Periostitis and Hypertrophic Pulmonary Osteoarthropathy: Report of 2 Cases and Review of the Literature

Hypertrophic osteoarthropathy in a child due to thoracic Hodgkin’s disease

“…Hypertrophic osteoarthropathy in children is rare and is typically considered to be a sign of pulmonary disease [1][2][3]. However, it may be related to a variety of inflammatory or neoplastic disorders, including congenital heart diseases, pulmonary or cystic fibrosis, infectious airway disease, chronic liver and inflammatory bowel disease as well as various intrathoracic and gastrointestinal tumours [1][2][3].…”

“…However, it may be related to a variety of inflammatory or neoplastic disorders, including congenital heart diseases, pulmonary or cystic fibrosis, infectious airway disease, chronic liver and inflammatory bowel disease as well as various intrathoracic and gastrointestinal tumours [1][2][3]. While up to 90% of HPOA cases are associated with primary or metastatic pulmonary neoplasms in adults, intrathoracic malignant disease accounts for only 12% of HPOA cases in children [1][2][3]. The presence of HPOA has been regarded as a poor prognostic sign, since most of the patients with malignancy and HPOA have showed extensive metastases [2].…”

“…HPOA has a characteristic radiological appearance [1][2][3][4]. Initially, there is periosteal new bone formation forming a thin radiopaque line around the cortex and separated from it by a radiolucent band.…”

“…Subsequently, the two layers gradually fuse and lead to poorly demarcated or lamellar periosteal reaction. This appearance may be easily differentiated from bone involvement from the Hodgkin's disease itself, as periosteal infiltration by lymphoma usually affects the anterior surfaces of the vertebral bodies rather than the long bones and is always associated with cortical bone destruction [1][2][3][4].…”

Characterization of hypertrophic osteoarthropathy in an identified skeleton from Évora, Portugal, using combined and comparative morphology and microscopy

“…Only the secondary form is described in dogs. The primary form, also called pachyodermoperiostosis or Touraine-Solente-Golé syndrome, is a rare genetic disease that is inherited in an autosomal fashion (Latos-Bielenska et al, 2007), recently mapped to human chromosome 4q33-q34 and to gene mutations encoding 15-hydroxyprostangladin degradation (Uppal et al, 2008;Yao et al, 2009). This form, usually severe, affects primarily males (Aufderheide and Rodriguez-Martín, 1998), appears around the time of puberty, and its progression is limited to the puberal growth period (Christensen et al, 2013).…”