Multicentric osteolysis with nodulosis and arthropathy (MONA) with cardiac malformation, mimicking polyarticular juvenile idiopathic arthritis: case report and literature review

Eur J Pediatr. 2013 Dec;172(12):1657-63. doi: 10.1007/s00431-013-2102-8. Epub 2013 Jul 31.

Abstract

The 'vanishing bone' syndrome multicentric osteolysis with nodulosis and arthropathy (MONA) is a rare chronic skeleton disorder caused by matrix metalloproteinase 2 (MMP2) deficiency, mimicking erosive polyarticular juvenile idiopathic arthritis. MONA is characterised by facial dysmorphism, subcutaneous fibrocollagenous nodules, carpal and tarsal osteolysis and interphalangeal joint erosions. We present the case of a 5-year-old boy with double outlet right ventricle, ventricular septal defect, coarctation of the aorta and MONA. Previously, a total of 24 cases of MONA have been reported of which six also had congenital cardiac malformations. Despite treatment attempts of our patient with methotrexate, eternacept and prednisolone, serial X-ray studies documented continuous severe bone degeneration.

Conclusion: The case documents the natural history of MONA and establishes a link between MMP2 deficiency and heart development, and given the recurring cardiac association, we suggest that all MONA patients be examined for possible cardiac defects.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Abnormalities, Multiple / diagnosis
  • Abnormalities, Multiple / genetics
  • Antirheumatic Agents / therapeutic use*
  • Arthritis, Juvenile / diagnosis*
  • Child, Preschool
  • Diagnosis, Differential
  • Disease Progression
  • Hajdu-Cheney Syndrome / diagnosis*
  • Hajdu-Cheney Syndrome / drug therapy
  • Heart Defects, Congenital / diagnosis*
  • Heart Defects, Congenital / genetics
  • Humans
  • Male
  • Matrix Metalloproteinase 2 / analysis
  • Matrix Metalloproteinase 2 / deficiency*
  • Matrix Metalloproteinase 2 / genetics
  • Mutation, Missense
  • Sequence Analysis, DNA

Substances

  • Antirheumatic Agents
  • Matrix Metalloproteinase 2