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OBJECTIVE: To assess the health care costs and utilization in patients with hereditary hemorrhagic telangiectasia (HHT) in the United States. PATIENTS AND METHODS: Retrospective analysis of patients with HHT diagnosed between 2007 and 2017 was performed using deidentified administrative claims data from the OptumLabs Data Warehouse. Adult patients with new (incident) diagnosis of HHT between January 1, 2007, and December 31, 2017, were included. Comparisons were made using the Wilcoxon rank sum test. RESULTS: Three thousand nine hundred seventy-seven patients with a first diagnosis of HHT between 2007 and 2017 were identified, of which 3590 were matched 1:1 to non-HHT patients with similar baseline characteristics and comorbidities. These 3590 patients with HHT were 63.1% female and 83.9% white with a mean age of 51.1 ± 18.5 years, and a mean follow-up period of 3.2 ± 2.2 years (range, 1.0-11.7 years). Compared with the control group, the cumulative 5-year median total health care cost for patients with HHT was 41.4% higher ($21,118 vs $14,929; P < .001) in those with private commercial insurance and 31.7% higher ($35,462 vs $26,925; P < .001) in those with Medicare Advantage coverage. The median annual health care costs were significantly higher in patients with HHT with commercial insurance and Medicare Advantage in the first year after diagnosis ($4,333 vs $1,804; P < .001), and ($7,322 vs $5,245; P < .001), respectively, and remained higher throughout the duration of follow-up. Further analysis showed that outpatient clinic visits, hospital admission, imaging rates, invasive procedures, iron infusions, and blood transfusions were all significantly higher in the HHT group. CONCLUSION: Patients with HHT have significantly higher health care costs compared with a matched control group. A better understanding of the reasons underlying these cost differences will provide opportunities for patients, providers, and other stakeholders to better manage this rare condition.
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BACKGROUND: Hypereosinophilia (HE) is defined by the presence of >1.5 × 109/L eosinophils in the peripheral blood. Paraneoplastic HE has been reported in a number of solid and hematologic malignancies including ovarian cancer. We present a case with paraneoplastic HE in the setting of underlying endometrioid ovarian carcinoma. CASE PRESENTATION: An 88-year-old woman presented with cough, dyspnea and 20-pound unintentional weight loss of one month duration. Evaluation revealed peripheral hypereosinophilia (HE) with absolute eosinophil count of 15.38 × 109/L (53.8%) and an elevated exhaled nitric oxide at 172 parts per billion (normal < 39 PPB). Given the HE and unintentional weight loss, computed tomography (CT) scan was obtained and showed a pelvic mass. The patient underwent bilateral salpingo-ophorectomy with pathology consistent with endometrioid ovarian carcinoma. The patient experienced complete resolution of her cough, dyspnea, and peripheral eosinophilia following surgical resection. CONCLUSION: This case highlights that solid malignancy should be considered in patients with marked HE.
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BACKGROUND: While chemotherapeutic agents result in an improvement in both disease-free and overall survival in cancer patients, treatment can result in short and long-term complications. One well-known complication is neuropathy which can result from a number of chemotherapeutic agents. However, chemotherapy-induced phrenic neuropathy is an exceedingly rare phenomenon with few cases reported in the literature. CASE: A 34-year-old male with metastatic testicular cancer presented with progressive dyspnea on exertion after initiation of chemotherapy with bleomycin, cisplatin, and etoposide. Multiple diagnostic studies were performed including pulmonary function testing, chest computed tomography, fluoroscopic sniff evaluation, in addition to phrenic nerve electromyography. Based on results of these tests, the diagnosis of chemotherapy-induced phrenic neuropathy was made. CONCLUSION: Chemotherapy-induced phrenic neuropathy, although rare, should be considered as a cause of dyspnea in cancer patients following initiation of chemotherapy.