Clinical perspectives of Treponema pallidum subsp. Endemicum infection in adults, particularly men who have sex with men in the Kansai area, Japan: A case series.

Bejel, caused by Treponema pallidum subsp. Endemicum (TEN), is a locally transmitted disease among children and juveniles in hot and dry regions. The number of adult cases of TEN infection outside of endemic areas has recently increased. We clinically examined five cases of TEN infection among adult cases previously reported in Japan. TEN infection mainly developed among young to middle-aged men who have sex with men (MSM). The clinical features of cases of TEN infection were similar to those of primary- and secondary-stage T. pallidum subsp. pallidum (TPA) infection. Genital lesions were common as the primary lesion. The clinical features and laboratory parameters of cases of TEN infection were similar to those of TPA infection. Most of the isolated strains had the A2058G mutation in 23S rDNA, which is responsible for resistance to macrolides. We also performed the systemic literature review of the TEN cases outside the endemic countries. The recent reported cases diagnosed with molecular methods shared the clinical features, occurred in young-to middle-aged sexually active persons in urban areas of developed countries and often accompanied with genital lesions, which were distinct from the classic description of bejel. This case series and the literature review provides important clinical insights and will contribute to the clinical detection of this rarely identified disease in developed countries. The surveillance of treponematoses, including TEN infection, using molecular diagnostic techniques is also warranted in developed countries, for the purpose of grasping the epidemic situation and control the local transmission.

Bejel, a Nonvenereal Treponematosis, among Men Who Have Sex with Men, Japan.

Bejel, an endemic treponematosis caused by infection with Treponema pallidum subspecies endemicum, has not been reported in eastern Asia and the Pacific region. We report local spread of bejel among men who have sex with men in Japan. Spread was complicated by venereal syphilis.

Bejel in Cuba: molecular identification of Treponema pallidum subsp. endemicum in patients diagnosed with venereal syphilis.

OBJECTIVES: Bejel, caused by Treponema pallidum subsp. endemicum (TEN), was until now considered as a non-venereal disease endemic in areas with hot and dry climates. This study has identified TEN in clinical samples from Cuban patients previously diagnosed with syphilis. METHODS: We performed sequencing-based molecular typing on 92 samples from Cuban individuals diagnosed with syphilis. Moreover, to differentiate T. pallidum subspecies, multi-locus sequence analysis (MLSA) was designed and was applied to suspicious samples. RESULTS: Nine samples, from six patients, had a nucleotide sequence similarity (at all typing loci) to the Bosnia A genome, which is the infectious agent of bejel. Additionally, MLSA clearly supported a TEN classification for the treponemal samples. Clinical and epidemiological data from the six patients also suggested sexual transmission of bejel as well as the endemicity of this rare treponematosis in Cuba. CONCLUSIONS: Molecular identification of Treponema pallidum subsp. endemicum, the agent of bejel, in Cuban patients diagnosed with syphilis indicates the clear limitations of a diagnosis based exclusively on serology, geographical occurrence, clinical symptoms and anamnestic data. This finding has important implications for Global Public Health Systems, including paradigm changes regarding the location of endemic outbreaks, clinical aspects and transmission of this neglected disease.