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.
CONCLUSIONS: Bisphosphonates provide an effective and safe treatment option for primary and secondary HPOA. However, there is a lack of randomized controlled trials.

Arthritis is an unusual manifestation of paraneoplastic syndrome, appearing in a variety of cancers, including pulmonary and colorectal. It can often pose a diagnostic challenge to physicians, since it may be difficult to distinguish from more commonly encountered rheumatic illnesses. Moreover, synchronous cancers are rare and unexpected in patients with symmetrical polyarthritis. Hypertrophic pulmonary osteoarthropathy is to be considered in patients with polyarthritis and lung neoplasia. The aim of this report is to highlight the case of a patient presenting with paraneoplastic polyarthritis, which led to identifying the presence of underlying synchronous lung and colorectal malignancies. Lymph node biopsy was performed raising suspicion of Caplan\'s syndrome but lung lobectomy confirmed adenocarcinoma. Rheumatologists should be reacquainted with rheumatic manifestations in malignant diseases.

A 71-year-old male with hypertrophic osteoarthropathy was referred to our hospital because of a nocturnal fever and tenderness stretching from the left parotid region to the left front neck, in which antibacterials were ineffective. He was diagnosed with Takayasu\'s arteritis following findings of contrast-enhanced computed tomography and neck ultrasound. This is the first report to describe the development of Takayasu\'s arteritis after a prolonged course of typical hypertrophic osteoarthropathy, and the proposed hypertrophic osteoarthropathy and Takayasu\'s arteritis have common underlying pathophysiological factors.

Hypertrophic osteoarthropathy (HOA) is a paraneoplastic syndrome, the exact pathogenesis of which remains to be elucidated. The case of a 69-year-old man who developed intractably painful HOA secondary to lung cancer is presented. Contrast-enhanced computed tomography of the chest showed an 80-mm solid nodule with a large low-density area. The patient was diagnosed as having stage IIIA undifferentiated non-small cell lung cancer. The combination of carboplatin and paclitaxel with bevacizumab reduced tumor size and plasma vascular endothelial growth factor (VEGF) levels, relieving his leg pain. On immunohistochemical examination, lung cancer cells were positive for VEGF. A hypoxic tumor microenvironment may have caused some lung cancer cells to express hypoxia-inducible factor-1α, which contributed, at least in part, to the production of VEGF. The deep dermis vessels showed proliferation in the shin, with their thickened walls positive for VEGF. These findings may encourage investigators to explore novel management strategies for painful HOA.

暂无摘要。

Rheumatic paraneoplastic syndromes are rare syndromes that occur at distant sites from the underlying tumor and may involve the bones, joints, fasciae, muscles, or vessels. In the absence of a known tumor, early recognition of a rheumatic syndrome as paraneoplastic permits dedicated work-up for, and potentially early treatment of an occult malignancy. Although there is a continuously growing list of paraneoplastic rheumatic disorders, not all of these disorders have a well-established association with a neoplastic process. The goals of this article are to review the clinical characteristics, diagnostic work-up, and imaging findings of well-documented rheumatic paraneoplastic disorders.

Giant-cell carcinoma of the lung (GCCL) is a rare histological form of poorly differentiated non-small-cell lung cancer, which is classified as a subtype of pulmonary sarcomatoid carcinomas. In this case report, we describe the case of a 57 year old female with a past medical history of HIV on HAART (CD4 count at the time was 621 cell/μl). She presented to the hospital with a two months history of productive cough with yellowish sputum containing streaks of blood, twelve pound weight loss, bilateral hand swelling, and knee pain with noticeable finger clubbing on physical examination. Chest computed tomography scan and subsequent bronchoscopy was performed and revealed a protruding endobronchial lesion in the right upper lobe (RUL) bronchus. Definitive diagnosis established by way of pathologic analysis of the resected specimen obtained from RUL lobectomy revealed sarcomatoid giant cell carcinoma, with tumor size 9.5 cm and invasion of the visceral pleura and 1/13 hilar lymph node involvement. The pathological stage was determined as pT3N1Mx based on the tumor node metastasis (TNM) staging system. The patient was started on adjuvant combination cisplatin and docetaxel therapy with supplemental G-CSF four months after surgery and followed as an outpatient. The significance of this case is that it highlights a very rare lung cancer, unveiling a possible paraneoplastic syndrome associated with this malignancy and the impact of HIV HAART therapy in carcinogenesis.

BACKGROUND: Hypertrophic osteoarthropathy (HOA) is a musculoskeletal pathology that often occurs as a paraneoplastic syndrome. 90% of HOA cases occur secondary to malignancy. 60 to 80% of which are lung cancers.
METHODS: We present a case of a 61-year-old man who had worsening knee pain. HOA was incidentally noted on extremity X-ray. The patient was found to have a soft-tissue attenuating mass on chest X-ray. The diagnosis of non-small cell lung cancer was made after bronchoscopy. WHY SHOULD AN EMERGENCY PHYSICIAN BE AWARE OF THIS?: HOA can be an indication of malignancy, most commonly lung cancer, so it is important to recognize the key radiographic findings associated with HOA. When treating patients with bone pain and clubbed digits, emergency physicians should strongly consider screening for more severe primary pathology.

Hypertrophic osteoarthropathy (HOA) is an orphan syndrome characterized by abnormal proliferation of the skin and osseous tissues at the distal parts of the extremities. The main clinical features are: a peculiar bulbous deformity of the tips of the digits conventionally described as \"clubbing,\" periosteal proliferation of the tubular bones, and synovial effusions. In most instances, HOA develops a reaction to a severe internal illness, such as lung cancer, cyanotic heart disease, or liver cirrhosis. There is a subgroup of patients who do not have underlying pathology. Such cases are classified as having primary HOA. Digital clubbing is easy to recognize. Any patient with newly developed digital clubbing should undergo careful search for an underlying illness with special attention to intra-thoracic pathologies. Painful HOA is treated with non-steroidal anti-inflammatory medications. Vascular endothelial growth factor and prostaglandin E2 have been proposed as key bone proliferating mediators.

Pachydermoperiostosis is a rare condition representing a primary form of hypertrophic osteoarthropathy. It presents in different stages. Patients often overlook early symptoms, because they are benign. The most common manifestations are clubbing of the fingers and toes, skin thickening with characteristic folds on the face and head and widening of joints accompanied by radiological changes. Surgical treatment is not often needed, and, consequently, there are no strict guidelines on surgical management, which is mainly based on case report ana-lysis. This paper presents a case of surgical management of pachydermoperiostosis.