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Urinary incontinence due to the presence of necrotic adult Schistosoma haematobium parasite in the bladder following travel to Egypt
Travel Medicine and Infectious Disease (2012) 10, 205e207
Available online at www.sciencedirect.com
journal homepage: www.elsevierhealth.com/journals/tmid
CASE REPORT
Urinary incontinence due to the presence of necrotic adult Schistosoma haematobium parasite in the bladder following travel to Egypt N. Papoutsoglou a,*, D. Tappe b, P. Demmer c, A. Kocot a, H. Riedmiller a a Department of Urology and Pediatric Urology, Julius Maximilian University Medical School, Oberdu¨rrbacher Str. 6, 97080 Wu¨rzburg, Germany b Institute of Hygiene and Microbiology, Julius Maximilian University Medical School, Josef-Schneider-Str. 2, 97080 Wu¨rzburg, Germany c Institute of Pathology, Julius Maximilian University Medical School, Josef-Schneider-Str. 2, 97080 Wu¨rzburg, Germany
Received 16 September 2011; received in revised form 30 April 2012; accepted 8 May 2012 Available online 1 June 2012
KEYWORDS Schistosoma haematobium; Parasitic infections; Urinary incontinence; Haematuria; Cystoscopy
Summary A case of seronegative urinary Schistosomiasis is reported in a 68-year-old Caucasian male presenting with urgency of micturition and incontinence several months after bathing in a chlorinated pool of a first class hotel in Egypt. The symptoms were initiated by a necrotic adult Schistosoma haematobium parasite found in the urinary bladder following a cystoscopic examination. The purpose of this report is to describe this probable and uncommon source of Schistosomiasis, to demonstrate that Schistosoma parasites can also be found in the urinary bladder and to emphasize the importance of travel history. ª 2012 Elsevier Ltd. All rights reserved.
Case report A 68-year-old Caucasian male with a past medical history of Parkinson’s disease was referred with urgency of micturition and incontinence, which developed a few weeks previously. Previous cystoscopy had shown an area of abnormal bladder
* Corresponding author. Tel.: þ49 93120132001; fax: þ49 931 20132009. E-mail address: [email protected] (N. Papoutsoglou).
urothelium. On digital rectal examination the prostate was found to be enlarged and benign. Urinalysis showed microscopic haematuria (250 erythrocytes/mL) and leucocyturia. Bacterial growth was not detected by urine culture. The patient had taken no anticoagulants and no malignancies of the urogenital tract were reported in his personal and family past medical history. Full blood count, eosinophil count, prothrombin time, bleeding time, creatinine and prostate specific antigen (PSA) were normal. Ultrasonographic examination of the abdomen, kidneys and bladder as well as the intravenous urogram was unremarkable. Endoscopic
1477-8939/$ - see front matter ª 2012 Elsevier Ltd. All rights reserved. doi:10.1016/j.tmaid.2012.05.001
206 investigation of the lower urinary tract was performed, revealing normal urethra and mild prostatic hypertrophy. Atypical bladder mucosa with an aspect of “grains of sand” measuring about 10 mm in diameter was seen between the posterior and superior wall of the urinary bladder. No bleeding site or exophytic bladder tumour was detected. However, a vermiform white convolute with a length of 2 cm was found floating in the urine from the bladder and was transurethrally biopsied. Histopathological examination showed mostly scaled-off urothelium with mediocre sclerotic thickening of the lamina propria, minimal inflammatory reaction with a few eosinophils and, in the epithelial layer of the mucosa, a necrotic Schistosoma egg (Fig. 1) and a necrotic adult parasite (Fig. 2). Serology for Schistosomiasis using indirect immunofluorescence, indirect haemagglutination test and ELISA was negative. Parasitological examination of urine or stool samples was not performed. When asked about his travel history, the patient recalled a 2 weeks vacation in a first class hotel in Egypt (Red Sea Coast) 7 months before the onset of the urge urinary incontinence, where he had experienced a short episode of diarrhoea. No other visits in Schistosomiasis endemic countries were reported. Owing to his motor deficiency due to Parkinson’s disease, he had used the hotel’s chlorinated swimming pool only once. Any other contact with possibly Schistosome containing freshwater, such as canals, rivers and ponds, was denied. The patient received Praziquantel 40 mg/kg as antihelminthic chemotherapy. Urinary urgency and incontinence decreased rapidly and urinalysis showed normal results. Six months later, free of urinary symptoms, the patient developed cancer of the colon and underwent hemicolectomy and palliative antineoplastic chemotherapy.
