Hypertrophic osteoarthropathy in childhood malignancy

Our plain-English summary of Hypertrophic osteoarthropathy in childhood malignancy, by C.R. Staalman, U. Umans (Medical and Pediatric Oncology, 1993).

The study

• Authors: C.R. Staalman, U. Umans
• Journal: Medical and Pediatric Oncology
• Published: 1993;21(9):676-9
• Type of article: Case report, Review
• Cited by: 45 articles, as counted by NIH iCite
• Read it: PubMed · Publisher

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.

In plain English

This article combined one case report with a review of published cases of hypertrophic osteoarthropathy (HOA) in children with cancer. HOA involves clubbing of the fingers and toes and new bone growth along the long bones. The case was an 11-year-old girl with carcinoma of the nasopharynx. After treatment and a remission lasting 1.5 years, she developed a very painful ossifying periostitis, which led to the discovery that her cancer had returned. She died 2 months later. The authors found 24 published cases in children no older than 17, making 25 with their own. Of these, 11 had carcinoma of the nasopharynx, 8 osteosarcoma, 3 Hodgkin's disease, 1 periosteal sarcoma, 1 mesothelioma of the pleura and 1 carcinoma of the thymus. The article does not discuss asbestos. Twenty of the 25 were male. HOA led to discovery of recurrence or metastasis in 15 of 25 patients. Nineteen patients died or showed extensive metastases, and among these 19 the mean survival after HOA was discovered was about 9 months. The authors concluded that HOA is a bad prognostic sign in these children.

We wrote this plain-English summary from the full article with the help of AI, and a second AI model checked each sentence against the article. It is not the authors' wording; the abstract and the article are their own account.

What the study did and found

• Design: case report combined with a literature review of published cases
• Who was studied: One 11-year-old girl treated at the Academic Hospital at the University of Amsterdam, plus 24 published cases of children not older than 17 years with a proven malignancy and HOA, 25 patients in total
• Exposure: No asbestos or other exposure was studied. The article concerns HOA occurring in children with malignancies, one of which was mesothelioma of the pleura.
• Main results:
  • In the present case, a very painful ossifying periostitis led to discovery of a recurrence of the nasopharynx carcinoma. A chest X-ray showed mediastinal lymph node enlargement, and the child died 2 months later.
  • Of the 25 patients, 11 had carcinoma of the nasopharynx, 8 osteosarcoma, 3 Hodgkin's disease, 1 periosteal sarcoma, 1 mesothelioma of the pleura and 1 carcinoma of the thymus.
  • Of the 25 cases, only 2 were under age 10 and 20 were male.
  • The appearance of HOA led to discovery of recurrence or metastasis in 15 of 25 patients.
  • In 20 of 25 cases, chest X-ray abnormalities were present at onset of HOA or later. Of these 20 children, only 2 had a known survival of more than 18 months.
  • The other 5 children all had carcinoma of the nasopharynx and showed no chest X-ray abnormality. Their maximum survival was 14 months.
  • Reversal of HOA complaints after treatment was mentioned in 6 of the 25 cases. In 2 of these, remission lasted more than 18 months.
  • Nineteen patients died or showed extensive metastases. Among these 19 patients, the mean life span after HOA was discovered was about 9 months, and only 1 lived more than 2 years.
• The authors' conclusion: The authors concluded that HOA with a malignant tumor is very rare in children, that its appearance in a child with a tumor has a very serious meaning and is a bad prognostic sign, and that the exact cause of HOA is still unknown.
• Limitations the authors state:
  • The authors could not translate one article, describing a child with a schwannoma of the lung who developed HOA, so that case was not included.

Topics

Medical terms in this study, explained in our Lexicon: Clubbing, Fibrosis, Carcinoma, Prognosis, Sarcoma, Mesothelioma, Pleura, Mediastinum.

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