A case of secondary hypertrophic osteoarthropathy from medieval Tuscany (central Italy, 10th-12th centuries CE).

OBJECTIVES: This study aims to provide a detailed evaluation of a case of secondary hypertrophic osteoarthropathy (HOA) and to explore insights into the presence and consequences of disease in medieval rural Italy. MATERIALS: The skeleton of a male (US 4405) with an estimated age at death of 51-69 years excavated from the medieval rural site of Pieve di Pava (Siena, Italy). METHODS: Macroscopic and radiological (x-ray, CT) analyses were performed. RESULTS: Symmetrical extensive periosteal new bone formation on the diaphyseal and metaphyseal regions of this individual's long bones; the lower limbs were more extensively and severely affected than the upper limbs and the distal segments were more severely altered in comparison to the proximal ones. CONCLUSIONS: The macroscopic and radiological features are highly consistent with a diagnosis of secondary HOA. SIGNIFICANCE: The excellent state of preservation allowed the evaluation of rarely noted skeletal manifestations of HOA and provided insight into aspects of rural life in medieval Italy. LIMITATIONS: Molecular analysis was not successful in sequencing the aDNA of tuberculosis, therefore the underlying primary cause of secondary HOA, whether pulmonary or extrapulmonary, remains obscure in this case. SUGGESTION FOR THE FUTURE RESEARCH: It is advisable to regularly revisit the data available from osteoarchaeological collections in order to identify further cases of HOA, along with to further investigate the known cases to search for the underlying primary disease.

Massive periostosis in a child from Neolithic Gebel es-Silsileh, Egypt.

In 2015 a surprising find of human bone fragments from a child was made in a collection of the Egyptian Museum and Papyrus Collection, Berlin. These bone fragments from Southern Egypt date to 3400-3300 BC and represent the distal parts of both femora and the proximal parts of both tibiae (bones around the knee joint). The bones have a specific appearance, probably indicating a systemic disease. Due to the incomplete state of the skeletal remains, the distribution of the lesions throughout the entire skeleton could not be observed, thus preventing a better diagnosis of the underlying pathological process. The poor collagen preservation of the bone precluded aDNA testing for pathogens. The bone fragments were instead subjected to radiographic and microscopic analysis which revealed a recurrent periosteal process accompanied by a distinct osteoclastic component. A possible diagnosis might be an underlying unknown pathological process, leading to the development of a secondary Hypertrophic Osteoarthropathy (HOA).

Solitary fibrous tumour of the pleura: a rare cause of digital clubbing.

A woman in her early 70s was found to have incidental finger clubbing at a fracture clinic consultation for an unrelated problem. She reported no associated respiratory symptoms and was referred back to her General Practitioner for further investigation. A chest radiograph revealed a large left-sided mass. This was characterised as a pleural-based mass on CT, resulting in localised atelectasis and mediastinal shift. A CT guided biopsy revealed histology consistent with a solitary fibrous tumour of the pleura and the patient was referred for thoracotomy and resection.

[Bilateral periostitis and arthritis: about a case of hypertrophic osteoarthropathy]. / Périostite bilatérale et polyarthrite : à propos d'un cas d'ostéoarthropathie hypertrophique pneumique.

We report the case of a 67-year-old female patient presenting swelling of the hands and feet and pain in both legs. Clinical examination and bone scintigraphy identify the triad "digital clubbing - arthritis - bilateral periostitis of the long bones", leading to a diagnosis of hypertrophic osteoarthropathy, a syndrome usually associated with pulmonary neoplasia. The thoracic CT-scan, followed by a biopsy, effectively diagnosed a right upper lobe adenocarcinoma. Surgical treatment of the neoplasia allowed the resolution of the clinical complaints and the pathological scintigraphic findings.

Nous rapportons le cas d'une patiente de 67 ans présentant des gonflements des mains et des pieds ainsi que des douleurs des deux jambes. L'examen clinique et la scintigraphie osseuse identifient la triade «hippocratisme digital - arthrites - périostite bilatérale des os longs¼, permettant de poser un diagnostic d'ostéoarthropathie hypertrophique, un syndrome habituellement associé à une néoplasie pulmonaire. Le scanner thoracique, suivi d'une biopsie, ont en effet diagnostiqué un adénocarcinome localisé au niveau du lobe supérieur droit. La prise en charge chirurgicale de la néoplasie a permis la résolution des plaintes cliniques et de l'aspect scintigraphique pathologique.

[Surgical Treatment for Lung Cancer Associated with Hypertrophic Pulmonary Osteoarthropathy].