Discussion Schistosomiasis, which is also known as Bilharziasis, is a significant health problem in many tropical and
Figure 1 Necrotic schistosoma egg (arrow) in mucosal layer of the urinary bladder. The typical small nuclei in irregular distribution are still visible. The structure measures 112 43 mm and is thus consistent with ova of Schistosoma haematobium, which are slightly smaller than those of Schistosoma mansoni. Magnification 100, periodic acid-Schiff stain.
N. Papoutsoglou et al. subtropical countries. According to the World Health Organization, an estimated 200 million people, mainly children, are affected.1,2 Schistosoma haematobium, which is responsible for the urinary Schistosomiasis, is endemic in most African countries and the Middle East. Human infection occurs after penetration of the skin by cercariae following exposure to standing waters that harbor infected snails and have been contaminated by urine of infected persons. Salted and chlorinated water count as safe. The snails represent the intermediate host of infection; humans on the other hand are the definitive host. After penetrating the skin S. haematobium adult parasites lodge in the pelvic blood vessels, multiply and give birth to thousands of ovules. The deposition of ovules provokes damage of the bladder mucosa and may promote carcinogenesis.2e5 The diagnosis depends on the physician’s awareness especially by a history of bathing in standing waters in endemic areas. The definitive diagnosis of urinary Schistosomiasis is established by the detection of species-specific, typically formed eggs in urine, by detection of Schistosome antigens in serum or urine, or by genus-specific serology.6 In this case the only symptom that determined the patient to seek medical assistance was the urinary urgency and incontinence and not the usual finding of frank haematuria although urinalysis revealed microscopic haematuria. Interestingly, a detached free-floating convolute of the bladder mucosa containing a necrotic adult parasite was retrieved endoscopically and it was the histological examination of the specimen which led to the diagnosis of Schistosomiasis. In lack of other medical causes, this tissue convolute presumably initiated the urinary urgency and incontinence. The finding of an adult Schistosome parasite in the bladder mucosal convolute appears unusual, as the parasites normally lay eggs in the venous plexus of the bladder and eliminate viable eggs by urine. The reported gastrointestinal symptoms were most likely an episode of traveller’s diarrhoea, as the symptoms of acute Schistosomiasis (i.e. exanthema, urticaria, fever, abdominal pain and also diarrhoea) usually start 2e6 weeks after infection. Such symptoms as well as the typical swimmer’s itch due to cercarial penetration were not reported. Serology for Schistosomiasis was negative despite the use of multiple tests. The enzyme-linked immunoelectrotransfer blot assay though (EITB) was not performed.6,7 However, a negative serology does not exclude infection, and the seronegative window for Schistosomiasis may be up to 27 weeks long.8 Also noteworthy, the patient bathed only once in an outdoor chlorinated swimming pool of a first class hotel, which retrospectively was identified as the most likely source of infection. As swimming in salt water and chlorinated pools is generally considered to be safe, the question remains if the chlorine level of the pool’s water has been too low. However, the use of showers or even tap and table water from dubious sources cannot be excluded. Cases of urinary Schistosomiasis in Brazilian soldiers returning from Mozambique after serving four months on a United Nations peace mission have also been reported. The infection occurred after swimming in a contaminated river during their leisure time. Brazil and South America though, do not count as endemic regions for urinary Schistosomiasis and no autochthonous cases have been reported so far.9 In Egypt, Schistosomiasis due to S. haematobium is endemic in the
Urinary incontinence due to the presence of Schistosoma haematobium parasite in the bladder
207
Figure 2 Partial sections through entirely necrotic adult schistosoma. A, vacuolization of parasite parenchyma (arrowhead). Note small nuclei of the parasite. B, two phagocytes (arrow) are ingesting remnants of the adult schistosoma (arrowhead). Magnification 400, periodic acid-Schiff stain.
