forming in high-risk patients should alert the clinicians to the possibility of lymphogranuloma venereum. The diagnosis can also be made by NAAT on material aspirated from fluctuant and even nonfluctuant nodes in high-risk patients.4 In our case this could have led to an earlier diagnosis. Also NAAT-testing on atypical anogenitale lesions should be considered in MSM, especially the HIV-positive, when tests for syphilis and herpes are negative.
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