Endocrinology and Metabolism

Case Reports

A Case of Intravenous Pamidronate-Related Osteonecrosis of the Jaw in a Patient with Waldenstrom's Macroglobulinemia.: Da Mi Lee, Mi Sun Ann, Tae Ho Kim, Tae Jin Park, Yoon Sok Chung, Joon Seong Park, Seung Il Song, Kyi Beom Lee, Hee Jae Joo; J Korean Endocr Soc. 2008;23(3):210-214. Published online June 1, 2008; DOI: https://doi.org/10.3803/jkes.2008.23.3.210

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Bisphosphonate-related osteonecrosis of the jaw (BRONJ) is a rare, but serious, side effect of bisphosphonate therapy that produces significant morbidity in affected patients. It is characterized by poor wound healing and spontaneous intra-oral soft tissue breakdown, which lead to exposure of necrotic maxillary and mandibular bone.

Citations

Citations to this article as recorded by

Dental sinus tracts mimicking scrofuloderma: cutaneous presentation of underlying bisphosphonate-related osteonecrosis of the jaw
H.-S. Kang, J.-W. Lee, J.-S. Kim
Clinical and Experimental Dermatology.2011; 36(6): 624. CrossRef

A Case of Type I Osteogenesis Imperfecta Differentially Diagnosed as a Cause of a Spinal Compression Fracture.: Sang Youl Rhee, Soo Young Moon, Suk Chon, In Kyung Jeong, Seungjoon Oh, Kyu Jeung Ahn, Ho Yeon Chung, Jeong Taek Woo, Sung Woon Kim, Young Seol Kim, Jin Woo Kim; J Korean Endocr Soc. 2007;22(6):446-452. Published online December 1, 2007; DOI: https://doi.org/10.3803/jkes.2007.22.6.446

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Abstract PDF: Osteogenesis imperfecta (OI) is a genetic disease that is caused by a synthetic anomaly of type I collagen. It is characterized by such features as low bone density, multiple fractures, bone deformities and chronic bone pain. According to the hereditary pattern and degree of phenotypical expression, it also has various extraskeletal manifestations such as blue sclera, hearing deformities and dentinogenesis imperfecta. Recently, an expanded seven subgroup classification of OI has been suggested by means of its clinical severity and mutational characteristics. However, most of the OI cases reported in Korea have been classified as type II or III that can be diagnosed easily and present with severe clinical manifestations. Only rare type I OI cases have been currently reported in Korea. Herein, we report a case of type I OI that was differentially diagnosed as a cause of a spinal compression fracture.

Original Articles

Effects of Pamidronate Treatment on Osteogenesis Imperfecta.: Seung Won Lee, Hyon J Kim, Jae Hyun Cho, Hyoung Suk Lee, Youn Mu Jung, Dae Jung Kim, Kwan Woo Lee, Yoon Sok Chung; J Korean Endocr Soc. 2004;19(5):485-491. Published online October 1, 2004

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Abstract PDF: BACKGROUND
Osteogenesis imperfecta (OI) is a congenital disorder of type I collagen, with variable phenotypes, due to increased bone fragility and low bone mass. Previous pharmacological treatments for OI have been attempted with calcitonin and growth hormone but with little beneficial effects. Recently, Glorieux reported the beneficial effects of bisphosphonates in OI. METHODS: In this study, the effects of pamidronate treatment were evaluated in 9 patients with OI. All patients received intravenous pamidronate infusions, which was dose adjusted according to the patients' age. The outcome measures included the biochemical bone markers; serum alkaline phosphatase, urine deoxy-pyridinoline, urine Ca/Cr ratio, and bone mineral density (BMD). RESULTS: Serum alkaline phosphatase, urine deoxypyridinoline, and urine Ca/Cr ratio were slightly decreased after 1 year of therapy, although these changes were not statistically significant. The BMDs of the lumbar spine and proximal femur were significantly increased after 1-year of pamidronate treatment. No fractures were reported during the 1 year treatment periods. CONCLUSION: Pamidronate treatment had an effect on the BMD in osteogenesis imperfecta, probably due to decreasing bone resorption

Cyclic Pamidronate Infusion in Primary Osteoporotic Women.: Bong Nam Chae, Eun Gyoung Hong, Seone Kyu Lee, Yoon Sok Chung, Kwan Wook Lee, Hyeon Man Kim; J Korean Endocr Soc. 2001;16(2):221-230. Published online April 1, 2001

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Abstract PDF: BACKGROUND
Bisphosphonates are now well established as successful antiresorptive agents for the prevention and treatment of osteoporosis. We investigated the effect of cyclic intravenous treatment with an aminobisphosphonate, pamidronate in cases of primary osteoporosis. METHODS: Eighteen patients with primary osteoporosis (bone mineral density BMD t-score < -2.5) received four courses of pamidronate (30 mg with 500 mL normal saline over 2 hours every 3 months). The serum biochemical parameters and bone turnover markers were measured before each treatment. The bone pain score, medication score, and the side effects were also monitored. BMD and simple spine X-ray were performed before and 1 year after of treatment. RESULTS: BMD at the lumbar spine (L2-4) significantly increased from 0.798+/-0.110 g/cm2 to 0.860+/-0.107 g/cm2 after 1 year of treatment with pamidronate: by +8.3+/-9.4% of baseline. BMDs at the femoral neck, Ward s triangle and the trochanter also increased, but not significantly. Serum total alkaline phosphatase (p<0.05) and urine deoxypyridinoline/creatinine (p=0.069) decreased with treatment. Other bone turnover markers were unchanged. The bone pain score decreased significantly. None of the patients experienced a new fracture during treatment. The frequency of the side effects following the first infusion was 61.1% (a transient fever and myalgia with flu-like symptoms in 10 patients and mild phlebitis in 1 patient). However, only two patients complained of flu-like symptoms after second infusion, and no patient complained following the third infusion. CONCLUSION: Cyclic intravenous treatment of pamidronate every three months was effective in increasing BMD and in the decreasing bone turnover rate, and was relatively well tolerated in primary osteoporotic women.

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