Systemic Manifestations, Tooth Eruption and Enamel Defects in Children with Congenital Zika Virus Syndrome: 36-Month Follow-up Case Series

Alidianne Fábia Cabral  Cavalcanti; Taynná Dantas  de Arruda; Yeska Paola Costa  Aguiar; Adriana Suely de Oliveira  Melo; Jeime Iara Bezerra de Freitas  Leal; Emna Hidoussi Sakly; Myroslav  Goncharuk-Khomyn; Sérgio D’Avila Lins Bezerra  Cavalcanti; Alessandro Leite Cavalcanti

Authors

Alidianne Fábia Cabral Cavalcanti
Taynná Dantas de Arruda
Yeska Paola Costa Aguiar
Adriana Suely de Oliveira Melo
Jeime Iara Bezerra de Freitas Leal
Emna Hidoussi Sakly
Myroslav Goncharuk-Khomyn
Sérgio D’Avila Lins Bezerra Cavalcanti
Alessandro Leite Cavalcanti

Keywords:

Arbovirus Infection, Craniofacial Abnormalities, Microcephaly, Tooth Eruption, Dental Enamel

Abstract

Objective: To describe systemic manifestations, the characteristics related to tooth eruption, and the occurrence of enamel defects in children with Congenital Zika Virus Syndrome (CZS). Material and Methods: Prospective case series based on nine children with confirmed CZS diagnosis assisted at a reference center in a municipality in the Northeast Region of Brazil. Through a structured interview directed to mothers, information related to prenatal, delivery, and postpartum periods was collected. Tooth eruption was monitored through clinical examinations for 36 months. The modified developmental defect of enamel index (DDE) was used to identify opacities and hypoplasia. Data were presented using descriptive statistics. Results: A high proportion (77.8%) had microcephaly, and 55.5% had low birth weight. Musculoskeletal disorders, swallowing difficulty, and self-injury practices were present in all children. Among the systemic findings, visual impairment (77.8%) and seizures (77.8%) were widely reported. Concerning disorders related to the stomatognathic system, bruxism (66.7%) and difficulty in sucking (33.3%) were present. For most children (77.8%), the deciduous right lower central incisor was the first tooth to erupt (minimum 8 months and maximum 17 months). Enamel defects were diagnosed in only two children (22.2%). Conclusion: A wide range of systemic manifestations was observed in children with CZS, including visual impairment and musculoskeletal disorders. Delayed eruption of the first deciduous tooth was also observed. Enamel defects were present in a small proportion of children.

References

Oliveira Melo AS, Malinger G, Ximenes R, Szejnfeld PO, Alves Sampaio S, Bispo de Filippis AM. Zika virus intrauterine infection causes fetal brain abnormality and microcephaly: tip of the iceberg? Ultrasound Obstet Gynecol 2016; 47(1):6-7. https://doi.org/10.1002/uog.15831

Aguiar RS, Pohl F, Morais GL, Nogueira FCS, Carvalho JB, Guida L, et al. Molecular alterations in the extracellular matrix in the brains of newborns with congenital Zika syndrome. Sci Signal 2020; 13(635):eaay6736. https://doi.org/10.1126/scisignal.aay6736

Ticconi C, Rezza G. Defining Zika virus infection in pregnant women. Pathog Glob Health 2019; 113(7):290.

Del Campo M, Feitosa IM, Ribeiro EM, Horovitz DD, Pessoa AL, França GV, et al. The phenotypic spectrum of congenital Zika syndrome. Am J Med Genet A 2017; 173(4):841-57. https://doi.org/10.1002/ajmg.a.38170

Duarte JS, Santos LOF, Sette GCS, Santos TFC, Alves FAP, Coriolano-Marinus MWL. Children's needs with congenital syndrome related to Zika virus in the domiciliary context. Cad Saúde Colet 2019; 27(3):249-56. https://doi.org/10.1590/1414-462X201900030237

Cavalcanti AL. Challenges of dental care for children with microcephaly carrying Zika congenital syndrome. Contemp Clin Dent 2017; 8(3):345-6.

