title: Buruli ulcer caused by Mycobacterium ulcerans subsp shinshuense: a rare case of familial concurrent occurrence and detection of insertion sequence 2404 in Japan.
abstract: OBJECTIVE
  Buruli ulcer, a severe skin infectious disease caused by Mycobacterium ulcerans, is prevalent in tropical and subtropical areas. Recently, cases of Buruli ulcer have been increasing in Japan. All cases have been sporadic, and to date the pathogenic organism has not been detected in materials from the environment.
  
  
  METHODS
  Three members of the same family were seen with large indurated plaques on their face and extremities in the winter of 2010. Skin biopsy specimens of their lesions showed extensive necrosis of deep dermis and subcutaneous fat. Acid-fast bacilli were detected in each biopsy specimen by Ziehl-Neelsen stain, and bacteriological analysis of cultured microorganisms revealed the strains to be M ulcerans subsp shinshuense. The patients were treated with a combination of antibiotics and surgical debridement. Insertion sequence 2404 was detected from a crayfish captured in a stagnant water channel in the backyard of the family's house.
  
  
  CONCLUSIONS
  We report a rare instance of familial occurrence of Buruli ulcer in Japan. Detection of insertion sequence 2404 from a crayfish suggests that the pathogenic organism may reside in an aquatic environment in Japan, as in other endemic areas. To prevent this serious infectious disease, further investigation is needed to clarify the transmission pathway.
authors.affiliation: Department of Dermatology, Fukushima Medical University School of Medicine, Fukushima, Japan.
authors.forename: Mikio
authors.initials: M
authors.lastname: Ohtsuka
authors.affiliation: Department of Dermatology, Fukushima Medical University School of Medicine, Fukushima, Japan.
authors.forename: Nobuyuki
authors.initials: N
authors.lastname: Kikuchi
authors.affiliation: Department of Dermatology, Fukushima Medical University School of Medicine, Fukushima, Japan.
authors.forename: Toshiyuki
authors.initials: T
authors.lastname: Yamamoto
authors.affiliation: Department of Microbiology, Fukushima Medical University School of Medicine, Fukushima, Japan.
authors.forename: Tatsuo
authors.initials: T
authors.lastname: Suzutani
authors.affiliation: Leprosy Research Center, National Institute of Infectious Diseases, Tokyo, Japan.
authors.forename: Kazue
authors.initials: K
authors.lastname: Nakanaga
authors.affiliation: Leprosy Research Center, National Institute of Infectious Diseases, Tokyo, Japan.
authors.forename: Koichi
authors.initials: K
authors.lastname: Suzuki
authors.affiliation: Leprosy Research Center, National Institute of Infectious Diseases, Tokyo, Japan.
authors.forename: Norihisa
authors.initials: N
authors.lastname: Ishii
mesh_terms: D000328:Adult; D000818:Animals; D000900:Anti-Bacterial Agents / Q000627:therapeutic use*; D003400:Astacoidea / Q000382:microbiology; D001706:Biopsy; D054312:Buruli Ulcer / Q000175:diagnosis / Q000382:microbiology* / Q000473:pathology; D002675:Child, Preschool; D003646:Debridement / Q000379:methods*; D005260:Female; D006801:Humans; D007564:Japan; D008297:Male; D016254:Mutagenesis, Insertional; D019911:Mycobacterium ulcerans / Q000235:genetics / Q000302:isolation & purification*; D009336:Necrosis; D013194:Staining and Labeling
chemical_list: D000900:Anti-Bacterial Agents