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Chronic polyarthritis as isolated manifestation of toxocariasis

ABSTRACT

Human toxocariasis is a parasitic zoonosis mainly caused by Toxocara canis or Toxocara catiand is acquired by ingestion of the parasite’s embryonated eggs. Arthralgia and/or arthri-tis were reported in up to 17% of the cases, generally with acute duration (less than 6weeks). However, to our knowledge, chronic polyarthritis, as the isolated presentation ofToxocara infection, was not reported. One of the 5809 patients that was followed up at ourservice (0.017%) had chronic polyarthritis as the single manifestation of toxocariasis and wasdescribed herein. A 3-year-old girl was referred to our service with severe painful chronicpolyarthritis for a period longer than 10 weeks and morning stiffness of 30 min. Dog contactexposure history in the recreational areas of neighborhood was reported. Her exams showedhigh levels of eosinophils in peripheral blood (29%), bone marrow aspirate revealed markedeosinophilia (32%) and Toxocara enzyme-linked immunosorbent assay (Elisa) was positive(1:1280). She was treated with paracetamol (40 mg/kg/day) and thiabendazole (25 mg/kg/day)for 10 days, and all manifestations reduced. After eight months of follow-up, she was onclinical and laboratorial remission. In conclusion, we described a case of chronic polyarthri-tis, as isolated manifestation of toxocariasis, mimicking juvenile idiopathic arthritis andleukemia. Importantly, this zoonosis should be considered in patients with arthritis andeosinophilia.

Keywords:
Systemic lupus erythematosus; Acute acalculous cholecystitis; Abdominal computed tomography; Abdominal computed tomographyInitial

RESUMO

A toxocaríase é uma zoonose parasitária causada principalmente pelo Toxocara canis ou peloT. cati. É adquirida pela ingestão de ovos embrionados do parasita. A artralgia e/ou artriteforam relatadas em até 17% dos casos, geralmente com duração aguda (menos de seis sema-nas). No entanto, que se tem conhecimento, a poliartrite crônica como manifestação isoladada infecção por Toxocara ainda não foi descrita na literatura. Um dos 5.809 pacientes acom-panhados em nosso serviço (0,017%) exibiu poliartrite crônica como manifestação únicada toxocaríase e foi descrito neste estudo. Uma menina de três anos foi encaminhada aonosso serviço com poliartrite crônica dolorosa grave por um período superior a 10 semanase rigidez matinal diária de 30 minutos. Foi relatada história de exposição a contato comcão nas áreas de lazer do bairro. Seus exames revelaram níveis elevados de eosinófilos nosangue periférico (29%), o aspirado de medula óssea revelou eosinofilia acentuada (32%)e o ensaio imunoenzimático ligado a enzima (ELISA) para Toxocara foi positivo (1:1.280). Acriança foi tratada com paracetamol (40 mg/kg/dia) e tiabendazol (25 mg/kg/dia) durante10 dias e houve regressão de todas as manifestações. Depois de oito meses de seguimento,a pequena paciente estava em remissão clínica e laboratorial. Em conclusão, descreve-seum caso de poliartrite crônica como manifestação isolada da toxocaríase, que mimetizouuma artrite idiopática juvenil e leucemia. É importante ressaltar que essa zoonose deve serconsiderada em pacientes com artrite e eosinofilia.

Palavras-chave:
Artrite crônica; Eosinofilia; Toxocaríase; Crianças; Artrite idiopática juvenil; Leucemiar

Introduction

Human toxocariasis is a parasitic zoonosis caused mainly by Toxocara canis or Toxocara cati and is acquired by ingestion of the embryonated eggs of the parasite.1Wisniewska-Ligier M, Wozniakowska-Gesicka T, Sobolewska-Dryjanska J, Markiewicz–Jozwiak A, Wieczorek M. Analysis of the course and treatment of toxocariasis in children – a long term observation. Parasitol Res. 2012;110:2363-2371.,2Mazur-Melewska K, Mania A, Figlerowicz M, Kemnitz P, Sluzewski W, Michalak M. The influence of age on a clinical presentation of Toxocara spp. infection in children. Ann Agric Environ Med. 2012;19:233-236. The clinical forms are systemic (visceral larva migrans), localized (ocular and neurological), asymptomatic and covert.2Mazur-Melewska K, Mania A, Figlerowicz M, Kemnitz P, Sluzewski W, Michalak M. The influence of age on a clinical presentation of Toxocara spp. infection in children. Ann Agric Environ Med. 2012;19:233-236.-4Carvalho EA, Rocha RL. Toxocariasis: visceral larva migrans in children. J Pediatr. 2011;87:100-110.

