Role of bisphosphonates in hypertrophic osteoarthropathy: a systematic review.

BACKGROUND: The role of bisphosphonates (BP) in hypertrophic osteoarthropathy (HPOA) is unclear. We presented a case of primary HPOA and performed a systematic review of literature on the effect of BP on treatment response in primary and secondary HPOA. METHODS: The study was prospectively registered in PROSPERO (CRD42022343786). We performed a PubMed literature search that restricted to the English language. We included patients diagnosed with primary or secondary HPOA who received BP. The primary endpoint assessed was the effectiveness of BP on response to pain or arthritis. Secondary outcomes included timing, degree, and duration of response, comparison to other HPOA therapies, impact of BP on radiology, bone scan, bone turnover markers, and adverse effects of BP. RESULTS: Literature search retrieved only case reports. Forty-five patients (21 primary, 24 secondary HPOA) had received BP. Majority(88.3%) experienced improvement in pain or arthritis. Response was gradual for primary HPOA and within a median of 3 to 7 days for secondary HPOA after treatment with BP. Most patients had reduced bone scan uptake after BP. When other HPOA therapies were tried, half responded to BP after not having previously responded to other therapies, while a third received the treatments concurrently, making it difficult to attribute treatment response to a drug. Reporting of other secondary outcomes was very heterogenous and qualitative to draw conclusions. No major adverse effects have been reported for BP in HPOA. CONCLUSION: Bisphosphonates provide an effective and safe treatment option for primary and secondary HPOA. However, there is a lack of randomized controlled trials.

Hypertrophic pulmonary osteoarthropathy with esophageal sarcomatoid carcinoma: a case report.

Hypertrophic pulmonary osteoarthropathy (HPOA), mainly manifested clubbing, is rare in patients with esophageal sarcomatoid carcinoma. We herein describe a 48-year-old Chinese man whose advanced sarcomatoid carcinoma was diagnosed while examining his symptoms of HPOA. The patient had no opportunity of surgery after surgical evaluation. Chemoradiation, including 5 cycles of chemotherapy (Paclitaxel liposome 60 mg day 1 and nedaplatin 30 mg day 1, q1w) and 6MV-X/VMAT-95%PGTV 59.92 Gy/2.14 Gy/28 F and 95%PTV 50.4 Gy/1.8 Gy/28 F, and subsequent 6 cycles of chemotherapy (paclitaxel liposome 210 mg day 1 and nedaplatin 50 mg day 1, 60 mg day 2, q3w) shrank the tumor and the condition of the patient became stable without clubbing remission or exacerbation. No medical case report found in a PubMed search in the indexed English-language literature in the past 20 years, though there are some reports of HPOA combined with other pathologic types of esophageal carcinoma. The patient's condition was effectively controlled by chemotherapy and radiotherapy and showed stable disease. However, the five-year survival rate of advanced esophageal carcinoma patients is very low with low life quality, and the adverse reactions also contribute to low life quality. The purpose of this report is to present the feature and our treatment for primary esophageal sarcomatoid carcinoma with HPOA, which could be helpful for further understanding of the disease and clinical decision making. Moreover, this article also reviews esophageal carcinoma with HPOA and sarcomatoid carcinoma in the esophagus. We look forward to the breakthrough of immunotherapy and molecular targeting therapy to improve the situation.

Successful Treatment of ROS1-rearranged Lung Cancer Complicated by Hypertrophic Pulmonary Osteoarthropathy with Crizotinib Therapy.

Hypertrophic pulmonary osteoarthropathy (HPO) is a paraneoplastic syndrome characterized by digital clubbing, arthritis, and periostitis. Tumor removal usually leads to the resolution of these symptoms. We herein report the efficacy of crizotinib treatment for treating the symptoms of HPO associated with c-ros oncogene 1 receptor tyrosine kinase (ROS1)-rearranged lung cancer. A 71-year-old woman presented with a pulmonary tumor and arthritis. She was diagnosed with a ROS1-rearranged lung adenocarcinoma [stage IIIB (cT4N2M0) ] with HPO. Crizotinib dramatically reduced the tumor size and resolved the symptoms. After two months of crizotinib treatment, she underwent lobectomy, and a pathological evaluation revealed ypstage IIIA (ypT3a, ypN1). Crizotinib treatment was effective for reducing the tumor size and improving the symptoms of HPO.

You're the Flight Surgeon.

Chumbley EM. You're the flight surgeon: a case of bilateral leg pain and swelling in a loadmaster. Aerosp Med Hum Perform. 2017; 88(5):511-515.

Treatment of hypertrophic osteoarthropathy in the case of pulmonary metastasis secondary-to-nasopharyngeal carcinoma with zoledronic acid: an enlightening experience.

We describe a case of 23-year-old man, who presented with painful hypertrophic pulmonary osteoarthropathy involving bilateral upper and lower extremities, resulting from intrathoracic metastasis. The patient had a history of undifferentiated nasopharyngeal carcinoma which was treated successfully 2 years ago. The painful osteoarthropathy had made the patient incapacitated. A single dose of 4 mg of intravenous zoledronic acid (ZA) was given which resulted in complete resolution of pain along with reduction of swelling. There was no recurrence on follow-up. Bisphosphonates by their action on bone metabolism might alleviate the symptoms and its use should be encouraged in cancer patients with debilitating arthropathies. This article aims at highlighting the role of bisphosphonates, particularly ZA in managing patients with hypertrophic osteoarthropathy (HOA) and sharing our experience with this drug because of the rarity of the condition and lack of sufficient data in the medical literature.

Hypertrophic osteoarthropathy with acro-osteolysis in a patient with primary pulmonary hypertension.

