Abstract
Purpose
To investigate whether atrophy of the leg muscles present in congenital clubfoot (CCF) is primitive or secondary to treatment of the deformity.
Methods
Magnetic resonance imaging (MRI) of both legs was taken in three cohorts of patients with unilateral congenital clubfoot (UCCF): eight untreated newborns (age range 10 days to 2 weeks); eight children who had been treated with the Ponseti method (age range 2–4 years); eight adults whose deformity had been corrected by manipulation and casting according to Ponseti, followed by a limited posterior release performed at age 2–3 months (age range 19–23 years). All of the treated patients wore a brace until 3 years of age. Muscles were measured on transverse MRI scans of both legs taken midway between the articular surface of the knee and the articular surface of the ankle, using a computer program (AutoCAD 2002 LT). The same program was used to measure leg muscles in the histologic cross sections of the legs of two fetuses with UCCF, spontaneously aborted at 13 and 19 weeks of gestation, respectively. Measurements of the whole cross section of the leg (total leg volume: TLV), of the muscular tissue (muscular tissue volume: MTV), and of the adipose tissue (adipose tissue volume: ATV) of the tibia, fibula, and of the other soft tissues (tendons, nerves, and vessels) were taken by using an interactive image analyzer (IAS 2000, Delta System, Milan, Italy).
Results
Marked atrophy of the leg muscles on the clubfoot side was found in both fetuses and untreated newborns, with a percentage ratio of MTV between the normal and the affected leg of 1.3 and 1.5, respectively. Leg muscle atrophy increased with growth, and the percentage ratio of MTV between the normal and the affected leg was, respectively, 1.8 and 2 in treated children and adults. On the other hand, fatty tissue tended to increase relatively from birth to adulthood, but it could not compensate for the progressive muscular atrophy. As a result, the difference in TLV tended to increase from childhood to adulthood.
Conclusions
Our study shows that leg muscular atrophy is a primitive pathological component of CCF which is already present in the early stages of fetal CCF development and in newborns before starting treatment. Muscular atrophy increases with the patient’s age, suggesting a mechanism of muscle growth impairment as a possible pathogenic factor of CCF.
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References
Bechtol CO, Mossman HW (1950) Clubfoot; an embryological study of associated muscle abnormalities. J Bone Joint Surg Am 32:827–838
Flinchum D (1953) Pathological anatomy in talipes equinovarus. J Bone Joint Surg Am 35:111–114
Ippolito E, Ponseti IV (1980) Congenital club foot in the human fetus. A histological study. J Bone Joint Surg Am 62:8–22
Irani RN, Sherman MS (1963) The pathological anatomy of clubfoot. J Bone Joint Surg Am 45:45–52
Schlicht D (1963) The pathological anatomy of talipes equino-varus. Aust N Z J Surg 33:1–11
Waisbrod H (1973) Congenital club foot. An anatomical study. J Bone Joint Surg Br 55:796–801
Wiley AM (1959) Club foot: an anatomical and experimental study of muscle growth. J Bone Joint Surg Br 41:821–835
Laaveg SJ, Ponseti IV (1980) Long-term results of treatment of congenital club foot. J Bone Joint Surg Am 62:23–31
Hutchins PM, Foster BK, Paterson DC, Cole EA (1985) Long-term results of early surgical release in club feet. J Bone Joint Surg Br 67:791–799
Kránicz J, Than P, Kustos T (1998) Long-term results of the operative treatment of clubfoot: a representative study. Orthopedics 21:669–674
Ippolito E, Farsetti P, Caterini R, Tudisco C (2003) Long-term comparative results in patients with congenital clubfoot treated with two different protocols. J Bone Joint Surg Am 85:1286–1294
