Atypical and incomplete pulmonary hypertrophic osteoarthropathy in the left distal femur: a case report
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CASE REPORT
Atypical and incomplete pulmonary hypertrophic osteoarthropathy in the left distal femur: a case report Gang Xu, Norio Yamamoto* , Katsuhiro Hayashi, Akihiko Takeuchi, Shinji Miwa, Kentaro Igarashi, Yuta Taniguchi, Yoshihiro Araki, Hirotaka Yonezawa, Sei Morinaga and Hiroyuki Tsuchiya
Abstract Background: Pulmonary hypertrophic osteoarthropathy (PHO) is a rare paraneoplastic syndrome that mainly occurs in patients with lung cancer. Most symptoms occur symmetrically, and unilateral symptoms without clubbing are infrequent. This report presents the case of a rare atypical symptom that was highly suspected of being PHO. Case presentation: A 77-year-old woman with swelling and severe pain in the left femur for 2 months was referred to our hospital. Radiography revealed a remarkably osteogenic thickening and sclerotic lesion in her distal femur. Preliminary diagnoses of malignant bone tumor and hematological tumor were made based on laboratory test results, radiological examination, and clinical manifestation. A needle biopsy was performed, which ruled out the diagnosis of malignant bone tumors. A low-grade bone tumor was still suspected. After that, en bloc resection was performed, followed by replacement of the femur with a mega-prosthesis. Pathological analysis revealed no malignant tumor cells, and immunohistochemical staining for CDK4 and MDM2 yielded negative results. Meanwhile, pulmonary large cell carcinoma was identified on biopsy. Based on published studies, a diagnosis of atypical PHO was made according to clinical and imaging manifestations. Conclusions: This is an infrequent case of PHO with unilateral symptoms in the left femur. When clinical manifestations and radiological findings are inconsistent with the pathological results, the possibility of benign lesions with malignant clinical manifestations or imaging features should be carefully considered. Keywords: Pulmonary hypertrophic osteoarthropathy, Unilateral symptom, Low-grade osteosarcoma, Benign lesion, Clubbing Background Hypertrophic osteoarthropathy (HOA) is a rare syndrome that can be divided into two categories: primary (hereditary or idiopathic) and secondary HOA. Pulmonary hypertrophic osteoarthropathy (PHO) can be secondary to HOA, which is a paraneoplastic syndrome. Most patients are complicated by PHO secondary to intrathoracic malignancies, lung or heart diseases. PHO *Correspondence: [email protected]‑u.ac.jp Department of Orthopaedic Surgery, Kanazawa University School of Medicine, 13‑1 Takara‑machi, Kanazawa, Ishikawa 920‑8641, Japan
predominantly usually occurs in middle-aged and elderly patients. The incidence of PHO is relatively low, and prior studies have reported incidences of PHO of 0.2–17% in lung cancer patients [1–3]. Clinical symptoms of PHO usually appear symmetrically in the extremity and include clubbing, periostitis, arthritis, periosteal proliferation, and pain and swelling in the joints [4]. Radiological examination and bone scanning are the mainstays of diagnosis. Mainly, bone scint
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