Abstract
HHO in association with chronic liver disease is a rare syndrome reported in only 4 paediatric cases to date. A retrospective study in children with chronic liver disease due to neonatal hepatitis (NH) or biliary atresia (BA) at Baragwanath Hospital was performed. The prevalence of HHO, age of onset of its various components as well as predisposing factors were noted.
Of 40 patients studied, 19 (47%) had ongoing jaundice (3 with NH, 16 with DA). Seven infants had evidence of HHO (clubbing and arthropathy and/or periosteal reactions) and a further 3 had clubbing only. The earliest manifestation in all cases was clubbing (20 ± 10 months). Clinical joint involvement developed later, while bono thickening was the last sign to be elicited, although radiological periosteal reactions were evident much earlier. The mean age of patients, with HHO is 33.5 ± 11 months and those without HHO is 13.6 ± 8 months.
HHO in children occurs more frequently than deserted in the literature. Its pathogenesis is unknown, although several mechanism have been postulated including failure of degradation of humoral substances. In view of the high prevalence of NH and BA at the hospital and the unavailability of liver trunsplantation, these infants are an ideal study population. A prospective study is in progress.
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Zuckerman, M. Hepatic Hypertrophic Osteoarthropathy (HHO) - An apparent high prevalence in a developing country. Pediatr Res 35, 41 (1994). https://doi.org/10.1203/00006450-199402000-00068
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DOI: https://doi.org/10.1203/00006450-199402000-00068
