Bertiella studeri Infection—A Rare Cause of Chronic Abdominal Pain in a Child from North India

Bertiella studeri Infection—A Rare Cause of Chronic Abdominal Pain in a Child from North India Abstract Bertiella is a common parasite seen in non-human primates. It is rarely seen in humans. We present the case of a 2-year-old child with bertiellosis. He had recurrent abdominal pain, and worm fragments were found in stool, which were refractory to albendazole therapy. Bertiella, zoonosis, non-human primates INTRODUCTION Bertiella is a common cestode parasite of non-human primates, which are the definitive hosts. The adult worm resides in the intestines of monkeys and sheds proglottids loaded with eggs in the faeces. The intermediate host, oribatid mite, gets infected on ingesting the oncospheres and develops cysticercoids, which are again transmitted to monkeys when they eat infected mites. There are 29 known species of Bertiella, of which only 2 have been known to cause human infections—Bertiellastuderi (from Africa and Asia) and Bertiellamucronata (from South America and Cuba) [1]. The first human case was reported in 1913 from Mauritius, after which >50 cases have now been reported from across the globe, including India [2, 3]. Here, we present a case of bertiellosis in a 2-year-old child from North India. CASE REPORT A developmentally normal 2-year-old boy from Rampur in Shimla, Himachal Pradesh, symptomatic for past 1 year, presented with abdominal pain and distension along with continuous perianal itching. The abdominal pain was generalized, intermittent, dull aching, sometimes gripping, usually after feeds and relieved by heavy passage of worms in stool. He had a voracious appetite with preserved weight. His parents noticed behavioural changes in him, such as excessive irritability, restlessness, anger tantrums, aggressive behaviour and peer abuse. These behavioural changes can be a manifestation of continuous perianal itching and abdominal pain, which can be poorly described by a 2-year-old boy. No other systemic manifestations were noted. Residing in a rural, hilly area, adjoining forest, this child had close proximity to wild monkeys. For past 1 year, he had received albendazole multiple times with no symptomatic improvement or relief from passage of worms in stool. On investigation, he had a haemoglobin of 13.6 g/dl, total leucocyte count of 9600 cells/dl and serum IgE levels of 301 IU/ml, and ultrasonography of abdomen and ophthalmic examination were normal. In view of the persistent worm infestation, flow cytometric immunophenotyping was done for lymphocyte subsets to look for any underlying immunodeficiency, and T and B lymphocytes were found within normal range. The stool sample and worm segments were submitted for parasitological workup. On gross examination, the segments were cestode proglottids, yellowish-white in colour, wider (0.68–1.1 cm) than long (0.1 cm), showed active contractions and frequently changing shape. The microscopic examination of stool revealed oval to spherical ova, 46–65 µm in size, containing hexacanth embryo with typical pyriform apparatus characteristic of B. studeri (Fig. 1). No evidence of other parasitic or bacterial infection was found in the stool sample, and serology for cysticercosis, trichinellosis, toxocariasis and filariasis was also negative. The child was administered with two doses of praziquantel on successive days (dose—20 mg/kg/day) along with purgatives, as niclosamide was not available. The child was stable during the hospital stay with no further passage of worms and hence was discharged. After 2 weeks, three stool examinations were repeated, which showed no eggs or proglottids. The child is in regular follow-up on OPD basis. Even after 7 months of treatment, he is asymptomatic with no abdominal pain and perianal itching. His irritability and anger tantrums have also improved considerably. Fig. 1. View largeDownload slide Bertiella studeri proglottids in contraction (A) and expansion (B), and eggs with pyriform apparatus and oncosphere with hooklets from stool sample of patient in low (C) and high (D) magnification. Fig. 1. View largeDownload slide Bertiella studeri proglottids in contraction (A) and expansion (B), and eggs with pyriform apparatus and oncosphere with hooklets from stool sample of patient in low (C) and high (D) magnification. DISCUSSION Bertiellosis is a zoonotic disease, which may be acquired by close ecological contact with monkeys or other primates. With shrinking natural habitat of primates because of deforestation and urbanization, there has been an invasion of primates into human dwellings. Our patient revealed a contact history with monkeys, dogs and other animals. There is an increase in number of cases of bertiellosis from common dwelling areas of humans and monkeys [4]. Monkey feeding is also a common religious practice in India, thereby increasing the chance of