Nile river delta. S. haematobium is also found throughout the Nile valley in canals and irrigation ditches and along the Suez Canal.8 Travel history can be beneficial in cases in which usual conditions or diseases cannot interpret the presenting symptoms even if they are not suggestive of tropical aetiology. In all cases of Schistosomiasis of the urinary tract the treatment of choice is a single oral dose of Praziquantel 40 mg/kg. Recent randomized exploratory open-label trials have also shown the benefit of Mefloquineartesunate in individuals coinfected with Plasmodium falciparum showing high efficacy against both.10,11 From a urological point of view cystoscopy and subsequent biopsy of atypical bladder mucosa should be offered to all symptomatic individuals coming or returning from endemic regions even in the absence of detectable parasite eggs in three consecutive 24-h urine samples. For symptomatic individuals cystoscopy should be performed even if they have already been treated with antihelminthics, as therapeutic failure of Praziquantel especially in chronic Schistosomiasis of the urinary tract has been described.9,12,13
Conflict of interest All authors have seen and agree with the contents and there is no financial interest to report. We certify that the submission is original, has not been published before and is not currently being considered for publication elsewhere. We further confirm that the order of authors listed in the manuscript has been approved by all of us. Mr. Papoutsoglou is the main author and guarantor of the paper.
References 1. Kehinde EO, Anim JT, Hira PR. Parasites of urological importance. Urol Int 2008;81(1):1e13.
2. Engels D, Chitsulo L, Montresor A, Savioli L. The global epidemiological situation of schistosomiasis and new approaches to control and research. Acta Trop 2002;82(2):139e46. 3. Tzanetou K, Adamis G, Delis V, Moustakas G, Choreftaki T, Papaliodi E, et al. Urinary tract Schistosoma haematobium infection: a case report. J Travel Med 2007;14(5):334e7. 4. Vennervald BJ, Polman K. Helminths and malignancy. Parasite Immunol 2009;31(11):686e96. 5. Sheweita SA, El-Shahat FG, Bazeed MA, Abu El-Maati MR, O’ Connor PJ. Effects of Schistosoma haematobium infection on drug-metabolizing enzymes in human bladder cancer tissues. Cancer Lett 2004;205(1):15e21. 6. Davis Andrew. Schistosomiasis. Manson’s tropical diseases. 22nd ed. Saunders Elsevier; 2009. 7. da Silva IM, Tsang V, Noh J, Rey L, Conceisao MH. Clinical and laboratorial evaluation of urinary schistosomiasis in Brazilians after staying in Mozambique. Rev Soc Bras Med Trop 2006; 39(3):272e4. 8. Jones ME, Mitchell RG, Leen CL. Long seronegative window in schistosoma infection. Lancet 1992;340(8834e8835):1549e50. 9. Enk MJ, Amaral GL, Costa e Silva MF, Silveira-Lemos D, Teixeira-Carvalho A, Martins-Filho OA, et al. Rural tourism: a risk factor for schistosomiasis transmission in Brazil. Mem Inst Oswaldo Cruz 2010;105(4):537e40. 10. Danso-Appiah A, Garner P, Olliaro PL, Utzinger J. Treatment of urinary schistosomiasis: methodological issues and research needs identified through a Cochrane systematic review. Parasitology 2009;136(13):1837e49. 11. Keiser J, N’Guessan NA, Adoubryn KD, Silue KD, Vounatsou P, Hatz C, et al. Efficacy and safety of mefloquine, artesunate, mefloquine-artesunate, and praziquantel against Schistosoma haematobium: randomized, exploratory open-label trial. Clin Infect Dis 2000;50(9):1205e13. 12. da Silva IM, Thiengo R, Conceicao MH, Rey L, Lenzi HL, Filho PE, et al. Therapeutic failure of praziquantel in the treatment of Schistosoma haematobium infection in Brazilians returning from Africa. Mem Inst Oswaldo Cruz 2005;100(4):445e9. 13. da Silva IM, Thiengo R, Conceicao MH, Rey L, Pereira Filho E, Ribeiro PC. Cystoscopy in the diagnosis and follow-up of urinary schistosomiasis in Brazilian soldiers returning from Mozambique, Africa. Rev Inst Med Trop Sao Paulo 2006;48(1): 39e42.
