Table Info

Table 2.

Joint and musculoskeletal manifestations in CZS

Study	Demographics	Findings
Magalhães et al. 2023 [16] Retrospective cohort study	• Cohort of 29 CZS cases • 5-year follow-up • Age > 24 months • Females (n = 16), males (n = 13)	• Microcephaly (100%, n = 29) • Spastic quadriparesis (93.1%, n = 27) • Spastic hemiparetic (6.9%, n = 2) • Arthrogryposis (3.4%, n = 1) • Hip abnormal acetabular index under 2 years of age • High hip femoral head migration and subluxation over 2 years of age
da Fonseca et al. 2022 [17] Prospective cohort study	• Cohort of 30 CZS cases • 2 pelvic radiographs performed within 12 months • Age range from 19 months to 43 months • Females (n = 11), males (n = 19)	• Microcephaly (87%, n = 26) • Optic nerve abnormalities and chorioretinal (53%, n = 30) • Spasticity, hip functional limitation, hip deformity in flexion, adduction, and medial rotation bilaterally (83%, n = 25) • Hip displacement (total 47%, n = 14; right hip 43%, n = 13, left hip 30%, n = 9) • Hip dislocation (20%, n = 6) associated with spasticity (P = 0.003; OR, 15.9) and ophthalmologic abnormalities (P = 0.016; OR, 16.9)
Matos et al. 2021 [18] Cross-sectional case series study	• Cohort of 47 CZS newborns • Females (n = 24), males (n = 23)	Propose a classification scheme for orthopedic deformities and musculoskeletal complications by incidence, severity, and progression (non-spastic, spastic, arthrogrypotic) • Foot deformities: vertical talus, clubfoot (41.2%, n = 19) • Hip deformity (29.8%, n = 14); hip adduction contracture (20.2%), hip displacement (7.5%) • Knee deformity (12.8%, n = 6); knee displacement (6.4%) • Hypertonia (72.3%, n = 34) • Spasticity (77.3%, n = 36) • Arthrogryposis (15.2%, n = 7) associated with spasticity and more severe deformities • Hip and knee dislocation (29.8%, n = 14; 6.4%, n = 3)
Contreras-Capetillo et al. 2021 [19] Case report	• Stillborn 35-week gestation • Sex unknown	• Zika viral RNA found in CSF • Craniofacial disproportion • Microcephaly • Irregular anterior and lower posterior hairline • Internal rotation and limited abduction of the shoulder • Limited hip abduction, knee extension, and flexion of both feet • Arthrogryposis • Absence of distal part of the right arm
Tavares et al. 2021 [20] Cross-sectional case series study	• Cohort of 96 CZS cases • Age range 25 months to 40 months • Females (n = 41), males (n = 55)	• Positive pull to sit maneuver (45.8%, n = 44) • Positive in ventral suspension (32.3%, n = 31) • Positive in shoulder suspension (30.2%, n = 29) • Positive in scarf sign (32.3%, n = 31) • Hypertonia of appendicular muscles (94.8%, n = 91) • Quadriparesis (91.7%, n = 88), diparesis (3.1%, n = 3), hemiparesis (1.0%, n = 1) • Severe motor impairments, could be result of brain damage
van der Linden et al. 2020 [21] Cross-sectional prospective study	• Cohort of 21 CZS cases • Age range 16 months to 30 months • Females (n = 14), males (n = 7)	Developmental delays in: • Total head control (14.3%, n = 3), partial head control (47.6%, n = 10) • Moro reflex (47.6%, n = 10), asymmetric tonic neck response (71.4%, n = 15), palmomandibular reflex (66.6%, n = 14), plantar grasp response (52.0%, n = 11), palmar grasp (76.2%, n = 16) • “125” posture of fingers (81.0%, n = 17), oromandibular dystonia (42.9%, n = 9), extensor axial hypertonia (38.1%, n = 8), “Swan neck” posture of fingers (18.8, n = 3), internal rotation of the shoulder (9.5%, n = 2) • Arthrogryposis (19.0%, n = 4)

Study

Demographics

Findings

Magalhães et al. 2023 [16]

Retrospective cohort study

• Cohort of 29 CZS cases

• 5-year follow-up

• Age > 24 months

• Females (n = 16), males (n = 13)

• Microcephaly (100%, n = 29)

• Spastic quadriparesis (93.1%, n = 27)

• Spastic hemiparetic (6.9%, n = 2)

• Arthrogryposis (3.4%, n = 1)

• Hip abnormal acetabular index under 2 years of age

• High hip femoral head migration and subluxation over 2 years of age

da Fonseca et al. 2022 [17]