Pekindil G, Aktas S, Saridogan K, Pekindil Y (2001) Magnetic resonance imaging in follow-up of treated clubfoot during childhood. Eur J Radiol 37:123–129
Kamegaya M, Shinohara Y, Kuniyoshi K, Moriya H (2001) MRI study of talonavicular alignment in club foot. J Bone Joint Surg Br 83:726–730
Pirani S, Zeznik L, Hodges D (2001) Magnetic resonance imaging study of the congenital clubfoot treated with the Ponseti method. J Pediatr Orthop 21:719–726
Diméglio A, Bensahel H, Souchet P, Mazeau P, Bonnet F (1995) Classification of clubfoot. J Pediatr Orthop Br 4:129–136
Ponseti IV (1992) Treatment of congenital club foot. J Bone Joint Surg Am 74:448–454
Fisher RA (1956) Statistical methods and scientific inference. Oxford University Press, Oxford. Reprinted in 1990, Oliver and Boyd, Edinburgh
Aronson J, Puskarich CL (1990) Deformity and disability from treated clubfoot. J Pediatr Orthop 10:109–119
Gasser T (1996) Development of fat tissue and body mass index from infancy to adulthood. Pediatr Nephrol 10:340–342
Schmelzle HR, Fusch C (2002) Body fat in neonates and young infants: validation of skinfold thickness versus dual-energy X-ray absorptiometry. Am J Clin Nutr 76:1096–1100
Gosztonyi G, Dorfmüller-Küchlin S, Sparmann M, Eisenschenk A (1989) Morphometric study of muscle in congenital idiopathic club foot. Pathol Res Pract 185:790–794
Gray DH, Katz JM (1981) A histochemical study of muscle in club foot. J Bone Joint Surg Br 63:417–423
Handelsman JE, Badalamente MA (1981) Neuromuscular studies in clubfoot. J Pediatr Orthop 1:23–32
Herceg MB, Weiner DS, Agamanolis DP, Hawk D (2006) Histologic and histochemical analysis of muscle specimens in idiopathic talipes equinovarus. J Pediatr Orthop 26:91–93
Loren GJ, Karpinski NC, Mubarak SJ (1998) Clinical implications of clubfoot histopathology. J Pediatr Orthop 18:765–769
Maffulli N, Capasso G, Testa V, Borrelli L (1992) Histochemistry of the triceps surae muscle in idiopathic congenital clubfoot. Foot Ankle 13:80–84
Sirca A, Erzen I, Pecak F (1990) Histochemistry of abductor hallucis muscle in children with idiopathic clubfoot and in controls. J Pediatr Orthop 10:477–482
Feldbrin Z, Gilai AN, Ezra E, Khermosh O, Kramer U, Wientroub S (1995) Muscle imbalance in the aetiology of idiopathic club foot. An electromyographic study. J Bone Joint Surg Br 77:596–601
Tönnis D (1969) Elektromyographische und histologische untersuchungen zur frage der entstehung des angeborenen klumpfusses. Z Orthop 105:595–615
Caiozzo VJ, Utkan A, Chou R, Khalafi A, Chandra H, Baker M, Rourke B, Adams G, Baldwin K, Green S (2002) Effects of distraction on muscle length: mechanisms involved in sarcomerogenesis. Clin Orthop Relat Res 403(Suppl):S133–S145
Goldspink G, Williams P, Simpson H (2002) Gene expression in response to muscle stretch. Clin Orthop Relat Res 403(Suppl):S146–S152
Caiozzo VJ, Green S (2002) Breakout session 3: issues related to muscle growth, atrophy, and tissue engineering. Clin Orthop Relat Res 403(Suppl):S252–S261
Ester AR, Tyerman G, Wise CA, Blanton SH, Hecht JT (2007) Apoptotic gene analysis in idiopathic talipes equinovarus (clubfoot). Clin Orthop Relat Res 462:32–37
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The authors certify that their institution has approved the reporting of this study, that all the investigations were conducted in conformity with ethical principles of research, and informed consent was obtained.
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Ippolito, E., De Maio, F., Mancini, F. et al. Leg muscle atrophy in idiopathic congenital clubfoot: is it primitive or acquired?. J Child Orthop 3, 171–178 (2009). https://doi.org/10.1007/s11832-009-0179-4
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DOI: https://doi.org/10.1007/s11832-009-0179-4