Accepted Manuscript Munchausen Syndrome: Multiple Episodes of Polymicrobial Bacteremia Shimon A. Goldberg, MBBS Zoryana Yezernytski, MD Yitchak Skorochod, MD Eli Ben-Chetrit, MD Amos M. Yinnon, MD Gabriel R. Munter, MD PII:
S0002-9343(14)00671-8
DOI:
10.1016/j.amjmed.2014.07.033
Reference:
AJM 12634
To appear in:
The American Journal of Medicine
Received Date: 20 July 2014 Revised Date:
27 July 2014
Accepted Date: 28 July 2014
Please cite this article as: Goldberg SA, Yezernytski Z, Skorochod Y, Ben-Chetrit E, Yinnon AM, Munter GR, Munchausen Syndrome: Multiple Episodes of Polymicrobial Bacteremia, The American Journal of Medicine (2014), doi: 10.1016/j.amjmed.2014.07.033. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Munchausen Syndrome: Multiple Episodes of Polymicrobial Bacteremia
RI PT
Running title: Munchausen Syndrome and recurrent bacterermia
Shimon A. Goldberg, MBBS,1 Zoryana Yezernytski, MD,1 Yitchak Skorochod, MD,1 Eli Ben-Chetrit, MD,1,2 Amos M. Yinnon, MD1,2
SC
and Gabriel R. Munter, MD1
M AN U
Division of Internal Medicine1 and Infectious Disease Unit,2 Shaare Zedek Medical Center, affiliated with the Hebrew University-Hadassah Medical School, Jerusalem, Israel.
TE D
Address for correspondence: Amos M. Yinnon, MD, Professor and Chair, Division of Internal Medicine, Shaare Zedek Medical Center, P.O. Box
EP
3235, Jerusalem 91031, Israel. Tel: +972-2-6666340, Fax +972-26666840
AC C
Keywords: Munchausen syndrome; bacteremia; polymicrobial bacteremia; factitious; Pichia fermentans
Word count, Case through Discussion: 650
Funding: none. Conflict of interest statement: signed by all authors. All authors had access to all data and a role in writing the manuscript.
1
ACCEPTED MANUSCRIPT
To the Editor,
RI PT
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 and wound
SC
exploration was performed in the operating theatre, but no abnormalities were detected. Post-operatively, the patient developed acute isolated urinary
M AN U
retention and, on uro-neurological assessment, she was diagnosed with Fowler’s syndrome.1 She was discharged after receiving guidance for selfcatheterization.
TE D
In the subsequent four 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
EP
170 sets of blood cultures were taken, of which 28 (16.5%) were positive. Of
AC C
these, 10 (36%) were mono-microbial, while 9 sets (32%) yielded two microbes and another 9 yielded three or more microbes. A total of 20 different microbes were isolated, including Pichia fermentans, a pathogen as yet not described in human disease.2 In the same period, 118 urine cultures were obtained, of which 66 (56%) were positive. Of these, 35 (53%) yielded
2
ACCEPTED MANUSCRIPT
one organism, 22 (33%) grew two organisms and 9 (13.6%) grew ≥3
RI PT
organisms (Table).
Thorough laboratory, immunological and imaging investigations were performed, but no anatomical abnormality was detected. Suspicion of
SC
Munchausen syndrome developed into certainty due to the following features. First, no anatomical or other explanation could be found for
M AN U
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 the medical team with a stone she
TE D
claimed had passed through her urinary catheter. Analysis using x-ray diffraction showed it to be a dolomite stone from the street. Ultrasound and CT at that time showed no evidence of nephrolithiasis or hydronephrosis.
EP
Third, occasionally the oral temperature measured a degree higher than
AC C
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 3
ACCEPTED MANUSCRIPT
manifestations have been reported, including self-inflicted trauma,3 metabolic conditions, dermatologic abnormalities and infectious diseases.
RI PT
Purposely inflicted bacteremia has been described in seven case reports of children, the so-called Munchausen-by-proxy syndrome. A Pubmed search detected only two other cases of self-inflicted bacteremia in an adult, one
SC
involving a single instance bacteremia with three different Bacillus species,
M AN U
the other involving 13 episodes of polymicrobial sepsis.4,5
While with hindsight the diagnosis is clear, the process of diagnosing Munchausen syndrome was complex and gradual. It is difficult to imagine
TE D
that a patient would subject herself to the serious risks associated with selfinduced bloodstream infection. Only after multiple investigations did the treating physicians reluctantly raise the only reasonable explanation for this
EP
patient’s recurrent septic episodes. Psychiatric consultation confirmed a
AC C
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.
