Periostitis and hypertrophic pulmonary osteoarthropathy: report of 2 cases and review of the literature.

OBJECTIVE

To demonstrate the clinical spectrum of hypertrophic osteoarthropathy (HOA).

METHODS

We report 2 cases of HOA and performed a computer-assisted search of Medline/PubMed for the medical literature from 1960 to June 2008 using the keywords HOA, periostitis, and clubbing. These were also combined with the text words cancer, rheumatic disease, etiology, pathogenesis, hypothesis, transplant, and treatment. Only the English language literature, with pertinent information, was included.

RESULTS

Our 2 cases include 1 HOA case with clubbing, in a patient with a right to left shunt from cryptogenic cirrhosis and interstitial lung disease, and 1 HOA case without apparent clubbing, in a patient with chronic lung transplant rejection secondary to tobacco smoking and related emphysema. Review of the literature has shown that HOA is associated with a wide variety of disorders, approximately 80% are found with primary or metastatic pulmonary malignancies. Various rheumatic diseases, such as systemic vasculitis, can also be associated with HOA. With respect to the pathogenesis, vascular endothelial growth factor, platelet-derived growth factor, and platelets may play crucial roles. Therapeutically, bisphosphonates, such as pamidronade or octreotide, may be tried to relieve symptoms in refractory cases.

CONCLUSIONS

HOA, especially periostitis without clubbing, may go unrecognized. Involvement of vascular endothelial growth factor, platelet-derived growth factor, and platelets in the pathogenesis of HOA has been postulated and supported by recent data. HOA may present as a partial syndrome without clubbing and about 20% of cases have HOA without detectable malignancy. One of our cases represents the first report of the association of HOA with lung transplantation.