Craniofacial and oral manifestations in CZS

StudyDemographicsFindings

Díaz et al. 2023 [27]

Cross-sectional case-control study

• Cohort of 14 CZS cases

• Matched 12 healthy controls

• Age range from 3 years to 5 years

• Females (n = 9), males (n = 5)

• Microcephaly (92.9%, n = 13)

• Dolichocephaly (71.4%, n = 10)

• Mesocephalic (21.4%, n = 3)

• Protruding maxilla (78.6%, n = 11)

• Retrognathic mandible (64.3%, n = 9)

• Asymmetric pupillary line (64.3%, n = 9)

• Open bite (21.4%, n = 3)

• Bruxism (85.7%, n = 12)

• CZS microcephaly, maxillary prognathism, altered facial thirds, asymmetric pupillary line, bruxism (P = 0.006), deep and anterior open bite, and distal step decidual molar relationship (P = 0.031)

da Costa et al. 2022 [28]

Cross-sectional case series study

• Cohort of 10 children with CZS

• Age range from 3 years to 5 years

• Females (n = 3), males (n = 7)

• Microcephaly (90%, n = 9)

• Cerebral palsy (100%, n = 10)

• Oligodontia (20%, n = 2)

• 3-year-old male had 12 primary dental absences, was microcephalic, had cerebral palsy, visual impairment, seizures, and hearing loss

• 5-year-old male had 15 primary dental absences, had loss of bone density in maxilla and mandible, cerebral palsy, foot syndactyly, and ventriculomegaly

Gomes et al. 2022 [29]

Cross-sectional case-control study

• 62 cases of microcephaly

CZS (n = 49), cytomegalovirus (CMV) (n = 4), toxoplasmosis (n = 1), other congenital infections (n = 8)

• Age range from 7 months to 35 months

• Females (n = 30), males (n = 32)

• Delayed tooth eruption (94%, n = 46) in CZS

• Altered sequence of tooth eruption (72%, n = 33) in CZS

• No statistically significant difference between CZS and other congenital infections with regards to delayed tooth eruption, alterations in sequence of tooth eruption of primary teeth, or developmental defects of enamel (opacity and hypoplasia)

de Oliveira et al. 2020 [30]

Cross-sectional case-control study

• Cohort of 45 CZS cases

• 50 healthy controls

• Mean age of 17 months

• Females (n = 24), males (n = 21)

CZS is more likely to have:

• Low weight

• Nonexclusive breastfeeding (provision of food in addition to breast milk before age 6 months)

• Mouth breathing

• Swallowing difficulty

• Excessive salivation

• Hypoplasia of the middle third of the face (17.8%, n = 8)

• Short lingual frenulum (2.2%, n = 1)

• Abnormal insertion of upper labial frenulum (42.2%, n = 19)

• Ogival palate (44.4%, n = 20)

• Developmental defects of enamel (35.6%, n = 16)

• Delayed tooth eruption delay (17.8%, n = 8)

Carvalho et al. 2019 [31]

Cross-sectional case-control study

• Cohort of 30 children with CZS and microcephaly evaluated over 24 consecutive months

• 30 healthy controls

• Females (n = 14), males (n = 16)

• At the end of the study, 18 were younger than 25 months and 12 children were 25 months old or older

• Delayed first dental eruption is considered after 9 months of age

• Delayed eruption of first tooth until after 9 months of age (60%, n = 18, 26.6 times more prevalent than in controls, P < 0.001, mean age of first dental eruption 10.8 months ± 3.8 months)

• Alveolar ridge hyperplasia (3.3%, n = 1)

• Short labial or lingual frenum (60%, n = 18, 4.9 times more prevalent than in controls, P = 0.028)

• Inadequate lingual posture at rest (30%, n = 9, 26.9 times)

• Micrognathia (6.7%, n = 2, P = 0.002)

• Narrow palatine vaults (33.3%, n = 10, 24.8 times more prevalent than in controls, P = 0.01)