4
ACCEPTED MANUSCRIPT
References
RI PT
1. Swinn MJ, Wiseman OJ, Lowe E, Fowler CJ. The cause and natural history of isolated urinary retention in young women. J Urol 2002; 167:
SC
151-6.
2. Sanna ML, Zara G, Zara S, Migheli Q, Budroni M, Mannazzu I. A
M AN U
putative phospholipase C is involved in Pichia fermentans dimorphic transition. Biochim Biophys Acta. 2014;1840:344-9. 3. Yinnon AM, Zandman-Goddard G, Moreb J, Fisch R, Abraham AS.
1988; 37: 187-191.
TE D
Factitious injury of an extremity: a Munchausen variant. Am Fam Phys
EP
4. Galanos J, Perera S, Smith H, O’Neal D, Sheorey H, Waters MJ. Bacteremia due to three Bacillus species in a case of Munchausen's
AC C
syndrome. J Clin Microbiol. 2003; 4:2247-8. 5. Meessen NEL, Walenkamp GHIM, Jacobs JA. Munchausen's microbes? N Engl J Med. 1998; 339:1717-8.
5
ACCEPTED MANUSCRIPT
Table 1: Episodes of documented bacteremia and positive urine cultures*
RI PT
Total n=161 28 27 26 22 11 11 5 4 6 4 4 2 1 2 2 2 1 1 1 1 161
M AN U
TE D
EP
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20
Blood Urine n=55 n=106 Proteus mirabilis 6 22 Pseudomonas aeruginosa 3 24 Enterococcus 5 21 E. coli 2 20 Pichia fermentans 10 1 Candida 2 9 Lactobacillus spp 5 0 Mixed growth of Gram + species 4 0 Staphylococcus coag. negative 6 0 Streptococcus viridans 4 0 Klebsiella pneumoniae 0 4 Morganella morganii 0 2 Klebsiella oxytoca 0 1 Neisseria spp 2 0 Bacillus spp 2 0 Enterobacter cloacae 1 1 Stenotrophomonas maltophilia 1 0 Acinetobacter 0 1 Micrococcus 1 0 Diphtheroids 1 0 Total 55 106
SC
Organism
AC C
* 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
1
S0002-9343(14)00671-8
DOI:
10.1016/j.amjmed.2014.07.033
Reference:
AJM 12634
To appear in:
The American Journal of Medicine
Received Date: 20 July 2014 Revised Date:
27 July 2014
Accepted Date: 28 July 2014
Please cite this article as: Goldberg SA, Yezernytski Z, Skorochod Y, Ben-Chetrit E, Yinnon AM, Munter GR, Munchausen Syndrome: Multiple Episodes of Polymicrobial Bacteremia, The American Journal of Medicine (2014), doi: 10.1016/j.amjmed.2014.07.033. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Munchausen Syndrome: Multiple Episodes of Polymicrobial Bacteremia
RI PT
Running title: Munchausen Syndrome and recurrent bacterermia
Shimon A. Goldberg, MBBS,1 Zoryana Yezernytski, MD,1 Yitchak Skorochod, MD,1 Eli Ben-Chetrit, MD,1,2 Amos M. Yinnon, MD1,2
SC
and Gabriel R. Munter, MD1
M AN U
Division of Internal Medicine1 and Infectious Disease Unit,2 Shaare Zedek Medical Center, affiliated with the Hebrew University-Hadassah Medical School, Jerusalem, Israel.
TE D
Address for correspondence: Amos M. Yinnon, MD, Professor and Chair, Division of Internal Medicine, Shaare Zedek Medical Center, P.O. Box
EP
3235, Jerusalem 91031, Israel. Tel: +972-2-6666340, Fax +972-26666840
AC C
Keywords: Munchausen syndrome; bacteremia; polymicrobial bacteremia; factitious; Pichia fermentans
Word count, Case through Discussion: 650
Funding: none. Conflict of interest statement: signed by all authors. All authors had access to all data and a role in writing the manuscript.
1
ACCEPTED MANUSCRIPT
To the Editor,
RI PT
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 and wound
SC
exploration was performed in the operating theatre, but no abnormalities were detected. Post-operatively, the patient developed acute isolated urinary
M AN U
retention and, on uro-neurological assessment, she was diagnosed with Fowler’s syndrome.1 She was discharged after receiving guidance for selfcatheterization.
