Hypertrophic osteoarthropathy in childhood malignancy

Abstract

Hypertrophic osteoarthropathy (HOA), well known in adults, is rarely encountered in children. The clinical features include clubbing of the fingers and toes, arthritis, and a sometimes painful ossifying periostitis of the tubular bones. Apart from a hereditary form (primary HOA), most of the cases encountered in children are secondary and associated with conditions such as chronic suppurative lung processes (e.g., cystic fibrosis), congenital heart disease, biliary atresia, and polyposis coli. The association with malignant disorders, which is relatively common in adults, is very rare in children. In 1986 the authors published a case report of a patient with carcinoma of the nasopharynx who developed HOA. Another similar patient has been encountered. In both, the appearance of HOA was associated with a very poor prognosis. A meticulous research of the literature from 1890 to 1990 revealed only 24 children (19 boys, 5 girls) under the age of 18, with malignancy and associated HOA. Among them were 10 patients with a carcinoma of the nasopharynx, 8 with osteosarcoma, 3 with Hodgkin's lymphoma, 1 with a periosteal sarcoma, 1 with mesothelioma of the pleura, and 1 with carcinoma of the thymus. In five patients with HOA, there were no abnormalities of the lungs, mediastinum, or pleura, and none developed during the course of the disease. Many authors mention the predictive value of HOA, especially in association with malignant tumors. In contrast to suppurative processes in the lungs, in those with neoplastic disease involving the chest, HOA may precede pulmonary symptoms by 1-18 months. A striking feature of HOA in these instances is the reversibility of the complaints after successful treatment of the disorder of the chest, both in benign and malignant conditions. The present case is the second reported by the authors and the first description of a girl with carcinoma of the nasopharynx developing HOA.

A Case of Hypertrophic Osteoarthropathy Associated with Nasopharyngeal Carcinoma in a Child

“…From 1890 to 2002 only 31 children, including our case, under the age of 18 with malignancy and associated HOA have been reported (2)(3)(4)(5). Among them, 13 cases had nasophryngeal carcinoma.…”

“…It can be divided into primary HOA, which is not associated with any other medical conditions and secondary HOA, which can be further divided into pulmonary and non-pulmonary causes. Primary HOA is known as pachydermoperiostosis and transmitted as an autosomal dominant trait (2). In secondary HOA, common pulmonary causes include cystic fibrosis, pulmonary fibrosis, primary or metastatic carcinoma, and mesothelioma.…”

“…HOA in malignancy is very rare in children. To date, only 30 cases of HOA in association with childhood neoplasia have been published (2)(3)(4)(5). Among them 12 patients had carcinoma of the nasopharynx, 8 had an osteosarcoma, 5 had Hodgkin's disease, 3 had a carcinoma of the thymus, 1 had a periosteal sarcoma, and 1 had a mesothelioma of the pleura.…”

Childhood intrathoracic Hodgkin lymphoma with hypertrophic pulmonary osteoarthropathy: a case report and review of the literature

“…Secondary HOA is more common and was first defined as being secondary to intrathoracic inflammatory lesions. Secondary HOA may also be related to various acute/ chronic pulmonary, cardiac or gastrointestinal disorders, either infectious or neoplastic, examples of which include bacterial endocarditis, congenital heart diseases, infected vascular prostheses, cystic fibrosis, chronic airway infection, chronic lung disease, idiopathic pulmonary fibrosis, intrathoracic tumors such as bronchogenic carcinoma and mesothelioma, cirrhosis, inflammatory bowel disease and infections or malignancies of the gastrointestinal system [2,12,22]. HOA secondary to pulmonary causes is known as hypertrophic pulmonary osteoarthropathy.…”

“…The condition may be primary, with an absence of associated conditions [10], or secondary to various pulmonary, cardiac or gastrointestinal diseases [2,12,22]. Intrathoracic neoplasms are not major causes of HOA in children [22].…”