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Munchausen Syndrome: Multiple Episodes of Polymicrobial Bacteremia

      To the Editor:
      A 26-year-old female patient underwent abdominoplasty after significant weight loss due to dieting. One month later she was readmitted due to wound infection, which failed to heal with oral antibiotics. Wound exploration was performed in the operating theatre, but no abnormalities were detected. Postoperatively, the patient developed acute isolated urinary retention and, on uroneurological assessment, she was diagnosed with Fowler's syndrome.
      • Swinn M.J.
      • Wiseman O.J.
      • Lowe E.
      • Fowler C.J.
      The cause and natural history of isolated urinary retention in young women.
      She was discharged after receiving guidance for self-catheterization.
      In the subsequent 4 years the patient presented to the hospital 54 times and was admitted 40 times; in total, 596 inpatient days. Presenting complaints included abdominal pain, nausea, fever, and weakness. A total of 170 sets of blood cultures were taken, of which 28 (16.5%) were positive. Of these, 10 (36%) were mono-microbial, while 9 sets (32%) yielded 2 microbes and another 9 yielded 3 or more microbes. A total of 20 different microbes were isolated, including Pichia fermentans, a pathogen as yet not described in human disease.
      • Sanna M.L.
      • Zara G.
      • Zara S.
      • Migheli Q.
      • Budroni M.
      • Mannazzu I.
      A putative phospholipase C is involved in Pichia fermentans dimorphic transition.
      In the same period, 118 urine cultures were obtained, of which 66 (56%) were positive. Of these, 35 (53%) yielded one organism, 22 (33%) grew 2 organisms, and 9 (13.6%) grew ≥3 organisms (Table).
      TableEpisodes of Documented Bacteremia and Positive Urine Cultures
      Due to cases of polymicrobial growths, the total number of organisms in both blood and urine exceeds the number of positive sets appearing in the text.
      OrganismBlood

      n = 55
      Urine

      n = 106
      Total

      n = 161
      1Proteus mirabilis62228
      2Pseudomonas aeruginosa32427
      3Enterococcus52126
      4E. coli22022
      5Pichia fermentans10111
      6Candida2911
      7Lactobacillus spp505
      8Mixed growth of Gram + species404
      9Staphylococcus coag. negative606
      10Streptococcus viridans404
      11Klebsiella pneumoniae044
      12Morganella morganii022
      13Klebsiella oxytoca011
      14Neisseria spp202
      15Bacillus spp202
      16Enterobacter cloacae112
      17Stenotrophomonas maltophilia101
      18Acinetobacter011
      19Micrococcus101
      20Diphtheroids101
      Total55106161
      Due to cases of polymicrobial growths, the total number of organisms in both blood and urine exceeds the number of positive sets appearing in the text.
      Thorough laboratory, immunological, and imaging investigations were performed, but no anatomical abnormality was detected. Suspicion of Munchausen syndrome developed into certainty due to the following features. First, no anatomical or other explanation could be found for recurrent sepsis, especially the polymicrobial kind. Of the 28 episodes of bacteremia, only in 4 (14%) was the same organism isolated from the urine. Second, once the patient presented to the medical team with a stone she claimed had passed through her urinary catheter. Analysis using radiographic diffraction showed it to be a dolomite stone from the street. Ultrasound and computed tomography at that time showed no evidence of nephrolithiasis or hydronephrosis. Third, occasionally, the oral temperature measured a degree higher than rectal. Finally, on another occasion she complained of phlebitis secondary to an intravenous catheter and requested a new one. The “erythema” was successfully removed with an alcohol swab.
      Munchausen syndrome can range from a single episode of factitious illness to a chronic condition with significant morbidity and even mortality. Various manifestations have been reported, including self-inflicted trauma,
      • Yinnon A.M.
      • Zandman-Goddard G.
      • Moreb J.
      • Fisch R.
      • Abraham A.S.
      Factitious injury of an extremity: a Munchausen variant.
      metabolic conditions, dermatologic abnormalities, and infectious diseases. Purposely inflicted bacteremia has been described in 7 case reports of children, the so-called Munchausen-by-proxy syndrome. A PubMed search detected only 2 other cases of self-inflicted bacteremia in an adult, one involving a single-instance bacteremia with 3 different Bacillus species, the other involving 13 episodes of polymicrobial sepsis.
      • Galanos J.
      • Perera S.
      • Smith H.
      • O'Neal D.
      • Sheorey H.
      • Waters M.J.
      Bacteremia due to three Bacillus species in a case of Munchausen's syndrome.
      • Meessen N.E.L.
      • Walenkamp G.H.I.M.
      • Jacobs J.A.
      Munchausen's microbes?.
      While with hindsight the diagnosis is clear, the process of diagnosing Munchausen syndrome was complex and gradual. It is difficult to imagine that a patient would subject herself to the serious risks associated with self-induced bloodstream infection. Only after multiple investigations did the treating physicians reluctantly raise the only reasonable explanation for this patient's recurrent septic episodes. Psychiatric consultation confirmed a pathologic relationship with her mother, as well as other mental distress. When confronted, she denied infecting herself; unusually, this patient continues to present to the same hospital after confrontation, whereas patients typically turn to other institutions after confrontation.

      References

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        • Lowe E.
        • Fowler C.J.
        The cause and natural history of isolated urinary retention in young women.
        J Urol. 2002; 167: 151-156
        • Sanna M.L.
        • Zara G.
        • Zara S.
        • Migheli Q.
        • Budroni M.
        • Mannazzu I.
        A putative phospholipase C is involved in Pichia fermentans dimorphic transition.
        Biochim Biophys Acta. 2014; 1840: 344-349
        • Yinnon A.M.
        • Zandman-Goddard G.
        • Moreb J.
        • Fisch R.
        • Abraham A.S.
        Factitious injury of an extremity: a Munchausen variant.
        Am Fam Physician. 1988; 37: 187-191
        • Galanos J.
        • Perera S.
        • Smith H.
        • O'Neal D.
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        • Waters M.J.
        Bacteremia due to three Bacillus species in a case of Munchausen's syndrome.
        J Clin Microbiol. 2003; 4: 2247-2248
        • Meessen N.E.L.
        • Walenkamp G.H.I.M.
        • Jacobs J.A.
        Munchausen's microbes?.
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