Available online at www.sciencedirect.com
journal homepage: www.elsevierhealth.com/journals/tmid
CASE REPORT
Urinary incontinence due to the presence of necrotic adult Schistosoma haematobium parasite in the bladder following travel to Egypt N. Papoutsoglou a,*, D. Tappe b, P. Demmer c, A. Kocot a, H. Riedmiller a a Department of Urology and Pediatric Urology, Julius Maximilian University Medical School, Oberdu¨rrbacher Str. 6, 97080 Wu¨rzburg, Germany b Institute of Hygiene and Microbiology, Julius Maximilian University Medical School, Josef-Schneider-Str. 2, 97080 Wu¨rzburg, Germany c Institute of Pathology, Julius Maximilian University Medical School, Josef-Schneider-Str. 2, 97080 Wu¨rzburg, Germany
Received 16 September 2011; received in revised form 30 April 2012; accepted 8 May 2012 Available online 1 June 2012
KEYWORDS Schistosoma haematobium; Parasitic infections; Urinary incontinence; Haematuria; Cystoscopy
Summary A case of seronegative urinary Schistosomiasis is reported in a 68-year-old Caucasian male presenting with urgency of micturition and incontinence several months after bathing in a chlorinated pool of a first class hotel in Egypt. The symptoms were initiated by a necrotic adult Schistosoma haematobium parasite found in the urinary bladder following a cystoscopic examination. The purpose of this report is to describe this probable and uncommon source of Schistosomiasis, to demonstrate that Schistosoma parasites can also be found in the urinary bladder and to emphasize the importance of travel history. ª 2012 Elsevier Ltd. All rights reserved.
Case report A 68-year-old Caucasian male with a past medical history of Parkinson’s disease was referred with urgency of micturition and incontinence, which developed a few weeks previously. Previous cystoscopy had shown an area of abnormal bladder
* Corresponding author. Tel.: þ49 93120132001; fax: þ49 931 20132009. E-mail address: [email protected] (N. Papoutsoglou).
urothelium. On digital rectal examination the prostate was found to be enlarged and benign. Urinalysis showed microscopic haematuria (250 erythrocytes/mL) and leucocyturia. Bacterial growth was not detected by urine culture. The patient had taken no anticoagulants and no malignancies of the urogenital tract were reported in his personal and family past medical history. Full blood count, eosinophil count, prothrombin time, bleeding time, creatinine and prostate specific antigen (PSA) were normal. Ultrasonographic examination of the abdomen, kidneys and bladder as well as the intravenous urogram was unremarkable. Endoscopic
1477-8939/$ - see front matter ª 2012 Elsevier Ltd. All rights reserved. doi:10.1016/j.tmaid.2012.05.001
206 investigation of the lower urinary tract was performed, revealing normal urethra and mild prostatic hypertrophy. Atypical bladder mucosa with an aspect of “grains of sand” measuring about 10 mm in diameter was seen between the posterior and superior wall of the urinary bladder. No bleeding site or exophytic bladder tumour was detected. However, a vermiform white convolute with a length of 2 cm was found floating in the urine from the bladder and was transurethrally biopsied. Histopathological examination showed mostly scaled-off urothelium with mediocre sclerotic thickening of the lamina propria, minimal inflammatory reaction with a few eosinophils and, in the epithelial layer of the mucosa, a necrotic Schistosoma egg (Fig. 1) and a necrotic adult parasite (Fig. 2). Serology for Schistosomiasis using indirect immunofluorescence, indirect haemagglutination test and ELISA was negative. Parasitological examination of urine or stool samples was not performed. When asked about his travel history, the patient recalled a 2 weeks vacation in a first class hotel in Egypt (Red Sea Coast) 7 months before the onset of the urge urinary incontinence, where he had experienced a short episode of diarrhoea. No other visits in Schistosomiasis endemic countries were reported. Owing to his motor deficiency due to Parkinson’s disease, he had used the hotel’s chlorinated swimming pool only once. Any other contact with possibly Schistosome containing freshwater, such as canals, rivers and ponds, was denied. The patient received Praziquantel 40 mg/kg as antihelminthic chemotherapy. Urinary urgency and incontinence decreased rapidly and urinalysis showed normal results. Six months later, free of urinary symptoms, the patient developed cancer of the colon and underwent hemicolectomy and palliative antineoplastic chemotherapy.