contact with the parasite. Dogs have also been reported to be infected with B. studeri in the Philippines, thus expanding the potential zoonotic reservoirs [5]. Human infection is accidental and occurs mainly in infants or children who have a propensity of geophagia. Our patient was also likely to have ingested mite-infested soil because of his aggressive nature and habit of picking up anything. The infection may be asymptomatic or present with gastrointestinal disturbances, such as diarrhoea, recurrent abdominal pain, anorexia, weight loss, vomiting and constipation [5–7]. The diagnosis of bertiellosis is made by observing the morphological characteristics of the parasite elements in the stool sample [4, 6, 8]. Bertiella may be more prevalent than realized, and the cases may go under-reported because of apparent resemblance of segments to other tapeworms [9]. There is a lack of awareness amongst clinicians and microbiologists about the disease, which adds to the diagnostic dilemma and delay in management. The immature eggs from unripe proglottids have thin, flexible shells that deform easily and may look similar to vegetative artefacts and thus dismissed if not carefully observed under the microscope [3]. In our case also, the diagnosis of Bertiella was missed repeatedly on stool examination. Treatment of tape worms is traditionally done with praziquantel, which is well tolerated and has good efficacy. It acts by causing paralysis of the worm by acting on the membrane calcium channels. Niclosamide is the other drug that is used as a treatment for taeniasis and other tapeworms [10]. It is important to correctly identify Bertiella, a tape worm, as it is known to be resistant to common antihelminthic drugs. A complete resolution of symptoms can be achieved with praziquantel at a dose of 20 mg/kg/body weight for 2 successive days, as in our case [5]. CONCLUSION Bertiellosis is a rare zoonosis. It should be kept in the differential diagnosis in cases reporting recurrent tapeworm expulsion in faeces refractory to albendazole. A careful elicitation of the animal contact history may yield important clues to the diagnosis of zoonotic infections in many more ways than realized and remains a simple yet under used skill. As oribatid mites are natural components of the soil fauna with global distribution, a preventive programme or prophylactic attempt would be difficult and uneconomical to achieve [4]. Therefore, a timely diagnosis and appropriate case-based treatment will remain the mainstay for the management of bertiellosis because of its rarity and ubiquity of the definitive and intermediate hosts. ACKNOWLEDGEMENT The authors acknowledge the contributions of ‘Department of Parasitology, Postgraduate Institute of Medical Education and Research, Chandigarh, India’ for identifying this rare worm. References 1 Galán-Puchades MT , Fuentes MV , Mas-Coma S. Morphology of Bertiella studeri (Blanchard, 1891) sensu Stunkard (1940) (Cestoda: Anoplocephalidae) of human origin and a proposal of criteria for the specific diagnosis of bertiellosis . Folia Parasitol 2000 ; 47 : 23 – 8 . Google Scholar CrossRef Search ADS PubMed 2 Blanchard R. Bertiella satyri de l’Orang-outang, est aussi parasite de l’homme . Bull Acad Méd 1913 ; 9 : 286 – 96 . 3 Malik S , Srivastava VK , Samantaray JC. Human bertiellosis from North India . Indian J Pediatr 2013 ; 80 : 258 – 60 . Google Scholar CrossRef Search ADS PubMed 4 Paçô JM , Campos DM , Araújo JL. Human bertiellosis in Goiás, Brazil: a case report on human infection by Bertiella sp. (Cestoda: Anoplocephalidae) . Rev Inst Med Trop Sao Paulo 2003 ; 45 : 159 – 61 . Google Scholar CrossRef Search ADS PubMed 5 El-Dib NA , Al-Rufaii A , El-Badry AA , et al. Human infection with Bertiella species in Saudi Arabia . Saudi Pharm J 2004 ; 12 : 168 – 9 . 6 Sun X , Fang Q , Chen X-Z , et al. Bertiella studeri infection, China . Emerg Infect Dis 2006 ; 12 : 176 – 7 . Google Scholar CrossRef Search ADS PubMed 7 Xuan LT , Anantaphruti MT , Tuan PA , et al. The first human infection with Bertiella studeri in Vietnam . Southeast Asian J Trop Med Public Health 2003 ; 34 : 298 – 300 . Google Scholar PubMed 8 Lopes VV , dos Santos HA , Silva AV , et al. First case of human infection by Bertiella studeri (Blanchard, 1891) Stunkard,1940 (Cestoda; Anoplocephalidae) in Brazil . Rev Inst Med Trop Sao Paulo 2015 ; 57 : 447 – 50 . Google Scholar CrossRef Search ADS PubMed 9 Bhagwant S. Human Bertiella studeri (family Anoplocephalidae) infection of probable Southeast Asian origin in Mauritian children and an adult . Am J Trop Med Hyg 2004 ; 70 : 225 – 8 . Google Scholar PubMed 10 Shafaghi A , Rezayat KA , Mansour-Ghanaei F , et al. Taenia: an uninvited guest . Am J Case Rep 2015 ; 16 : 501 – 4 . Google Scholar CrossRef Search ADS PubMed © The Author [2017]. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Tropical Pediatrics Oxford University Press