Aguiar YPC, Cavalcanti AFC, Alencar CRB, Melo ASO, Cavalcanti SDLB, Cavalcanti Al. Chronology of the first deciduous tooth eruption in Brazilian children with microcephaly associated with Zika virus: a longitudinal study. Pesqui Bras Odontopediatria Clín Integr 2018; 18(1):e3982. https://doi.org/10.4034/PBOCI.2018.181.16

Silva MCPMD, Arnaud MA, Lyra MCA, Alencar Filho AV, Rocha MÂW, Ramos RCF, et al. Dental development in children born to zikv-infected mothers: a case-based study. Arch Oral Biol 2020; 110:104598. https://doi.org/10.1016/j.archoralbio.2019.104598

Medina DT, Santos APPD, Rodrigues FMDF, Oliveira BH. Oral manifestations of congenital Zika virus infection in children with microcephaly: 18-month follow-up case series. Spec Care Dentist 2021; 2021. Nov 22. https://doi.org/10.1111/scd.12681

Cavalcanti AFC, Aguiar YPC, Arruda TD, Melo ASO, Cavalcanti AL, d’Ávila S. Quality of life of mothers of Brazilian children with congenital Zika virus syndrome and associated factors. Pesqui Bras Odontopediatria Clin Integr 2021; 21:e0071. https://doi.org/10.1590/pboci.2021.120

World Health Organization (WHO). Recommended definitions, terminology and format for statistical tables related to the perinatal period and use of a new certificate for cause of perinatal deaths. Modifications recommended by FIGO as amended October 14, 1976. Acta Obstet Gynecol Scand 1977; 56(3):247-53.

Brasil. Secretaria de Atenção a Saúde. Departamento de Ações Programáticas e Estratégicas. Protocolo de Atenção à Saúde e Resposta à Ocorrência de Microcefalia Relacionada à Infecção Pelo Vírus Zika. Plano Nacional de Enfrentamento à Microcefalia. Brasília: Ministério da Saúde; 2016, 46p. [In Portuguese].

Walter LRF, Ferelle A, Issao M. Odontologia Para o Bebê: Odontopediatria do Nascimento aos Três Anos. São Paulo: Artes Médicas; 1996. 246p. [In Portuguese].

Lunt RC, Law DB. A review of the chronology of eruption of deciduous teeth. J Am Dent Assoc 1974; 89(4):872-79.

Federation Dentaire Internationale (FDI). Comission of oral health research and epidemiology. A review of the developmental defects of enamel of dental index (DDE index). Int Dent J 1992; 42(6):411-26.

Torres-Duque CA, Patino CM, Ferreira JC. Case series: an essential study design to build knowledge and pose hypotheses for rare and new diseases. J Bras Pneumol 2020; 46(4):e20200389. https://doi.org/10.36416/1806-3756/e20200389

Campos GS, Bandeira AC, Sardi SI. Zika virus outbreak, Bahia, Brazil. Emerg Infect Dis 2015; 21(10):1885-86.

Brasil. Portaria nº 1.813, de 11 de novembro de 2015. Declara Emergência em Saúde Pública de importância Nacional (ESPIN) por alteração do padrão de ocorrência de microcefalias no Brasil. Available from: http://bvsms.saude.gov.br/bvs/saudelegis/gm/2015/prt1813_11_11_2015.html. [Access on June 22, 2019]. [In Portuguese].

World Health Organization (WHO). WHO statement on the third meeting of the International Health Regulations (2005) (IHR(2005)) Emergency committee on zika virus and observed increase in neurological disorders and neonatal malformations. Available from: https://www.who.int/news-room/detail/14-06-2016-who-statement-on-the-third-meeting-of-the-international-health-regulations-(2005)-(ihr(2005))-emergency-committee-on-zika-virus-and-observed-increase-in-neurological-disorders-and-neonatal-malformations. [Accessed on June 29, 2020].

Brasil. Ministério da Saúde. Secretaria de Vigilância em Saúde. Boletim Epidemiológico 04. Informe quinzenal sarampo – Brasil. Semanas epidemiológicas 43 de 2020 a 1 de 2021. Available from: https://www.gov.br/saude/pt-br/centrais-de-conteudo/publicacoes/boletins/boletins-epidemiologicos/edicoes/2021/boletim_epidemiologico_svs_4.pdf. [Access on August 11, 2021]. [In Portuguese].