Of note, the covert form presents non-specific symptoms, such as lymphadenopathy, dermatological disorders, asthma and joint manifestations.2Mazur-Melewska K, Mania A, Figlerowicz M, Kemnitz P, Sluzewski W, Michalak M. The influence of age on a clinical presentation of Toxocara spp. infection in children. Ann Agric Environ Med. 2012;19:233-236.-4Carvalho EA, Rocha RL. Toxocariasis: visceral larva migrans in children. J Pediatr. 2011;87:100-110. Arthralgia and/or arthritis were reported in up to 17% of the cases,2Mazur-Melewska K, Mania A, Figlerowicz M, Kemnitz P, Sluzewski W, Michalak M. The influence of age on a clinical presentation of Toxocara spp. infection in children. Ann Agric Environ Med. 2012;19:233-236. generally with acute duration (less than 6 weeks). In Brazil, the prevalence of toxocariasis in urban areas ranged from 3.6% to 24.7%.5Alderete JM, Jacob CM, Pastorino AC, Elefant GR, Castro AP, Fomin AB, et al. Prevalence of Toxocara infection in schoolchildren from the Butantã region, São Paulo, Brazil. Mem Inst Oswaldo Cruz. 2003;98:593-597.,6Chieffi PP, Ueda M, Camargo ED, de Souza AM, Guedes ML, Gerbi LJ, et al. Visceral larva migrans: a seroepidemiological survey in five municipalities of São Paulo State, Brazil. Rev Inst Med Trop São Paulo. 1990;32:204-210. However, to our knowledge, severe and painful chronic polyarthritis, as the isolated presentation of Toxocara infection, was not reported.

From January 1983 to November 2013, we revised the charts of patients followed up at the Pediatric Rheumatology Unit of the Children's Institute of Hospital das Clínicas da Faculdade de Medicina Universidade de São Paulo. Only one of the 5809 patients (0.017%) had chronic polyarthritis as the single manifestation of toxocariasis and was described herein.

Case report

A 3-year-old girl was referred to our service with chronic polyarthritis in knees, wrists, shoulders, elbows and hips, with painful joint effusions in knees and wrists. The total duration of polyarthritis was 10 weeks. At that moment, she refused to walk due to severe pain. She had 30 min of morning stiffness and loss of appetite. Dog contact history in the recreational areas of neighborhood was reported. Laboratory examinations showed hemoglobin 10.1 g/dL, hematocrit 32%, white blood cell (WBC) 17,800 mm3 (neutrophils 42%, lymphocytes 25%, eosinophils 29%, and monocytes 4%), platelets 464,000 mm3, erythrocyte sedimentation rate (ESR) 55 mm/1st hour, C-reactive protein (CRP) 42.8 mg/dL, and lactate dehydrogenase (LDH) 879 mg/dL (normal range 117-213). Serologic test for hepatitis A, B and C, human immunodeficiency virus (HIV), cytomegalovirus, rubella, Epstein-Barr virus, toxoplasmosis and antistreptolysin O were negative. Three consecutive stool examinations did not identify any parasite infestation. Immunoglobulin E was 272 µg/L (normal <60 µg/L). Her bone marrow aspirate revealed marked eosinophilia (32%; normal range 0.5-7%) without neoplastic cells. The rheumatoid factor also was negative, and ophthalmological examination was normal. Toxocara enzyme-linked immunosorbent assay (ELISA) was 1:1280. She was treated with paracetamol (40 mg/kg/day) and thiabendazole (25 mg/kg/day) for 10 days, and all manifestations reduced. After 2 months, the ESR was 14 mm/1st hour, CRP 0.93 mg/dL and WBC 8200 mm3 (neutrophils 35%, lymphocytes 53%, eosinophils 6% and monocytes 6%), platelets 224,000 mm3, and immunoglobulin E was 162 µg/L. After eight months of follow-up, she was on clinical and laboratorial remission.

Discussion

Chronic polyarthritis, mimicking neoplasia and juvenile idiopathic arthritis (JIA), as the main manifestation of toxocariasis, have rarely been observed in our tertiary University Hospital in the last 30 years. The presence of concomitant arthritis and eosinophilia suggests the diagnosis.

