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骨质石化症。当前的临床考量

Osteopetrosis. Current clinical considerations.

作者信息

Shapiro F

机构信息

Department of Orthopaedic Surgery, Children's Hospital, Boston, MA 02115.

出版信息

Clin Orthop Relat Res. 1993 Sep(294):34-44.

PMID:8358940
Abstract

Osteopetrosis is an inherited skeletal condition characterized by increased bone radiodensity. There are three clinical groups: infantile-malignant autosomal recessive, fatal within the first few years of life (in the absence of effective therapy); intermediate autosomal recessive, appears during the first decade of life but does not follow a malignant course; and autosomal dominant, with full-life expectancy but many orthopaedic problems. The infantile variant shows a myelophthisic anemia, granulocytopenia, and thrombocytopenia, and patients eventually die from infection or bleeding or both. Neurologic sequelae include cranial nerve compression (optic nerve, blindness; auditory nerve, deafness; facial nerve, paresis), hydrocephalus, convulsions, and mental retardation. Radiographs show uniform bone density without corticomedulary demarcation, broadened metaphyses, "bone within a bone" or endobone phenomena (tarsals, carpals, phalanges, vertebra, ilium), and thickened growth plates if there is superimposed rickets. Transverse pathologic fractures occur, often followed by massive periosteal bone formation. Computed tomographic scans, magnetic resonance imaging, and bone scans provide specific information. Iliac crest bone biopsy is valuable to quantitate osteoclast and marrow changes by light and electron microscopy. Medical treatments involve high-dose calcitriol to stimulate osteoclast differentiation and bone marrow transplantation to provide monocytic osteoclast precursors. Orthopaedic problems in the intermediate and autosomal dominant forms include increased fractures, coxa vara, long-bone bowing, hip and knee degenerative arthritis, and mandibular and long-bone osteomyelitis. Cranial nerve compression also occurs. Osteotomy, plating, intramedullary rodding, and joint arthroplasty can be done, but are difficult because of bone hardness.

摘要

骨硬化症是一种遗传性骨骼疾病,其特征是骨放射密度增加。有三个临床类型:婴儿恶性常染色体隐性遗传型,在生命的最初几年内致命(若无有效治疗);中间型常染色体隐性遗传型,出现在生命的第一个十年,但不呈恶性病程;以及常染色体显性遗传型,预期寿命正常,但有许多骨科问题。婴儿型变体表现为骨髓化生贫血、粒细胞减少和血小板减少,患者最终死于感染或出血或两者兼有。神经后遗症包括颅神经受压(视神经,失明;听神经,耳聋;面神经,麻痹)、脑积水、惊厥和智力迟钝。X线片显示骨密度均匀,无皮质骨髓分界,干骺端增宽,“骨中骨”或内生骨现象(跗骨、腕骨、指骨、椎骨、髂骨),如果合并佝偻病则生长板增厚。会发生横向病理性骨折,常伴有大量骨膜骨形成。计算机断层扫描、磁共振成像和骨扫描可提供具体信息。髂嵴骨活检通过光镜和电镜对破骨细胞和骨髓变化进行定量很有价值。医学治疗包括大剂量骨化三醇以刺激破骨细胞分化和骨髓移植以提供单核破骨细胞前体。中间型和常染色体显性遗传型的骨科问题包括骨折增加、髋内翻、长骨弯曲、髋和膝关节退行性关节炎以及下颌骨和长骨骨髓炎。也会发生颅神经受压。可以进行截骨术、钢板固定、髓内棒固定和关节置换术,但由于骨质坚硬而困难。

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2
Spontaneous Anterior Arch Fracture of the Atlas Following C1 Laminectomy in a Patient With Osteopetrosis: A Case With Five Years of Follow-Up.一名患有骨硬化症的患者在C1椎板切除术后发生寰椎自发性前弓骨折:一例五年随访病例
Cureus. 2025 Feb 22;17(2):e79471. doi: 10.7759/cureus.79471. eCollection 2025 Feb.
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Genetic testing confirmed osteopetrosis with initial presentation of nystagmus.
基因检测确诊为骨质石化症,最初表现为眼球震颤。
Taiwan J Ophthalmol. 2023 Apr 28;14(3):437-440. doi: 10.4103/tjo.TJO-D-22-00152. eCollection 2024 Jul-Sep.
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