Hypertrophic osteoarthropathy is an entity characterized by a triad of periostitis of long bones, clubbing and arthritis. Radiologically there are two patterns, one characterized by new bone formation which predominates in patients with pulmonary disease, and another by acro-osteolysis that is most frequently associated with congenital heart disease. We report the case of a 30-year-old man diagnosed with primary pulmonary hypertension for two years, developing hypertrophic osteoarthropathy with a mixed radiological pattern.

Pain management in hypertrophic pulmonary osteoarthropathy: an illustrative case and review.

Hypertrophic pulmonary osteoarthropathy (HOA) is a condition associated with lung cancer and many other diseases. Pain associated with HOA can be disabling, unremitting, and refractory to conventional analgesic medications. We present a challenging case of HOA in a patient with nonsmall cell lung cancer and review specific therapies for management of HOA-related pain.

Treatment of hypertrophic osteoarthropathy with zoledronic acid: case report and review of the literature.

OBJECTIVES: Zoledronic acid (ZA) is rarely used to manage hypertrophic osteoarthropathy (HOA). We report our experience with ZA treatment of a patient with HOA and sarcoidosis who had also undergone lung transplant. We also conducted a literature review of the usefulness of bisphosphonates in HOA. METHODS: We performed a PubMed literature search using keywords HOA, periostitis, bisphosphonate, ZA, sarcoidosis, and lung transplant. A PRISMA flow diagram is presented to depict the data collection process, and a case is reported. RESULTS: A 62-year-old woman with bilateral lung transplant as a result of severe pulmonary sarcoidosis developed severe limb pain and inflammatory polyarthritis. HOA was diagnosed in the presence of periostitis with the symptoms. Failure of the refractory bone and joint pain to respond to low doses of prednisone, tramadol, or even pamidronate infusion prompted a trial of a single dose of intravenous ZA. Surprisingly, the pain completely resolved without recurrence. A total of 12 cases of HOA treated with bisphosphonates were retrieved from the literature and reviewed. CONCLUSIONS: Bisphosphonates are generally effective therapy for HOA-related pain. ZA may be even more efficacious and longer lasting than pamidronate for management of the bone and joint pain associated with HOA irrespective of the underlying disorders.

Clubbing and hypertrophic osteoarthropathy in two patients taking long-term bevacizumab for metastatic colorectal cancer.

INTRODUCTION: The authors report two cases of young patients who developed clubbing and hypertrophic osteoarthropathy in one case or lung diffusion disorder in the second, after a long-term use of bevacizumab plus chemotherapy in a palliative setting of metastatic colorectal cancer. DISCUSSION: We propose that patients on long-term bevacizumab be examined for clubbing and undergo respiratory function tests and that this would be studied prospectively before beginning trials in evaluating this monoclonal antibody given for 2 years in an adjuvant setting.

Treatment of painful hypertrophic osteoarthropathy associated with non-small cell lung cancer with octreotide: a case report and review of the literature.

Hypertrophic osteoarthropathy (HOA) is a syndrome most commonly associated with non-small cell lung cancer and consists of periostitis, digital clubbing and painful polyarthropathy. Its symptoms may be disabling and are reportedly difficult to manage effectively with conventional analgesia. We present a case of a lung cancer patient with opioid resistant painful HOA in whom analgesia was achieved with octreotide.

Hypertrophic osteoarthropathy in intestinal transplant recipients.

We report 3 patients with periosteal new bone formation consistent with hypertrophic osteoarthropathy (HOA), in the context of intestinal allograft rejection. Patient 1 developed thick periosteal new bone formation of the right arm during a prolonged episode of intestinal acute cellular rejection (ACR) 2 months posttransplant. Patient 2 developed ankle pain and swelling during an episode of severe ACR. Plain films showed periosteal new bone formation of the left ankle. In patient 3, the right wrist became swollen during an episode of moderate ACR, whereas plain films demonstrated mild periosteal reaction. Patients 2 and 3 had resolution of their symptoms once the ACR resolved with treatment. This is the first case series of HOA occurring in association with intestinal ACR. We speculate that an immune-mediated process is responsible for the bone disease. Further inquiry will help establish if HOA is related to transplant status, intestinal inflammation, or allograft rejection in general.

[A woman with red, swollen lower legs]. / Een vrouw met rode, gezwollen onderbenen.

A 32-year old female patient with Graves' disease was diagnosed with pretibial myxedema. It is part of the triad of extrathyreoidal manifestations of Graves' disease: exophthalmia, pretibial myxedema, and acropachy. The patient was successfully treated with topical corticosteroids.

Hypertrophic osteoarthropathy masquerading as lower extremity cellulitis and response to bisphosphonates.

Hypertrophic osteoarthropathy (HOA) is characterized by clubbing of digits and, in more advanced stages, by periosteal new bone formation and synovial effusions. Secondary HOA typically occurs in the setting of intrathoracic malignancy, infections or lung metastases. We report a case with an atypical presentation of HOA. The prominent systemic signs, presentation limited to lower extremities only and an excellent response to bisphosphonates make this case unusual.

Hypertrophic osteoarthropathy effectively treated with zoledronic acid.

Hypertrophic osteoarthropathy (HOA) is a syndrome characterized by digital clubbing, arthralgias, and tissue swelling and is frequently described in association with bronchogenic carcinoma. The associated pain can be disabling, and symptoms are often resistant to conventional analgesic medications. We present a patient with HOA of the lower extremities and wrists that developed after bronchogenic carcinoma. The pain and swelling completely resolved after a single administration of 4 mg of zoledronic acid. The proposed pathogenesis of HOA and the mechanisms by which bisphosphonates might alleviate symptoms are reviewed.