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Constant inhibition in congenital lower extremity shortening: does it begin in utero?

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Abstract

Background

Limb-length discrepancy (LLD) in children with congenital lower extremity shortening is constant in proportion from birth to skeletal maturity (known as constant inhibition), but its developmental pattern in utero is unknown. The popular prenatal multiplier method to predict LLD at birth assumes constant inhibition in utero to be true. Verifying the in utero developmental pattern of LLD, and thus confirming the validity of the prenatal multiplier method, is crucial for meaningful prenatal parental counseling.

Objective

To elucidate the in utero developmental pattern of LLD in fetuses with congenital lower extremity shortening.

Materials and methods

Clinical indications for 3,605 lower extremity radiographs performed on infants (<1 year old) at a large tertiary hospital over a 17-year period were reviewed. Inclusion criteria were (1) diagnosis of congenital lower extremity shortening, (2) bilateral lower limb postnatal radiographs documenting LLD and (3) fetal ultrasound (US) documenting LLD. Available measurements of femoral, tibial and fibular lengths on fetal US and postnatal radiographs were collected. Prenatal and postnatal length ratios of shorter-to-longer bones were calculated and compared.

Results

Eighteen infants met inclusion criteria. Diagnoses were proximal focal femoral deficiency=4, congenital short femur=2, tibial hemimelia=3, posteromedial tibial bowing=6 and fibular hemimelia=3. The correlations between postnatal and prenatal length ratios were high for the femur, tibia and fibula (R>0.98, P<0.0001). The relative differences in the postnatal and prenatal length ratios of these bones were small (|average|<0.026, standard deviation <0.068).

Conclusion

Our data indicate that the postnatal and prenatal length ratios were equivalent, supporting the constant inhibition pattern of LLD in utero, thus validating the prenatal multiplier method for predicting LLD.

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Authors and Affiliations

  1. Department of Radiology, Boston Children’s Hospital, 300 Longwood Ave., Boston, MA, 02115, USA

    Andy Tsai, Tal Laor & Judy A. Estroff

  2. Advanced Fetal Care Center, Boston Children’s Hospital, Boston, MA, USA

    Judy A. Estroff & James R. Kasser

  3. Department of Orthopedics, Boston Children’s Hospital, Boston, MA, USA

    James R. Kasser

Authors
  1. Andy Tsai
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  2. Tal Laor
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  3. Judy A. Estroff
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  4. James R. Kasser
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Correspondence to Andy Tsai.

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Tsai, A., Laor, T., Estroff, J.A. et al. Constant inhibition in congenital lower extremity shortening: does it begin in utero?. Pediatr Radiol 48, 1451–1462 (2018). https://doi.org/10.1007/s00247-018-4153-5

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  • DOI: https://doi.org/10.1007/s00247-018-4153-5

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