Discussion Schistosomiasis, which is also known as Bilharziasis, is a significant health problem in many tropical and
Figure 1 Necrotic schistosoma egg (arrow) in mucosal layer of the urinary bladder. The typical small nuclei in irregular distribution are still visible. The structure measures 112 43 mm and is thus consistent with ova of Schistosoma haematobium, which are slightly smaller than those of Schistosoma mansoni. Magnification 100, periodic acid-Schiff stain.
N. Papoutsoglou et al. subtropical countries. According to the World Health Organization, an estimated 200 million people, mainly children, are affected.1,2 Schistosoma haematobium, which is responsible for the urinary Schistosomiasis, is endemic in most African countries and the Middle East. Human infection occurs after penetration of the skin by cercariae following exposure to standing waters that harbor infected snails and have been contaminated by urine of infected persons. Salted and chlorinated water count as safe. The snails represent the intermediate host of infection; humans on the other hand are the definitive host. After penetrating the skin S. haematobium adult parasites lodge in the pelvic blood vessels, multiply and give birth to thousands of ovules. The deposition of ovules provokes damage of the bladder mucosa and may promote carcinogenesis.2e5 The diagnosis depends on the physician’s awareness especially by a history of bathing in standing waters in endemic areas. The definitive diagnosis of urinary Schistosomiasis is established by the detection of species-specific, typically formed eggs in urine, by detection of Schistosome antigens in serum or urine, or by genus-specific serology.6 In this case the only symptom that determined the patient to seek medical assistance was the urinary urgency and incontinence and not the usual finding of frank haematuria although urinalysis revealed microscopic haematuria. Interestingly, a detached free-floating convolute of the bladder mucosa containing a necrotic adult parasite was retrieved endoscopically and it was the histological examination of the specimen which led to the diagnosis of Schistosomiasis. In lack of other medical causes, this tissue convolute presumably initiated the urinary urgency and incontinence. The finding of an adult Schistosome parasite in the bladder mucosal convolute appears unusual, as the parasites normally lay eggs in the venous plexus of the bladder and eliminate viable eggs by urine. The reported gastrointestinal symptoms were most likely an episode of traveller’s diarrhoea, as the symptoms of acute Schistosomiasis (i.e. exanthema, urticaria, fever, abdominal pain and also diarrhoea) usually start 2e6 weeks after infection. Such symptoms as well as the typical swimmer’s itch due to cercarial penetration were not reported. Serology for Schistosomiasis was negative despite the use of multiple tests. The enzyme-linked immunoelectrotransfer blot assay though (EITB) was not performed.6,7 However, a negative serology does not exclude infection, and the seronegative window for Schistosomiasis may be up to 27 weeks long.8 Also noteworthy, the patient bathed only once in an outdoor chlorinated swimming pool of a first class hotel, which retrospectively was identified as the most likely source of infection. As swimming in salt water and chlorinated pools is generally considered to be safe, the question remains if the chlorine level of the pool’s water has been too low. However, the use of showers or even tap and table water from dubious sources cannot be excluded. Cases of urinary Schistosomiasis in Brazilian soldiers returning from Mozambique after serving four months on a United Nations peace mission have also been reported. The infection occurred after swimming in a contaminated river during their leisure time. Brazil and South America though, do not count as endemic regions for urinary Schistosomiasis and no autochthonous cases have been reported so far.9 In Egypt, Schistosomiasis due to S. haematobium is endemic in the
Urinary incontinence due to the presence of Schistosoma haematobium parasite in the bladder
207
Figure 2 Partial sections through entirely necrotic adult schistosoma. A, vacuolization of parasite parenchyma (arrowhead). Note small nuclei of the parasite. B, two phagocytes (arrow) are ingesting remnants of the adult schistosoma (arrowhead). Magnification 400, periodic acid-Schiff stain.