Bertiella studeri Infection—A Rare Cause of Chronic Abdominal Pain in a Child from North India

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Oxford University Press
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© The Author [2017]. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com
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0142-6338
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Abstract

Abstract Bertiella is a common parasite seen in non-human primates. It is rarely seen in humans. We present the case of a 2-year-old child with bertiellosis. He had recurrent abdominal pain, and worm fragments were found in stool, which were refractory to albendazole therapy. Bertiella, zoonosis, non-human primates INTRODUCTION Bertiella is a common cestode parasite of non-human primates, which are the definitive hosts. The adult worm resides in the intestines of monkeys and sheds proglottids loaded with eggs in the faeces. The intermediate host, oribatid mite, gets infected on ingesting the oncospheres and develops cysticercoids, which are again transmitted to monkeys when they eat infected mites. There are 29 known species of Bertiella, of which only 2 have been known to cause human infections—Bertiellastuderi (from Africa and Asia) and Bertiellamucronata (from South America and Cuba) [1]. The first human case was reported in 1913 from Mauritius, after which >50 cases have now been reported from across the globe, including India [2, 3]. Here, we present a case of bertiellosis in a 2-year-old child from North India. CASE REPORT A developmentally normal 2-year-old boy from Rampur in Shimla, Himachal Pradesh, symptomatic for past 1 year, presented with abdominal pain and distension along with continuous perianal itching. The abdominal pain was generalized, intermittent, dull aching, sometimes gripping, usually after feeds and relieved by heavy passage of worms in stool. He had a voracious appetite with preserved weight. His parents noticed behavioural changes in him, such as excessive irritability, restlessness, anger tantrums, aggressive behaviour and peer abuse. These behavioural changes can be a manifestation of continuous perianal itching and abdominal pain, which can be poorly described by a 2-year-old boy. No other systemic manifestations were noted. Residing in a rural, hilly area, adjoining forest, this child had close proximity to wild monkeys. For past 1 year, he had received albendazole multiple times with no symptomatic improvement or relief from passage of worms in stool. On investigation, he had a haemoglobin of 13.6 g/dl, total leucocyte count of 9600 cells/dl and serum IgE levels of 301 IU/ml, and ultrasonography of abdomen and ophthalmic examination were normal. In view of the persistent worm infestation, flow cytometric immunophenotyping was done for lymphocyte subsets to look for any underlying immunodeficiency, and T and B lymphocytes were found within normal range. The stool sample and worm segments were submitted for parasitological workup. On gross examination, the segments were cestode proglottids, yellowish-white in colour, wider (0.68–1.1 cm) than long (0.1 cm), showed active contractions and frequently changing shape. The microscopic examination of stool revealed oval to spherical ova, 46–65 µm in size, containing hexacanth embryo with typical pyriform apparatus characteristic of B. studeri (Fig. 1). No evidence of other parasitic or bacterial infection was found in the stool sample, and serology for cysticercosis, trichinellosis, toxocariasis and filariasis was also negative. The child was administered with two doses of praziquantel on successive days (dose—20 mg/kg/day) along with purgatives, as niclosamide was not available. The child was stable during the hospital stay with no further passage of worms and hence was discharged. After 2 weeks, three stool examinations were repeated, which showed no eggs or proglottids. The child is in regular follow-up on OPD basis. Even after 7 months of treatment, he is asymptomatic with no abdominal pain and perianal itching. His irritability and anger tantrums have also improved considerably. Fig. 1. View largeDownload slide Bertiella studeri proglottids in contraction (A) and expansion (B), and eggs with pyriform apparatus and oncosphere with hooklets from stool sample of patient in low (C) and high (D) magnification. Fig. 1. View largeDownload slide Bertiella studeri proglottids in contraction (A) and expansion (B), and eggs with pyriform apparatus and oncosphere with hooklets from stool sample of patient in low (C) and high (D) magnification. DISCUSSION Bertiellosis is a zoonotic disease, which may be acquired by close ecological contact with monkeys or other primates. With shrinking natural habitat of primates because of deforestation and urbanization, there has been an invasion of primates into human dwellings. Our patient revealed a contact history with monkeys, dogs and other animals. There is an increase in number of cases of bertiellosis from common dwelling areas of humans and monkeys [4]. Monkey feeding is also a common religious practice