Chest
Selected ReportsPulmonary Capillary Hemangiomatosis With Atypical Endotheliomatosis*: Successful Antiangiogenic Therapy With Doxycycline
Section snippets
Case Report
Over 6 months, a previously healthy 20-year-old male college student developed hemoptysis, cough, hoarseness, and sore throat. Symptoms responded partially to therapy with oral ampicillin that was prescribed for possible sinusitis. Initially, the patient continued normal activities and denied any fatigue or dyspnea, but eventually minimal physical activity severely exhausted him, and his hemoptysis increased.
A physical examination revealed a pale, chronically ill-appearing individual without
Results
Within weeks of initiating doxycycline therapy, the patient reported a substantial increase in exercise tolerance and a decrease in hemoptysis. The prednisone dose was decreased, and the patient was weaned approximately 4 months after starting therapy. After 6 months of doxycycline therapy, the α-interferon dose was decreased. The patient stopped taking the α-interferon on his own 2 months later. Nine months after starting doxycycline therapy, the patient was symptom-free. Improvement was seen
Discussion
To our knowledge, this case of PCH with atypical endotheliomatosis is the first case of PCH, or any other vascular malformation or neoplasm, that may have been treated successfully with doxycycline. This is one of only two well-documented cases in which a patient has responded to medical therapy. In the earlier case, α-interferon therapy was discontinued after 5 years without disease recurrence (CW White; personal communication; October 10, 2000). While α-interferon therapy has been shown in
ACKNOWLEDGMENT
The authors thank Judah Folkman for his thoughtful comments on this case, and his laboratory for performing urinary bFGF enzyme-linked immunosorbent assays. We thank the laboratory of Marsha Moses for performing the urinary zymography.
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Dr. Roberts was supported by a Nirenberg Center for Advanced Lung Diseases Fellowship, and Dr. Marler was the Garrett Smith Fellow in the laboratory of Dr. Judah Folkman.