Schimke versus non-Schimke chronic kidney disease: an anthropometric approach

Pediatrics. 2006 Aug;118(2):e400-7. doi: 10.1542/peds.2005-2614. Epub 2006 Jun 30.

Abstract

Schimke-immuno-osseous dysplasia is a rare autosomal-recessive multisystem disorder with the main clinical features of disproportionate growth deficiency, defective cellular immunity, and progressive renal disease. It is caused by mutations of SMARCAL1, a gene encoding a putative chromatin remodeling protein of unknown function. Because a detailed description of the clinical features is an essential first step in elucidating the function of SMARCAL1, we present the first detailed anthropometric data for Schimke-immuno-osseous dysplasia patients. By comprehensive anthropometric examination (28 parameters) of 8 patients (3 females) with the typical findings of Schimke-immuno-osseous dysplasia (mean age: 14.8 years; range: 4.9-30.5 years) and 304 patients (117 females) with congenital and hereditary chronic kidney disease (mean age: 10.7 +/- 4.8 years; range: 3-21.8 years), we show that Schimke-immuno-osseous dysplasia patients differ significantly from those with other forms of chronic kidney disease. z scores were calculated with reference limits derived from 5155 healthy children (2591 females) aged 3 to 18 years. The key finding was that, in the latter group, median leg length was significantly more reduced than sitting height, whereas in Schimke-immuno-osseous dysplasia patients, the reduction of sitting height was significantly more pronounced than for leg length. Therefore, the ratio of sitting height/leg length might be a simple tool for the clinician to distinguish Schimke-immuno-osseous dysplasia from other chronic kidney disease patients. Schimke-immuno-osseous dysplasia is very likely if this ratio is < 0.83. However, other forms of chronic kidney disease have to be discussed in case of a ratio > 1.01.

Publication types

  • Comparative Study

MeSH terms

  • Adolescent
  • Adult
  • Anthropometry*
  • Arm / pathology
  • Body Height
  • Bone Diseases, Developmental / genetics
  • Bone Diseases, Developmental / pathology*
  • Cephalometry
  • Child
  • Child, Preschool
  • Chromosome Disorders / genetics
  • Chromosome Disorders / pathology*
  • DNA Helicases
  • Disease Progression
  • Dwarfism / genetics
  • Dwarfism / pathology*
  • Female
  • Glomerulosclerosis, Focal Segmental / genetics
  • Glomerulosclerosis, Focal Segmental / pathology*
  • Glomerulosclerosis, Focal Segmental / surgery
  • Humans
  • Immunologic Deficiency Syndromes / genetics
  • Immunologic Deficiency Syndromes / pathology*
  • Kidney Failure, Chronic / genetics
  • Kidney Failure, Chronic / pathology
  • Kidney Failure, Chronic / surgery
  • Kidney Transplantation
  • Leg / pathology*
  • Male
  • Nephrotic Syndrome / genetics
  • Nephrotic Syndrome / pathology*
  • Nephrotic Syndrome / surgery
  • Phenotype
  • Skinfold Thickness

Substances

  • SMARCAL1 protein, human
  • DNA Helicases