Literature DB >> 29797037

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

Andy Tsai1, Tal Laor2, Judy A Estroff2,3, James R Kasser3,4.   

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.

Entities:  

Keywords:  Constant inhibition; Fetus; Infants; Limb-length discrepancy; Lower extremity; Prenatal multiplier method; Radiography; Ultrasound

Mesh:

Year:  2018        PMID: 29797037     DOI: 10.1007/s00247-018-4153-5

Source DB:  PubMed          Journal:  Pediatr Radiol        ISSN: 0301-0449


  32 in total

1.  Prenatal ultrasonographic diagnosis of posteromedial bowing of the leg: two case reports.

Authors:  P E Zollinger; M W Wessels; J W Wladimiroff; A F Diepstraten
Journal:  Ultrasound Obstet Gynecol       Date:  2000-02       Impact factor: 7.299

2.  Multiplier method for predicting limb-length discrepancy.

Authors:  D Paley; A Bhave; J E Herzenberg; J R Bowen
Journal:  J Bone Joint Surg Am       Date:  2000-10       Impact factor: 5.284

Review 3.  Prenatal diagnosis of proximal focal femoral deficiency: Literature review of prenatal sonographic findings.

Authors:  Valentina D'Ambrosio; Gaia Pasquali; Antonia Squarcella; Eleonora Marcoccia; Angela De Filippis; Silvia Gatto; Aliberti Camilla; Antonio Pizzuti; Renato La Torre; Antonella Giancotti
Journal:  J Clin Ultrasound       Date:  2015-09-26       Impact factor: 0.910

4.  Prenatal diagnosis of unilateral tibial hemimelia.

Authors:  M Dreyfus; J J Baldauf; E Rigaut; J M Clavert; B Gasser; J Ritter
Journal:  Ultrasound Obstet Gynecol       Date:  1996-03       Impact factor: 7.299

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Authors:  Katherine L Grantz; Mary L Hediger; Danping Liu; Germaine M Buck Louis
Journal:  Am J Obstet Gynecol       Date:  2017-12-22       Impact factor: 8.661

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Journal:  J Med Genet       Date:  1974-09       Impact factor: 6.318

Review 7.  Major limb malformations following intrauterine exposure to ethanol: two additional cases and literature review.

Authors:  R M Pauli; P F Feldman
Journal:  Teratology       Date:  1986-06

8.  De novo 21q interstitial deletion in a retarded boy with ulno-fibular dysostosis.

Authors:  J F Reynolds; H E Wyandt; T E Kelly
Journal:  Am J Med Genet       Date:  1985-01

9.  Congenital posteromedial bowing of the tibia and fibula.

Authors:  A M Pappas
Journal:  J Pediatr Orthop       Date:  1984-09       Impact factor: 2.324

10.  Congenital longitudinal deficiency of the fibula: follow-up of treatment by Syme amputation.

Authors:  G W Westin; D N Sakai; W L Wood
Journal:  J Bone Joint Surg Am       Date:  1976-06       Impact factor: 5.284

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