in India, thereby increasing the chance of contact with the parasite. Dogs have also been reported to be infected with B. studeri in the Philippines, thus expanding the potential zoonotic reservoirs [5]. Human infection is accidental and occurs mainly in infants or children who have a propensity of geophagia. Our patient was also likely to have ingested mite-infested soil because of his aggressive nature and habit of picking up anything. The infection may be asymptomatic or present with gastrointestinal disturbances, such as diarrhoea, recurrent abdominal pain, anorexia, weight loss, vomiting and constipation [5–7]. The diagnosis of bertiellosis is made by observing the morphological characteristics of the parasite elements in the stool sample [4, 6, 8]. Bertiella may be more prevalent than realized, and the cases may go under-reported because of apparent resemblance of segments to other tapeworms [9]. There is a lack of awareness amongst clinicians and microbiologists about the disease, which adds to the diagnostic dilemma and delay in management. The immature eggs from unripe proglottids have thin, flexible shells that deform easily and may look similar to vegetative artefacts and thus dismissed if not carefully observed under the microscope [3]. In our case also, the diagnosis of Bertiella was missed repeatedly on stool examination. Treatment of tape worms is traditionally done with praziquantel, which is well tolerated and has good efficacy. It acts by causing paralysis of the worm by acting on the membrane calcium channels. Niclosamide is the other drug that is used as a treatment for taeniasis and other tapeworms [10]. It is important to correctly identify Bertiella, a tape worm, as it is known to be resistant to common antihelminthic drugs. A complete resolution of symptoms can be achieved with praziquantel at a dose of 20 mg/kg/body weight for 2 successive days, as in our case [5]. CONCLUSION Bertiellosis is a rare zoonosis. It should be kept in the differential diagnosis in cases reporting recurrent tapeworm expulsion in faeces refractory to albendazole. A careful elicitation of the animal contact history may yield important clues to the diagnosis of zoonotic infections in many more ways than realized and remains a simple yet under used skill. As oribatid mites are natural components of the soil fauna with global distribution, a preventive programme or prophylactic attempt would be difficult and uneconomical to achieve [4]. Therefore, a timely diagnosis and appropriate case-based treatment will remain the mainstay for the management of bertiellosis because of its rarity and ubiquity of the definitive and intermediate hosts. ACKNOWLEDGEMENT The authors acknowledge the contributions of ‘Department of Parasitology, Postgraduate Institute of Medical Education and Research, Chandigarh, India’ for identifying this rare worm. References 1 Galán-Puchades MT , Fuentes MV , Mas-Coma S. Morphology of Bertiella studeri (Blanchard, 1891) sensu Stunkard (1940) (Cestoda: Anoplocephalidae) of human origin and a proposal of criteria for the specific diagnosis of bertiellosis . Folia Parasitol 2000 ; 47 : 23 – 8 . Google Scholar CrossRef Search ADS PubMed 2 Blanchard R. Bertiella satyri de l’Orang-outang, est aussi parasite de l’homme . Bull Acad Méd 1913 ; 9 : 286 – 96 . 3 Malik S , Srivastava VK , Samantaray JC. Human bertiellosis from North India . Indian J Pediatr 2013 ; 80 : 258 – 60 . Google Scholar CrossRef Search ADS PubMed 4 Paçô JM , Campos DM , Araújo JL. Human bertiellosis in Goiás, Brazil: a case report on human infection by Bertiella sp. (Cestoda: Anoplocephalidae) . Rev Inst Med Trop Sao Paulo 2003 ; 45 : 159 – 61 . Google Scholar CrossRef Search ADS PubMed 5 El-Dib NA , Al-Rufaii A , El-Badry AA , et al. Human infection with Bertiella species in Saudi Arabia . Saudi Pharm J 2004 ; 12 : 168 – 9 . 6 Sun X , Fang Q , Chen X-Z , et al. Bertiella studeri infection, China . Emerg Infect Dis 2006 ; 12 : 176 – 7 . Google Scholar CrossRef Search ADS PubMed 7 Xuan LT , Anantaphruti MT , Tuan PA , et al. The first human infection with Bertiella studeri in Vietnam . Southeast Asian J Trop Med Public Health 2003 ; 34 : 298 – 300 . Google Scholar PubMed 8 Lopes VV , dos Santos HA , Silva AV , et al. First case of human infection by Bertiella studeri (Blanchard, 1891) Stunkard,1940 (Cestoda; Anoplocephalidae) in Brazil . Rev Inst Med Trop Sao Paulo 2015 ; 57 : 447 – 50 . Google Scholar CrossRef Search ADS PubMed 9 Bhagwant S. Human Bertiella studeri (family Anoplocephalidae) infection of probable Southeast Asian origin in Mauritian children and an adult . Am J Trop Med Hyg 2004 ; 70 : 225 – 8 . Google Scholar PubMed 10 Shafaghi A , Rezayat KA , Mansour-Ghanaei F , et al. Taenia: an uninvited guest . Am J Case Rep 2015 ; 16 : 501 – 4 . Google Scholar CrossRef Search ADS PubMed © The Author [2017]. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com

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Journal of Tropical PediatricsOxford University Press

Published: Oct 18, 2017

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