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A case of autoimmune hepatitis and bisphosphonate-related osteonecrosis of the jaw.

de Boer YS, Bouma G, Wattjes MP, Lips P, Mulder CJ, van Nieuwkerk CM - Case Rep Gastroenterol (2012)

Bottom Line: BRONJ is an uncommon complication of BP treatment regimes that occurs at increased frequency in the presence of other risk factors, including chronic inflammatory conditions.Our patient suffered from a severe and complicated clinical course of BRONJ which, despite adequate therapy, resulted in death of the patient.Here we discuss the risk factors for the development and clinical course of BRONJ in AIH and the implications for management of these patients.

View Article: PubMed Central - PubMed

Affiliation: Department of Gastroenterology and Hepatology, VU University Medical Center, Amsterdam, The Netherlands.

ABSTRACT
Autoimmune hepatitis (AIH) is a chronic inflammatory liver disease of unknown aetiology usually requiring long-term immunosuppressive therapy. We present the case of an AIH patient who received long-term corticosteroids and azathioprine. As treatment for concomitant osteoporosis she was also treated with potent intravenous bisphosphonate (BP). This treatment was complicated by the development of BP-related osteonecrosis of the jaw (BRONJ). BRONJ is an uncommon complication of BP treatment regimes that occurs at increased frequency in the presence of other risk factors, including chronic inflammatory conditions. Our patient suffered from a severe and complicated clinical course of BRONJ which, despite adequate therapy, resulted in death of the patient. Here we discuss the risk factors for the development and clinical course of BRONJ in AIH and the implications for management of these patients.

No MeSH data available.


Related in: MedlinePlus

Spiral computed tomography in the bone window setting of the mandible after sequestrectomy and tooth extraction. Note the bilateral cortical bone defects (arrows) suggestive of multifocal osteonecrosis in the mandibular body in the multiplanar reconstructions in axial (a), coronal (b) and sagittal (c) orientations.
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Figure 1: Spiral computed tomography in the bone window setting of the mandible after sequestrectomy and tooth extraction. Note the bilateral cortical bone defects (arrows) suggestive of multifocal osteonecrosis in the mandibular body in the multiplanar reconstructions in axial (a), coronal (b) and sagittal (c) orientations.

Mentions: After 3 years of intravenous BP therapy she developed malnutrition due to eating difficulties as a result of severe pain in the jaw. She was therefore admitted to our hospital. Examination of the jaw revealed that this was due to an infected necrotic mandibular bone, which had an open connection with the oral cavity. Computed tomography later confirmed multifocal osteonecrosis of the mandible (fig. 1, fig. 2). Given the BP use and the absence of any known other cause she was diagnosed with BRONJ and therefore subsequently treated with analgesics and prophylactic antibiotics. This did not prevent progression of the osteonecrotic infection, from which Pseudomonas aeruginosa and Candida albicans were cultured. She subsequently underwent repeated mandibular sequestrectomy and tooth extraction. Despite intense therapy she eventually succumbed to respiratory failure due to disseminated C. albicans pneumonia.


A case of autoimmune hepatitis and bisphosphonate-related osteonecrosis of the jaw.

de Boer YS, Bouma G, Wattjes MP, Lips P, Mulder CJ, van Nieuwkerk CM - Case Rep Gastroenterol (2012)

Spiral computed tomography in the bone window setting of the mandible after sequestrectomy and tooth extraction. Note the bilateral cortical bone defects (arrows) suggestive of multifocal osteonecrosis in the mandibular body in the multiplanar reconstructions in axial (a), coronal (b) and sagittal (c) orientations.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC3376336&req=5

Figure 1: Spiral computed tomography in the bone window setting of the mandible after sequestrectomy and tooth extraction. Note the bilateral cortical bone defects (arrows) suggestive of multifocal osteonecrosis in the mandibular body in the multiplanar reconstructions in axial (a), coronal (b) and sagittal (c) orientations.
Mentions: After 3 years of intravenous BP therapy she developed malnutrition due to eating difficulties as a result of severe pain in the jaw. She was therefore admitted to our hospital. Examination of the jaw revealed that this was due to an infected necrotic mandibular bone, which had an open connection with the oral cavity. Computed tomography later confirmed multifocal osteonecrosis of the mandible (fig. 1, fig. 2). Given the BP use and the absence of any known other cause she was diagnosed with BRONJ and therefore subsequently treated with analgesics and prophylactic antibiotics. This did not prevent progression of the osteonecrotic infection, from which Pseudomonas aeruginosa and Candida albicans were cultured. She subsequently underwent repeated mandibular sequestrectomy and tooth extraction. Despite intense therapy she eventually succumbed to respiratory failure due to disseminated C. albicans pneumonia.

Bottom Line: BRONJ is an uncommon complication of BP treatment regimes that occurs at increased frequency in the presence of other risk factors, including chronic inflammatory conditions.Our patient suffered from a severe and complicated clinical course of BRONJ which, despite adequate therapy, resulted in death of the patient.Here we discuss the risk factors for the development and clinical course of BRONJ in AIH and the implications for management of these patients.

View Article: PubMed Central - PubMed

Affiliation: Department of Gastroenterology and Hepatology, VU University Medical Center, Amsterdam, The Netherlands.

ABSTRACT
Autoimmune hepatitis (AIH) is a chronic inflammatory liver disease of unknown aetiology usually requiring long-term immunosuppressive therapy. We present the case of an AIH patient who received long-term corticosteroids and azathioprine. As treatment for concomitant osteoporosis she was also treated with potent intravenous bisphosphonate (BP). This treatment was complicated by the development of BP-related osteonecrosis of the jaw (BRONJ). BRONJ is an uncommon complication of BP treatment regimes that occurs at increased frequency in the presence of other risk factors, including chronic inflammatory conditions. Our patient suffered from a severe and complicated clinical course of BRONJ which, despite adequate therapy, resulted in death of the patient. Here we discuss the risk factors for the development and clinical course of BRONJ in AIH and the implications for management of these patients.

No MeSH data available.


Related in: MedlinePlus