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Orbito-facial dysmorphology in patients with different degrees of trigonocephaly severity: quantitative morpho-volumetric analysis in infants with non-syndromic metopic craniosynostosis

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Abstract

Purpose

Craniofacial dysmorphology varies significantly along a wide spectrum of severity in metopic cranial synostosis (MCS). This study aimed to quantify craniofacial changes, in MCS, to investigate their relationships with the severity of trigonocephaly.

Methods

By combining the metopic ridge and interfrontal angles, we identified three groups of trigonocephaly severity (mild group n.14, moderate group n.19, severe group n.18). We perform a quantitative analysis using high-resolution CT images evaluating (1) cranial fossae dimensions; (2) vault indices and ratios: interparietal/ intercoronal (IPD/ICD), interparietal/intertemporal (IPD/ITD), cephalic index, vertico-longitudinal index; (3) orbito-facial distances (midfacial depth, maxillary height, upper facial index, orbital distances, globe protrusions), maxilla and orbital volumes; (4) supratentorial (ICV) and infratentorial (PCFV) cranial volumes and supratentorial (WBV) and infratentorial (PCFBV) brain volumes.

Results

In all groups, middle skull base lengths and upper midface index were increased. In moderate and severe groups: anterior hemifossa lengths were reduced, IPD/ICD and vertico-longitudinal index were changed; midfacial depth, anterior, mild, and lateral interorbital distances were reduced; globe protrusions were increased. The comparison between moderate and severe groups showed an increase of both globe protrusions and IPD/ICD. Among all groups, ICV and WBV were reduced in the severe group.

Conclusion

This morpho-volumetric study provides new insights in understanding the craniofacial changes occurring in infants at different severity of trigonocephaly. The increase of globe protrusions and the reduction of supratentorial volumes found in the severe group reflect the severity of trigonocephaly; these findings might have a clinical and surgical relevance.

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Authors and Affiliations

  1. UOC Radiologia e Neuroradiologia - Polo Diagnostica per immagini, radioterapia, oncologia ed ematologia, Area diagnostica per immagini, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Largo Francesco Vito, 1, 00168, Rome, Italy

    Rosalinda Calandrelli, Antonio Marrazzo, Marco Panfili & Cesare Colosimo

  2. UOC Neurologia - Polo scienze dell’invecchiamento, neurologiche, ortopediche e della testa-collo, Area Neuroscienze, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168, Rome, Italy

    Fabio Pilato

  3. UOC Neurochirurgia Infantile - Polo scienze dell’invecchiamento, neurologiche, ortopediche e della testa-collo, Area Neuroscienze, Fondazione Policlinico Universitario Agostino Gemelli IRCC, 00168, Rome, Italy

    Luca Massimi

  4. Istituto di Neurochirurgia, Università Cattolica del Sacro Cuore, Rome, Italy

    Luca Massimi

  5. International Neuroscience Institute, Hannover, Germany

    Concezio Di Rocco

  6. Istituto di Radiologia, Università Cattolica del Sacro Cuore, Rome, Italy

    Cesare Colosimo

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  1. Rosalinda Calandrelli
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Correspondence to Rosalinda Calandrelli.

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Conflict of interest

Rosalinda Calandrelli declares that she has no conflict of interest.

Fabio Pilato declares that he has no conflict of interest.

Luca Massimi declares that he has no conflict of interest.

Antonio Marrazzo declares that he has no conflict of interest.

Marco Panfili declares that he has no conflict of interest.

Concezio Di Rocco declares that he has no conflict of interest.

Cesare Colosimo declares that he is a scientific consultant for Bracco Diagnostics Inc. and Bayer HealthCare.

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Calandrelli, R., Pilato, F., Massimi, L. et al. Orbito-facial dysmorphology in patients with different degrees of trigonocephaly severity: quantitative morpho-volumetric analysis in infants with non-syndromic metopic craniosynostosis. Childs Nerv Syst 36, 1263–1273 (2020). https://doi.org/10.1007/s00381-019-04456-x

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