TE D
In the subsequent four 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
EP
170 sets of blood cultures were taken, of which 28 (16.5%) were positive. Of
AC C
these, 10 (36%) were mono-microbial, while 9 sets (32%) yielded two microbes and another 9 yielded three or more microbes. A total of 20 different microbes were isolated, including Pichia fermentans, a pathogen as yet not described in human disease.2 In the same period, 118 urine cultures were obtained, of which 66 (56%) were positive. Of these, 35 (53%) yielded
2
ACCEPTED MANUSCRIPT
one organism, 22 (33%) grew two organisms and 9 (13.6%) grew ≥3
RI PT
organisms (Table).
Thorough laboratory, immunological and imaging investigations were performed, but no anatomical abnormality was detected. Suspicion of
SC
Munchausen syndrome developed into certainty due to the following features. First, no anatomical or other explanation could be found for
M AN U
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 the medical team with a stone she
TE D
claimed had passed through her urinary catheter. Analysis using x-ray diffraction showed it to be a dolomite stone from the street. Ultrasound and CT at that time showed no evidence of nephrolithiasis or hydronephrosis.
EP
Third, occasionally the oral temperature measured a degree higher than
AC C
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 3
ACCEPTED MANUSCRIPT
manifestations have been reported, including self-inflicted trauma,3 metabolic conditions, dermatologic abnormalities and infectious diseases.
RI PT
Purposely inflicted bacteremia has been described in seven case reports of children, the so-called Munchausen-by-proxy syndrome. A Pubmed search detected only two other cases of self-inflicted bacteremia in an adult, one
SC
involving a single instance bacteremia with three different Bacillus species,
M AN U
the other involving 13 episodes of polymicrobial sepsis.4,5
While with hindsight the diagnosis is clear, the process of diagnosing Munchausen syndrome was complex and gradual. It is difficult to imagine
TE D
that a patient would subject herself to the serious risks associated with selfinduced bloodstream infection. Only after multiple investigations did the treating physicians reluctantly raise the only reasonable explanation for this
EP
patient’s recurrent septic episodes. Psychiatric consultation confirmed a
AC C
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.
4
ACCEPTED MANUSCRIPT
References
RI PT
1. Swinn MJ, Wiseman OJ, Lowe E, Fowler CJ. The cause and natural history of isolated urinary retention in young women. J Urol 2002; 167:
SC
151-6.
2. Sanna ML, Zara G, Zara S, Migheli Q, Budroni M, Mannazzu I. A
M AN U
putative phospholipase C is involved in Pichia fermentans dimorphic transition. Biochim Biophys Acta. 2014;1840:344-9. 3. Yinnon AM, Zandman-Goddard G, Moreb J, Fisch R, Abraham AS.
1988; 37: 187-191.
TE D
Factitious injury of an extremity: a Munchausen variant. Am Fam Phys
EP
4. Galanos J, Perera S, Smith H, O’Neal D, Sheorey H, Waters MJ. Bacteremia due to three Bacillus species in a case of Munchausen's
AC C
syndrome. J Clin Microbiol. 2003; 4:2247-8. 5. Meessen NEL, Walenkamp GHIM, Jacobs JA. Munchausen's microbes? N Engl J Med. 1998; 339:1717-8.
5
ACCEPTED MANUSCRIPT
Table 1: Episodes of documented bacteremia and positive urine cultures*
RI PT
Total n=161 28 27 26 22 11 11 5 4 6 4 4 2 1 2 2 2 1 1 1 1 161
M AN U
TE D
EP
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20
Blood Urine n=55 n=106 Proteus mirabilis 6 22 Pseudomonas aeruginosa 3 24 Enterococcus 5 21 E. coli 2 20 Pichia fermentans 10 1 Candida 2 9 Lactobacillus spp 5 0 Mixed growth of Gram + species 4 0 Staphylococcus coag. negative 6 0 Streptococcus viridans 4 0 Klebsiella pneumoniae 0 4 Morganella morganii 0 2 Klebsiella oxytoca 0 1 Neisseria spp 2 0 Bacillus spp 2 0 Enterobacter cloacae 1 1 Stenotrophomonas maltophilia 1 0 Acinetobacter 0 1 Micrococcus 1 0 Diphtheroids 1 0 Total 55 106
SC
Organism
AC C
* 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
1
