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A critical appraisal of surgical outcomes following orbital hypertelorism correction: what is the incidence of true bony relapse versus soft tissue telecanthus?

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Abstract

Background

Orbital hypertelorism (OHT) represents a congenital condition defined by lateralization of the bony orbit, unlike soft tissue telecanthus in which there is an increase in intercanthal distance without true bony lateralization. Existing literature remains very limited in its postoperative assessment of bony versus soft tissue relapse, which may both clinically present as telecanthus. We performed a critical appraisal of the literature to determine the postoperative incidence of bony versus soft tissue relapse following OHT repair.

Methods

The PubMed, MEDLINE, EMBASE, Scopus, Cochrane Central Register of Controlled Trials, and clinicaltrials.org were searched systematically for all English studies published in any time frame reporting relapse rates following primary OHT repair. The primary outcome was incidence of bony and soft tissue relapse defined as orbital lateralization and medial canthal drift, respectively. The secondary outcome measures include postoperative complications, predictors of postoperative complications, timing and type of surgery, and revision rates.

Results

Eleven articles were included. A total of 84 (35.3%) patients experienced bony relapse while 43 (27.2%) patients experienced soft tissue relapse. Age at time of intervention (p < 0.92), severity at presentation (p < 0.90), and surgical technique (p < 0.09) were not found be significantly associated with relapse rate. Methods for long-term follow-up were not standardized, and there was no consistent measure to objectively assess telecanthus.

Conclusions

There is no general consensus on predictive factors of long-term relapse following OHT repair in the form of box osteotomy or facial bipartition. These findings call for cross-sectional outcome standardization to better understand long-term outcomes across institutional, provider, and patient differences.

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Data availability

The datasets generated during and/or analyzed during the current study are available from the corresponding author on reasonable request.

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All authors contributed to the study conception and design. Material preparation, data collection, and analysis were performed by Beatrice Go, Sameer Shakir, Jordan Swanson, Scott Bartlett, and Jesse Taylor. The first draft of the manuscript was written by Beatrice Go and Sameer Shakir and all authors commented on previous versions of the manuscript. Jordan Swanson, Scott Bartlett, and Jesse Taylor critically revised the work. All authors read and approved the final manuscript.

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Correspondence to Jesse A. Taylor.

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Go, B.C., Shakir, S., Swanson, J.W. et al. A critical appraisal of surgical outcomes following orbital hypertelorism correction: what is the incidence of true bony relapse versus soft tissue telecanthus?. Childs Nerv Syst 37, 21–32 (2021). https://doi.org/10.1007/s00381-020-04890-2

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