Hypertrophic pulmonary osteoarthropathy( HPO) is a rare paraneoplastic manifestation of lung cancer that causes joint pain, joint swelling, and limited range of motion. Two surgical cases of lung cancer with HPO are presented. Case1:A 43-year-old female was referred to our department with a diagnosis of cStage ⅡB left hilar lung cancer. She had difficulty in walking due to arthralgia caused by HPO. Left pneumonectomy was performed and the arthralgia disappeared on the first postoperative day. The patient is being well after surgery without relapse of joint symptoms. Case2:The patient was a 65-year-old male with cStage ⅡA right lung cancer. The symptoms of HPO appeared after he was found to have lung cancer. After right upper lobectomy, the arthralgia disappeared on the first postoperative day. Currently, he is receiving adjuvant chemotherapy, without relapse of joint symptoms.

Epidemiology of Digital Clubbing and Hypertrophic Osteoarthropathy: A Systematic Review and Meta-analysis.

ABSTRACT: Digital clubbing and hypertrophic osteoarthropathy (HOA) are long-standing clinical entities, but their prevalence have not been synthesized. We aimed to estimate the prevalence of digital clubbing and HOA in people with existing medical conditions.We comprehensively searched PubMed, Embase, and Web of Science to select studies addressing HOA or digital clubbing and published through March 23, 2021. Summary estimates of the prevalence were derived through random-effects meta-analysis and narrative synthesis. The review protocol has been registered with PROSPERO, CRD42021243934.Of 3973 records, we included 142 studies. In adults, the pooled prevalence of digital clubbing was 33.4% (95% confidence interval [CI], 16.6-52.8), 31.3% (95% CI, 22.4-41.1), 27% (95% CI, 9.4-49.5), and 22.8% (95% CI, 10.8-37.6) in subjects with intestinal diseases, interstitial lung diseases, infective endocarditis, and hepatic diseases, respectively. In children and adolescents, the pooled prevalence of digital clubbing was 29.1% (95% CI, 19.4-39.9), 23% (95% CI, 9.0-41.1), 19.5% (95% CI, 4.1-42.4), and 17.1% (95% CI, 9.5-26.5) in subjects with human immunodeficiency virus infection, hemoglobinopathies, cystic fibrosis, and tuberculosis. The pooled prevalence of HOA was 10.1% (95% CI, 2.0-23.1) in adults with cancers, and 5% (95% CI, 2.5-8.2) in children and adolescents with cystic fibrosis.In conclusion, the prevalence of digital clubbing varied across disease groups in both adults and children. Full-spectrum HOA was mostly reported in adults with liver disease and cancers, and in children and adolescents with cystic fibrosis.

[A Case of Hypertrophic Pulmonary Osteoarthropathy Associated with Pulmonary Pleomorphic Carcinoma].

Hypertrophic pulmonary osteoarthropathy(HPO)is a tumor-associated syndrome that features the triad of clubbed fingers, periosteal bone growth in long bones, and arthritis, and is often associated with an adenocarcinoma or squamous cell carcinoma. This report presents details of a case of HPO associated with pleomorphic carcinoma, which was relieved by treatment. A 47-year-old woman was presented with a complaint of generalized arthralgia. A physical examination showed swollen joints in the body and clubbed fingers. Chest CT revealed a mass shadow in the left upper lobe and ultrasound- guided biopsy findings led to a diagnosis of non-small cell lung cancer. Furthermore, bone scintigraphy indicated symmetrical accumulation in bones and joints throughout the body. A right upper lobectomy was performed along with combined chest wall resection and mediastinal lymph node dissection with an open chest, and the presence of lung cancer complicated with HPO was indicated. Pathological examination results revealed a diagnosis of pleomorphic carcinoma(pT4N0M0, Stage ⅢA). Systemic arthralgia was resolved on the first postoperative day. One year after surgery, a solitary brain metastasis developed and was removed, with no recurrence at the time of writing. Joint symptoms related to HPO can be expected to improve with treatment of pulmonary lesions, thus aggressive procedures for diagnosis and treatment are desirable.

Clubbed Digits Presumably Caused by Lubiprostone.

Digital clubbing has been regarded as an important sign in medicine. A 33-year-old woman with no history of hepatic, pulmonary, or malignant disease was referred to our hospital. She had been taking lubiprostone every day for three years for constipation. Clubbing in her upper and lower limb digits began gradually about two years ago. The results of laboratory investigations were almost normal. We suspected the clubbed digits were a side effect of lubiprostone and confirmed that the levels of urinary prostaglandin E2 (PGE2), which can cause clubbed digits, were elevated. Thus, we instructed the woman to stop taking lubiprostone and monitored this lab value. However, the value continued to rise over 2 months to 41.9 µg/g Cr. During that time, she had been taking sennoside A B calcium instead of lubiprostone for constipation. Since sennoside A B calcium also has the effect of increasing PGE2, we ordered the discontinuation. Her urinary PGE2 to creatinine level normalized, and the clubbing improved after the discontinuation of these two medications.