Clinical features, diagnosis and treatment of Paget’s disease of bone in mainland China: A systematic review

Wang, Qinyi; Fu, Shanjiang; Ding, Ning; Liu, Shuying; Chen, Rong; Wen, Zhangxin; Fu, Sang; Sheng, Zhi-Feng; Ou, Yan

doi:10.1007/s11154-020-09544-x

Cited by 8 publications

(7 citation statements)

References 29 publications

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“…Table 3 compares clinical and management features of Paget's disease of bone among various countries, based on data from systemic reviews and large studies. [ 8 9 12 13 14 15 ] While men were more affected in China[ 9 ] and the United Kingdom (UK),[ 13 ] this study noted a slight predominance of the female gender. Female preponderance was also reported in Japanese[ 8 ] and Brazilian[ 16 ] studies.…”

Section: Discussionmentioning

confidence: 76%

“…[ 23 ] Calcitonin was the most commonly used specific drug in Japanese[ 8 ] and Chinese studies. [ 9 ] In this series, two patients received calcitonin and both reported aggravation of symptoms with higher alkaline phosphatase levels following treatment. Given its moderate efficacy, the use of calcitonin is mainly reserved for patients with contraindications to bisphosphonates.…”

Section: Discussionmentioning

confidence: 95%

“…Familial aggregation was observed in 26% in an Australian study,[ 7 ] 6.3% in a Japanese study,[ 8 ] and 0.3% in a recent Chinese study. [ 9 ] These findings suggest that genetic factors are important in the development of this disease. Mutations in the SQSTM1 gene were identified in sporadic and familial cases of Caucasian patients.…”

Section: Discussionmentioning

confidence: 99%

“…In a previous report about the origin of patients with Paget's disease of bone in Israel, 44% were Afro-Asians, with Tunisians representing the largest group. [ 9 ] A literature search did not shed light on the characteristics of Paget's disease of bone patients in the Maghreb. Thus, this study aimed to describe demographic, clinical, biochemical, and imaging characteristics, as well as treatment outcomes of Tunisian patients with Paget's disease of bone.…”

Section: Introductionmentioning

confidence: 99%

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Paget's disease of bone in Tunisia: A study of 69 patients

Maatallah¹,

Rahmouni²,

Miladi

et al. 2020

Indian J Endocr Metab

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Context: Paget's disease of bone is a common bone disease with a striking variation in its incidence and characteristics in different parts of the world. It is uncommonly reported in African patients. Aims: Given the lack of studies describing the characteristics of patients with Paget's disease of bone in North Africa, we aimed to describe demographic, clinical, biochemical, and imaging characteristics, as well as treatment outcomes of Tunisian patients with Paget's disease of bone. Subjects and Methods: This bicentric and retrospective study included patients with Paget's disease of bone. Clinical, laboratory, radiological profile, and response to treatment were analyzed. Results: Sixty-nine patients were identified. The mean age was 64.9 ± 11.6 years and 52.2% were women. One patient reported a positive family history. Seven patients were asymptomatic. Bone pain was the most common presenting symptom. Eight patients had a history of malignancy. In three patients, Paget's disease of bone was diagnosed as part of a metastatic workup. Monostotic disease was found in half of the cases. The most commonly involved sites were pelvis (43.5%), femur (21.7%), and spine (21.7%). The mean serum alkaline phosphatase level at presentation was 591 U/L (68–8380). Two patients received salmon calcitonin (2.8%) and 47 patients (68.1%) received bisphosphonates. After a mean follow-up of 55 months (2–240 months), bone pain improved in 43.1% of patients and the serum alkaline phosphate levels had normalized in 22 of them (43.1%). During follow-up, there was no malignant transformation. Conclusions: In this series of Tunisian patients, Paget's disease of bone had a female predominance and was usually monostotic. The clinical and radiological presentations were similar to the European series.

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Section: Discussionmentioning

confidence: 76%

Section: Discussionmentioning

confidence: 95%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Paget's disease of bone in Tunisia: A study of 69 patients

Maatallah¹,

Rahmouni²,

Miladi

et al. 2020

Indian J Endocr Metab

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“…Other presentations include fractures, osteoarthritis, bone deformities, spinal canal stenosis, and paraplegia. The skull can be affected, which can lead to an enlarged head, and compression of nerves and brain tissues may cause headache and hearing loss[ 13 ]. The majority of patients in this report had bone pain and deformities, but none had headache or hearing loss.…”

Section: Discussionmentioning

confidence: 99%

Paget’s disease of bone: Report of 11 cases

Miao¹,

Wang²,

Lyu³

et al. 2021

WJCC

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BACKGROUND Paget’s disease of bone (PDB) is a rare metabolic bone disease in China and is characterized by increased bone resorption and disorganized bone formation. The main clinical symptoms of PDB are focal or multiple bone pain and deformity with high disability. The disease has high missed diagnosis and misdiagnosis rates. This report summarizes the clinical manifestations, imaging and pathological features, and treatments of 11 patients with PDB at our hospital from 1993 to 2020 in order to improve the recognition and prognosis of PDB. CASE SUMMARY There were eight male and three female patients whose average age was 48.7 ± 11.0 years with a PDB course of 1-16 years. Nine patients had bone pain and bone deformities in different parts of the body, the majority of which involved the long bones. Laboratory examinations revealed elevated serum alkaline phosphatase (ALP) in all patients with an average of 618 ± 460 IU/L (normal range 0-130 IU/L), and serum calcium and phosphorus levels were in the normal range. Imageology showed that osteolysis was usually combined with osteosclerosis and/or bone deformities in single or multiple bones. 99m Tc-methylene diphosphonate bone scintigraphy revealed increased radionuclide uptake in the bone lesions. Six patients underwent bone tissue biopsy, and the typical pathological changes were a mosaic structure of the bone trabeculae with irregularly arranged cement lines and multinuclear osteoclasts. Ten of the 11 patients were effectively treated with bisphosphonates. CONCLUSION Early diagnosis of the rare disease PDB can be made through elevated ALP levels and typical presentations on bone X-ray and from bone tissue biopsy.

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