Prospective cohort study

• Cohort of 30 CZS cases

• 2 pelvic radiographs performed within 12 months

• Age range from 19 months to 43 months

• Females (n = 11), males (n = 19)

• Microcephaly (87%, n = 26)

• Optic nerve abnormalities and chorioretinal (53%, n = 30)

• Spasticity, hip functional limitation, hip deformity in flexion, adduction, and medial rotation bilaterally (83%, n = 25)

• Hip displacement (total 47%, n = 14; right hip 43%, n = 13, left hip 30%, n = 9)

• Hip dislocation (20%, n = 6) associated with spasticity (P = 0.003; OR, 15.9) and ophthalmologic abnormalities (P = 0.016; OR, 16.9)

Matos et al. 2021 [18]

Cross-sectional case series study

• Cohort of 47 CZS newborns

• Females (n = 24), males (n = 23)

Propose a classification scheme for orthopedic deformities and musculoskeletal complications by incidence, severity, and progression (non-spastic, spastic, arthrogrypotic)

• Foot deformities: vertical talus, clubfoot (41.2%, n = 19)

• Hip deformity (29.8%, n = 14); hip adduction contracture (20.2%), hip displacement (7.5%)

• Knee deformity (12.8%, n = 6); knee displacement (6.4%)

• Hypertonia (72.3%, n = 34)

• Spasticity (77.3%, n = 36)

• Arthrogryposis (15.2%, n = 7) associated with spasticity and more severe deformities

• Hip and knee dislocation (29.8%, n = 14; 6.4%, n = 3)

Contreras-Capetillo et al. 2021 [19]

Case report

• Stillborn 35-week gestation

• Sex unknown

• Zika viral RNA found in CSF

• Craniofacial disproportion

• Microcephaly

• Irregular anterior and lower posterior hairline

• Internal rotation and limited abduction of the shoulder

• Limited hip abduction, knee extension, and flexion of both feet

• Arthrogryposis

• Absence of distal part of the right arm

Tavares et al. 2021 [20]

Cross-sectional case series study

• Cohort of 96 CZS cases

• Age range 25 months to 40 months

• Females (n = 41), males (n = 55)

• Positive pull to sit maneuver (45.8%, n = 44)

• Positive in ventral suspension (32.3%, n = 31)

• Positive in shoulder suspension (30.2%, n = 29)

• Positive in scarf sign (32.3%, n = 31)

• Hypertonia of appendicular muscles (94.8%, n = 91)

• Quadriparesis (91.7%, n = 88), diparesis (3.1%, n = 3), hemiparesis (1.0%, n = 1)

• Severe motor impairments, could be result of brain damage

van der Linden et al. 2020 [21]

Cross-sectional prospective study

• Cohort of 21 CZS cases

• Age range 16 months to 30 months

• Females (n = 14), males (n = 7)

Developmental delays in:

• Total head control (14.3%, n = 3), partial head control (47.6%, n = 10)

• Moro reflex (47.6%, n = 10), asymmetric tonic neck response (71.4%, n = 15), palmomandibular reflex (66.6%, n = 14), plantar grasp response (52.0%, n = 11), palmar grasp (76.2%, n = 16)

• “125” posture of fingers (81.0%, n = 17), oromandibular dystonia (42.9%, n = 9), extensor axial hypertonia (38.1%, n = 8), “Swan neck” posture of fingers (18.8, n = 3), internal rotation of the shoulder (9.5%, n = 2)

• Arthrogryposis (19.0%, n = 4)

OR: odds ratio

Declarations

Acknowledgments

We are grateful for the support of Dr. Branka Filipovic, head of research, at the Saint James School of Medicine, for her early input in designing this study. We thank Mr. Oscar Andrade, chief librarian, at the Saint James School of Medicine, as well as the Kean University Library Nancy Thompson Learning Commons for assistance with accessing the full-text, peer-reviewed articles that were needed to conduct this review.

Author contributions

Dhaara S and Dhairavi S: Conceptualization, Methodology, Investigation, Writing—original draft, Writing—review & editing. OM: Conceptualization, Methodology, Investigation, Writing—original draft. RZ: Conceptualization, Methodology, Investigation, Writing—review & editing.

Conflicts of interest

The authors declare that they have no conflicts of interest.

Ethical approval

Not applicable.

Consent to participate

Not applicable.

Consent to publication

Not applicable.

Availability of data and materials

The data and materials obtained for this systematic review were obtained online using search engines and the research databases mentioned in the methodology. The articles referenced in this paper can be accessed freely by searching for them on PubMed, PMC, Ovid, Cochrane, or Google Scholar.

Funding

This research was supported by Kean University Office of Research and Sponsored Programs (ORSP). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.

Copyright

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