Early-onset facioscapulohumeral muscular dystrophy - significance of pelvic extensors in sagittal spinal imbalance.

Although facioscapulohumeral muscular dystrophy (FSHD) is the third most common inherited myopathy, cases of infantile or early-childhood onset have rarely been reported. The purpose of this study was to describe a case of early-onset FSHD with lumbar hyperlordosis, which shows the significance of the dynamic component of sagittal spinal imbalance. An 11-year-old girl presented with progressive gait disturbance and lumbar hyperlordosis. The motor power of her pelvic extensor muscles was grade 3. Pelvic tilt and hip flexion were markedly increased as determined by gait analysis. The most important factor in the development of hyperlordosis is the weakness of the pelvic extensor muscles, and the results of gait analysis exquisitely explain the pathophysiology. The patient stands with her spine hyperextended to maintain upright posture by a compensatory mechanism of relatively strong back extensor muscles. Corrective surgery for lumbar hyperlordosis was not considered because it could have eliminated the compensatory lumbar hyperextension, thus making the spine of the patient stoop forward through her hip joint during walking by the weakness of her pelvic extensor muscles. This FSHD case is an impressive example of a patient showing the concept that weak pelvic extensor muscles cannot keep the spine upright and balanced.

[1]  R. Vialle,et al.  Lumbar hyperlodosis in cerebral palsy: anatomic analysis and surgical strategy for correction , 2006, Child's Nervous System.

[2]  K. Felice,et al.  Facioscapulohumeral dystrophy presenting as infantile facial diplegia and late‐onset limb‐girdle myopathy in members of the same family , 2005, Muscle & nerve.

[3]  M. Dorobek,et al.  A severe case of facioscapulohumeral muscular dystrophy (FSHD) with some uncommon clinical features and a short 4q35 fragment. , 2004, European journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society.

[4]  J. Yoo,et al.  Dynamic Sagittal Imbalance of the Spine in Degenerative Flat Back: Significance of Pelvic Tilt in Surgical Treatment , 2001, Spine.

[5]  L. Lenke,et al.  Isolated thoracolumbar and lumbar hyperlordosis in a patient with cerebral palsy. , 2000, Journal of spinal disorders.

[6]  K Goto,et al.  Two Cases of Chromosome 4q35-Linked Early Onset Facioscapulohumeral Muscular Dystrophy with Mental Retardation and Epilepsy , 1998, Neuropediatrics.

[7]  R. Abresch,et al.  Profiles of neuromuscular diseases. Facioscapulohumeral muscular dystrophy. , 1995, American journal of physical medicine & rehabilitation.

[8]  C. Wijmenga,et al.  Facioscapulohumeral muscular dystrophy in early childhood. , 1994, Archives of neurology.

[9]  M. Fardeau,et al.  Diagnostic criteria for facioscapulohumeral muscular dystrophy , 1991, Neuromuscular Disorders.

[10]  B. Korf,et al.  Locomotor problems in infantile facioscapulohumeral muscular dystrophy. Retrospective study of 9 patients. , 1991, Acta orthopaedica Scandinavica.

[11]  Gordon V. Watters,et al.  A Clinician's View of Neuromuscular Diseases , 1978 .

[12]  Brooke Mh Clinical examination of patients with neuromuscular disease. , 1977 .