Musso D, Ko AI, Baud D. Zika virus infection - after the pandemic. N Engl J Med 2019; 381(15):1444-57. https://doi.org/10.1056/NEJMra1808246

Aragão MFV, van der Linden V, Brainer-Lima AM, Coeli RR, Rocha MA, Silva PS, et al. Clinical features and neuroimaging (CT and MRI) findings in presumed Zika virus related congenital infection and microcephaly: retrospective case series study. BMJ 2016; 13:353. https://doi.org/10.1136/bmj.i1901

Fujimori K, Yamada M, Maekawa T, Yotani N, Tamura EI, Imadome KI, et al. A case of neonatal cytomegalovirus infection with severe thrombocytopenia that was sucessfully managed with empiric antiviral terapy. IDCases 2019; 19:e00675. https://doi.org/10.1016/j.idcr.2019.e00675

Gusmão TPL, Faria ABS, Leão Filho JC, Carvalho AAT, Gueiros LAM, Leão JC. Dental changes in children with congenital Zika syndrome. Oral Dis 2020; 26(2):457-64. https://doi.org/10.1111/odi.13238

Siqueira RMP, Santos MTBR, Cabral GMP. Alterations in the primary teeth of children with microcephaly in northeast Brazil: a comparative study. Int J Paediatr Dent 2018; 28(5):523-32. https://doi.org/10.1111/ipd.12402

Moura da Silva AA, Ganz JS, Sousa PD, Doriqui MJ, Ribeiro MR, Branco MD, et al. Early growth and neurologic outcomes of infants with probable congenital Zika virus syndrome. Emerg Infect Dis 2016; 22(11):1953-6. https://doi.org/10.3201/eid2211.160956

Santa Rita TH, Barra RB, Peixoto GP, Mesquita PG, Barra GB. Association between suspected zika virus disease during pregnancy and giving birth to a newborn with congenital microcephaly: a matched case-control study. BMC Res Notes 2017; 10(1):457. https://doi.org/10.1186/s13104-017-2796-1

Oliveira-Filho J, Felzemburgh R, Costa F, Nery N, Mattos A, Henriques DF, et al. Seizures as a complication of congenital Zika syndrome in early infancy. Am J Trop Med Hyg 2018; 98(6):1860-2. https://doi.org/10.4269/ajtmh.17-1020

Moura da Silva AA, Ganz JS, Sousa PD, Doriqui MJ, Ribeiro MR, Branco MD, et al. Early growth and neurologic outcomes of infants with probable congenital Zika virus syndrome. Emerg Infect Dis 2016; 22(11):1953-1956. doi: 10.3201/eid2211.160956

Ventura LO, Ventura CV, Dias NC, Vilar IG, Gois AL, Arantes TE, et al. Visual impairment evaluation in 119 children with congenital Zika syndrome. J AAPOS 2018; 22(3):218-222.e1. https://doi.org/10.1016/j.jaapos.2018.01.009

Leal MC, van der Linden V, Bezerra TP, de Valois L, Borges ACG, Antunes MMC, et al. Characteristics of dysphagia in infants with microcephaly caused by congenital Zika virus infection, Brazil, 2015. Emerg Infect Dis 2017; 23(8):1253-9. https://doi.org/10.3201/eid2308.170354

Carvalho IF, Alencar PNB, Carvalho de Andrade MD, Silva PGB, Carvalho EDF, Araújo LS, et al. Clinical and x-ray oral evaluation in patients with congenital Zika Virus. J Appl Oral Sci 2019; 27:e20180276. https://doi.org/10.1590/1678-7757-2018-0276

D'Agostino ÉS, Chagas JRLP, Cangussu MCT, Vianna MIP. Chronology and sequence of deciduous teeth eruption in children with microcephaly associated to the Zika vírus. Spec Care Dentist 2020; 40(1):3-9. https://doi.org/10.1111/scd.12435

Vaz FFS, da Silva Sobrinho AR, Athayde FRRS, de Vasconcelos Carvalho M, Sette-de-Souza PH, Ferreira SJ. Might Zika virus-associated microcephaly's severity impact deciduous tooth eruption and orofacial structures? Oral Dis 2022; 2022. Apr 29. https://doi.org/10.1111/odi.14227

Cavalcanti AFC, Aguiar YPC, de Oliveira Melo AS, de Freitas Leal JIB, Cavalcanti AL, Cavalcanti SDLB. Teething symptoms in children with congenital Zika syndrome: A 2-year follow-up. Int J Paediatr Dent 2019; 29(1):74-8. https://doi.org/10.1111/ipd.12431

Franzolin SOB, Pardini MIMC, Francischone LA, Deffune E, Consolaro A, et al. A. Explanation for the signs and symptoms of tooth eruption: mast cells. Dental Press J Orthod 2019; 24(2):20-31. https://doi.org/10.1590/2177-6709.24.2.020-031.oin

Massignan C, Cardoso M, Porporatti AL, Aydinoz S, Canto GL, Mezzomo LA, et al. Signs and symptoms of primary tooth eruption: a meta-analysis. Pediatrics 2016; 137(3):e20153501. https://doi.org/10.1542/peds.2015-3501