Nile river delta. S. haematobium is also found throughout the Nile valley in canals and irrigation ditches and along the Suez Canal.8 Travel history can be beneficial in cases in which usual conditions or diseases cannot interpret the presenting symptoms even if they are not suggestive of tropical aetiology. In all cases of Schistosomiasis of the urinary tract the treatment of choice is a single oral dose of Praziquantel 40 mg/kg. Recent randomized exploratory open-label trials have also shown the benefit of Mefloquineartesunate in individuals coinfected with Plasmodium falciparum showing high efficacy against both.10,11 From a urological point of view cystoscopy and subsequent biopsy of atypical bladder mucosa should be offered to all symptomatic individuals coming or returning from endemic regions even in the absence of detectable parasite eggs in three consecutive 24-h urine samples. For symptomatic individuals cystoscopy should be performed even if they have already been treated with antihelminthics, as therapeutic failure of Praziquantel especially in chronic Schistosomiasis of the urinary tract has been described.9,12,13
Conflict of interest All authors have seen and agree with the contents and there is no financial interest to report. We certify that the submission is original, has not been published before and is not currently being considered for publication elsewhere. We further confirm that the order of authors listed in the manuscript has been approved by all of us. Mr. Papoutsoglou is the main author and guarantor of the paper.
References 1. Kehinde EO, Anim JT, Hira PR. Parasites of urological importance. Urol Int 2008;81(1):1e13.
2. Engels D, Chitsulo L, Montresor A, Savioli L. The global epidemiological situation of schistosomiasis and new approaches to control and research. Acta Trop 2002;82(2):139e46. 3. Tzanetou K, Adamis G, Delis V, Moustakas G, Choreftaki T, Papaliodi E, et al. Urinary tract Schistosoma haematobium infection: a case report. J Travel Med 2007;14(5):334e7. 4. Vennervald BJ, Polman K. Helminths and malignancy. Parasite Immunol 2009;31(11):686e96. 5. Sheweita SA, El-Shahat FG, Bazeed MA, Abu El-Maati MR, O’ Connor PJ. Effects of Schistosoma haematobium infection on drug-metabolizing enzymes in human bladder cancer tissues. Cancer Lett 2004;205(1):15e21. 6. Davis Andrew. Schistosomiasis. Manson’s tropical diseases. 22nd ed. Saunders Elsevier; 2009. 7. da Silva IM, Tsang V, Noh J, Rey L, Conceisao MH. Clinical and laboratorial evaluation of urinary schistosomiasis in Brazilians after staying in Mozambique. Rev Soc Bras Med Trop 2006; 39(3):272e4. 8. Jones ME, Mitchell RG, Leen CL. Long seronegative window in schistosoma infection. Lancet 1992;340(8834e8835):1549e50. 9. Enk MJ, Amaral GL, Costa e Silva MF, Silveira-Lemos D, Teixeira-Carvalho A, Martins-Filho OA, et al. Rural tourism: a risk factor for schistosomiasis transmission in Brazil. Mem Inst Oswaldo Cruz 2010;105(4):537e40. 10. Danso-Appiah A, Garner P, Olliaro PL, Utzinger J. Treatment of urinary schistosomiasis: methodological issues and research needs identified through a Cochrane systematic review. Parasitology 2009;136(13):1837e49. 11. Keiser J, N’Guessan NA, Adoubryn KD, Silue KD, Vounatsou P, Hatz C, et al. Efficacy and safety of mefloquine, artesunate, mefloquine-artesunate, and praziquantel against Schistosoma haematobium: randomized, exploratory open-label trial. Clin Infect Dis 2000;50(9):1205e13. 12. da Silva IM, Thiengo R, Conceicao MH, Rey L, Lenzi HL, Filho PE, et al. Therapeutic failure of praziquantel in the treatment of Schistosoma haematobium infection in Brazilians returning from Africa. Mem Inst Oswaldo Cruz 2005;100(4):445e9. 13. da Silva IM, Thiengo R, Conceicao MH, Rey L, Pereira Filho E, Ribeiro PC. Cystoscopy in the diagnosis and follow-up of urinary schistosomiasis in Brazilian soldiers returning from Mozambique, Africa. Rev Inst Med Trop Sao Paulo 2006;48(1): 39e42.