Toxocariasis is a very common parasitosis in Brazil, and its prevalence ranged from 3.6% to 24.7%.5Alderete JM, Jacob CM, Pastorino AC, Elefant GR, Castro AP, Fomin AB, et al. Prevalence of Toxocara infection in schoolchildren from the Butantã region, São Paulo, Brazil. Mem Inst Oswaldo Cruz. 2003;98:593-597.,6Chieffi PP, Ueda M, Camargo ED, de Souza AM, Guedes ML, Gerbi LJ, et al. Visceral larva migrans: a seroepidemiological survey in five municipalities of São Paulo State, Brazil. Rev Inst Med Trop São Paulo. 1990;32:204-210. This infection, as a cause of isolated eosinophilic arthritis, is very seldom seen in the literature.7Rayes AA, Lambertucci JR. Human toxocariasis as a possible cause of eosinophilic arthritis. Rheumatology (Oxford). 2001;40:109-110. Indeed, it was rarely described in arthritis of Henoch-Schönlein purpura.8Hamidou MA, Gueglio B, Cassagneau E, Trewick D, Grolleau JY. Henoch-Schönlein purpura associated with Toxocara canis infection. J Rheumatol. 1999;26:443-445.-10Bellanger AP, Bamoulid J, Millon L, Chalopin JM, Humbert P. Rheumatoid purpura associated with toxocariasis. Can Fam Physician. 2011;57:1413-1414. The diagnosis of toxocariasis is suspected in patients that presented household contact with dogs11Macpherson CN. The epidemiology and public health importance of toxocariasis: a zoonosis of global importance. Int J Parasitol. 2013;43:999-1008.,12Núñez CR, Martínez GD, Arteaga SY, Macotela MP, Montes PB, Durán NF. Prevalence and risk factors associated with Toxocara canis infection in children. Sci World J. 2013:572089 and it is confirmed by the presence of specific antibodies, detected by ELISA method, with sensitivity of 90-92% and specificity of 90-95%.1Wisniewska-Ligier M, Wozniakowska-Gesicka T, Sobolewska-Dryjanska J, Markiewicz–Jozwiak A, Wieczorek M. Analysis of the course and treatment of toxocariasis in children – a long term observation. Parasitol Res. 2012;110:2363-2371.,4Carvalho EA, Rocha RL. Toxocariasis: visceral larva migrans in children. J Pediatr. 2011;87:100-110. The high levels of eosinophils in peripheral blood was also observed in more than 60% of the patients,1Wisniewska-Ligier M, Wozniakowska-Gesicka T, Sobolewska-Dryjanska J, Markiewicz–Jozwiak A, Wieczorek M. Analysis of the course and treatment of toxocariasis in children – a long term observation. Parasitol Res. 2012;110:2363-2371.,4Carvalho EA, Rocha RL. Toxocariasis: visceral larva migrans in children. J Pediatr. 2011;87:100-110.,12Núñez CR, Martínez GD, Arteaga SY, Macotela MP, Montes PB, Durán NF. Prevalence and risk factors associated with Toxocara canis infection in children. Sci World J. 2013:572089 and IgE titers had high levels in children up to 3 years old,2Mazur-Melewska K, Mania A, Figlerowicz M, Kemnitz P, Sluzewski W, Michalak M. The influence of age on a clinical presentation of Toxocara spp. infection in children. Ann Agric Environ Med. 2012;19:233-236.,4Carvalho EA, Rocha RL. Toxocariasis: visceral larva migrans in children. J Pediatr. 2011;87:100-110. as observed herein.

Our patient had covert clinical form with chronic arthritis without other clinical manifestations.2Mazur-Melewska K, Mania A, Figlerowicz M, Kemnitz P, Sluzewski W, Michalak M. The influence of age on a clinical presentation of Toxocara spp. infection in children. Ann Agric Environ Med. 2012;19:233-236.-4Carvalho EA, Rocha RL. Toxocariasis: visceral larva migrans in children. J Pediatr. 2011;87:100-110. Indeed, lymphadenopathy, dermatological disorders, wheezing, arthralgia and acute arthritis may be associated with this form.2Mazur-Melewska K, Mania A, Figlerowicz M, Kemnitz P, Sluzewski W, Michalak M. The influence of age on a clinical presentation of Toxocara spp. infection in children. Ann Agric Environ Med. 2012;19:233-236.-4Carvalho EA, Rocha RL. Toxocariasis: visceral larva migrans in children. J Pediatr. 2011;87:100-110. However to our knowledge, chronic arthritis in children was not previously reported with this infection.

Importantly, the main differential diagnoses in our patient were acute lymphoblastic leukemia (ALL) and JIA. The presence of concomitant severe articular pain and high LDH levels, even without other systemic manifestations, indicate bone marrow aspiration to exclude this neoplasia, as in our case. Moreover, JIA is the most important cause of chronic arthritis with morning stiffness; however, the exclusion of infections is necessary to confirm this diagnosis.13Tamashiro MS, Aikawa NE, Campos LM, Cristofani LM, Odone-Filho V, Silva CA. Discrimination of acute lymphoblastic leukemia from systemic-onset juvenile idiopathic arthritis at disease onset. Clinics. 2011;66:1665-1669.

