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老年血液透析患者药物相关性颌骨骨坏死并病理性骨折导致下颌骨坏死的再生:一例病例报告及文献复习

An unusual case of bone regeneration of a necrotic mandible with pathologic fracture in an elderly hemodialysis patient with medication-related osteonecrosis of the jaw: a case report and review of the literature.

机构信息

Division of Medicine, Department of Oral and Maxillofacial Surgery, Interdisciplinary Graduate School, University of Yamanashi, 1110 Shimokato, Chuo, Yamanashi, 420-3898, Japan.

Department of Emergency and Critical Care Medicine, School of Medicine, University of Yamanashi, Yamanashi, Japan.

出版信息

J Med Case Rep. 2021 Dec 23;15(1):608. doi: 10.1186/s13256-021-03206-5.

Abstract

BACKGROUND

Bisphosphonates are frequently used for osteoporosis. Medication-related osteonecrosis of the jaw, a complication of bone-modifying agents, including bisphosphonates or angiogenic inhibitors, can be challenging to treat in elderly patients with numerous preexisting conditions. Achieving good treatment outcomes is especially difficult in patients with pathological fractures accompanied with extraoral fistulae.

CASE PRESENTATION

We report an unusual case of prominent bone regeneration following palliative surgical treatment in a 72-year-old Japanese female patient undergoing hemodialysis. She previously had severe osteoporosis due to renal osteodystrophy and was receiving antiresorptive intravenous bisphosphonate. Computed tomography revealed a discontinuous left lower mandibular margin with a pathologic fracture and extensive, morphologically irregular sequestrum formation (80 × 35 × 20 mm). The patient was diagnosed with stage III medication-related osteonecrosis of the jaw and pathologic mandibular fracture. Immediately before the surgery, the anticoagulant used for dialysis was changed from heparin to nafamostat mesylate to reduce the risk of intraoperative bleeding. Sequestrectomy was performed under general anesthesia. Postoperative infection was not observed, the intraoral and submandibular fistula disappeared, and, surprisingly, prominent spontaneous bone regeneration was observed postoperatively at 6 months. Despite the severe systemic condition of the patient, the conservative surgical approach with sequestrectomy has yielded desirable results for more than 6 years since the surgery.

CONCLUSIONS

This rare report of spontaneous bone regeneration in a patient of advanced age and poor general condition is the oldest case of mandibular regeneration ever reported.

摘要

背景

双膦酸盐常用于治疗骨质疏松症。颌骨骨坏死是一种药物相关并发症,常发生于使用骨修饰药物(包括双膦酸盐或血管生成抑制剂)的患者中,对于合并多种基础疾病的老年患者,其治疗颇具挑战。对于伴有口腔外瘘的病理性骨折患者,实现良好的治疗效果尤其困难。

病例介绍

我们报告了一例罕见病例,一名 72 岁日本女性接受姑息性手术治疗后出现明显骨再生。该患者因肾性骨营养不良而患有严重骨质疏松症,并接受了静脉用抗吸收双膦酸盐治疗。计算机断层扫描显示左下颌骨连续缘有一处病理性骨折,并伴有广泛的形态不规则死骨形成(80×35×20mm)。该患者被诊断为 III 期药物相关性颌骨骨坏死和病理性下颌骨骨折。在手术前,将用于透析的抗凝剂从肝素改为甲磺酸萘莫司他,以降低术中出血的风险。在全身麻醉下进行死骨切除术。术后未观察到感染,口腔和下颌下瘘消失,令人惊讶的是,术后 6 个月观察到明显的自发性骨再生。尽管患者的全身状况严重,但采用死骨切除术的保守手术方法已取得了 6 年以上的良好效果。

结论

本报告罕见地描述了一名高龄、一般状况较差的患者出现自发性骨再生,这是有报道的下颌骨再生最年长的病例。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c14b/8697441/14884d6872dd/13256_2021_3206_Fig1_HTML.jpg

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