The treatment of toxocariasis is necessary for symptomatic patients to reduce the number of potentially migratory larvae.9Pawlowska-Kamieniak A, Mroczkowska-Juchkiewicz A, Papierkowski A. Henoch-Schönlein purpura and toxocariasis. Pol Merkuriusz Lek. 1998;4:217-218. The use of thiabendazole (25 mg/kg/day) for a short period therapy is indicated to covert forms, as in the present case.4Carvalho EA, Rocha RL. Toxocariasis: visceral larva migrans in children. J Pediatr. 2011;87:100-110.

In conclusion, we described a case of chronic polyarthritis, as isolated manifestation of toxocariasis, mimicking JIA and ALL. This zoonosis should be considered in patients with arthritis and eosinophilia.

  • Fundings
    This study was supported by Fundação de Amparo à Pesquisa do Estado de São Paulo (FAPESP - grants 2008/58238-4 to CAS and 2011/12471-2 to CAS), by Conselho Nacional do Desenvolvimento Científico e Tecnológico (CNPQ - grant 302724/2011-7 to CAS), by Federico Foundation to CAS and by Núcleo de Apoio à Pesquisa “Saúde da Criança e do Adolescente” da USP (NAP-CriAd).

REFERENCES

  • Wisniewska-Ligier M, Wozniakowska-Gesicka T, Sobolewska-Dryjanska J, Markiewicz–Jozwiak A, Wieczorek M. Analysis of the course and treatment of toxocariasis in children – a long term observation. Parasitol Res. 2012;110:2363-2371.
  • Mazur-Melewska K, Mania A, Figlerowicz M, Kemnitz P, Sluzewski W, Michalak M. The influence of age on a clinical presentation of Toxocara spp. infection in children. Ann Agric Environ Med. 2012;19:233-236.
  • Guilherme EV, Marchioro AA, Araujo SM, Falavigna DL, Adami C, Falavigna-Guilherme G, et al. Toxocariasis in children attending a public health service pneumology unit in Parana State, Brazil. Rev Inst Med Trop Sao Paulo. 2013;55:189-192.
  • Carvalho EA, Rocha RL. Toxocariasis: visceral larva migrans in children. J Pediatr. 2011;87:100-110.
  • Alderete JM, Jacob CM, Pastorino AC, Elefant GR, Castro AP, Fomin AB, et al. Prevalence of Toxocara infection in schoolchildren from the Butantã region, São Paulo, Brazil. Mem Inst Oswaldo Cruz. 2003;98:593-597.
  • Chieffi PP, Ueda M, Camargo ED, de Souza AM, Guedes ML, Gerbi LJ, et al. Visceral larva migrans: a seroepidemiological survey in five municipalities of São Paulo State, Brazil. Rev Inst Med Trop São Paulo. 1990;32:204-210.
  • Rayes AA, Lambertucci JR. Human toxocariasis as a possible cause of eosinophilic arthritis. Rheumatology (Oxford). 2001;40:109-110.
  • Hamidou MA, Gueglio B, Cassagneau E, Trewick D, Grolleau JY. Henoch-Schönlein purpura associated with Toxocara canis infection. J Rheumatol. 1999;26:443-445.
  • Pawlowska-Kamieniak A, Mroczkowska-Juchkiewicz A, Papierkowski A. Henoch-Schönlein purpura and toxocariasis. Pol Merkuriusz Lek. 1998;4:217-218.
  • Bellanger AP, Bamoulid J, Millon L, Chalopin JM, Humbert P. Rheumatoid purpura associated with toxocariasis. Can Fam Physician. 2011;57:1413-1414.
  • Macpherson CN. The epidemiology and public health importance of toxocariasis: a zoonosis of global importance. Int J Parasitol. 2013;43:999-1008.
  • Núñez CR, Martínez GD, Arteaga SY, Macotela MP, Montes PB, Durán NF. Prevalence and risk factors associated with Toxocara canis infection in children. Sci World J. 2013:572089
  • Tamashiro MS, Aikawa NE, Campos LM, Cristofani LM, Odone-Filho V, Silva CA. Discrimination of acute lymphoblastic leukemia from systemic-onset juvenile idiopathic arthritis at disease onset. Clinics. 2011;66:1665-1669.

Publication Dates

  • Publication in this collection
    Mar-Apr 2016

History

  • Received
    20 Dec 2013
  • Accepted
    28 July 2014
Sociedade Brasileira de Reumatologia Av Brigadeiro Luiz Antonio, 2466 - Cj 93., 01402-000 São Paulo - SP, Tel./Fax: 55 11 3289 7165 - São Paulo - SP - Brazil
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