Malnas Mansoor, Dungarwal Arpit, Bhandari Hemant
Department of Orthopaedics, Bombay Hospital, New Marine Lines, Mumbai, Maharashtra, India.
J Orthop Case Rep. 2024 Oct;14(10):118-123. doi: 10.13107/jocr.2024.v14.i10.4832.
An atypical bone fracture is a complete or partial discontinuity of bone which occurs without sufficient trauma. Autoimmune polyglandular syndromes are complex conditions characterized by the simultaneous presence of at least two autoimmune-mediated endocrinopathies. These syndromes exhibit considerable heterogeneity in their manifestations and typically unfold sequentially, with a significant time gap between the onset of the initial and subsequent glandular autoimmune diseases. In this article, we report a case of bilateral subtrochanteric atypical fractures in a patient with autoimmune polyglandular syndrome. This case scenario has not been reported in the literature so far to the best of our knowledge.
A 37-year-old female patient presented at a tertiary care center in a metropolitan city with complaints of pain in both hips and the upper thigh. The patient faced difficulty walking and was unable to do so due to severe pain. Notably, there was no history of a fall or trauma. A radiographic examination revealed bilateral subtrochanteric atypical fractures. Upon further investigation, the patient was also found to be having hypothyroidism, hypoparathyroidism, pancytopenia, and an autoimmune disorder. The patient received a diagnosis of Autoimmune polyglandular syndrome with bilateral subtrochanteric atypical fractures. Treatment involved a comprehensive approach, including thyroxine for hypothyroidism, low-dose steroids, immunosuppressive agents, calcium supplementation, parathyroid hormone, and transfusion of blood and blood products. Due to the fractures being extra-articular, incomplete, and undisplaced, a conservative management approach was adopted.
The occurrence of atypical and pathological fractures, particularly when bilateral or recurrent, necessitates a comprehensive evaluation to determine the root cause. It is imperative to address the underlying etiology thoroughly in every patient. Neglecting to do so may lead to a recurrence or insufficient resolution of the pathology. In instances where two or more endocrine organs are involved, consideration should be given to autoimmune polyglandular syndrome as a potential diagnosis.
非典型骨折是指在没有足够外伤的情况下发生的骨的完全或部分连续性中断。自身免疫性多腺体综合征是一种复杂的病症，其特征是同时存在至少两种自身免疫介导的内分泌病。这些综合征在表现上具有相当大的异质性，通常呈序贯性发展，初始和后续腺体自身免疫性疾病发作之间存在显著的时间间隔。在本文中，我们报告了一例自身免疫性多腺体综合征患者发生双侧转子下非典型骨折的病例。据我们所知，这种病例情况迄今为止尚未见文献报道。
一名37岁女性患者到大城市的一家三级医疗中心就诊，主诉双髋部和大腿上部疼痛。患者行走困难，因剧痛无法行走。值得注意的是，患者无跌倒或外伤史。影像学检查显示双侧转子下非典型骨折。进一步检查发现，该患者还患有甲状腺功能减退、甲状旁腺功能减退、全血细胞减少症和一种自身免疫性疾病。该患者被诊断为患有双侧转子下非典型骨折的自身免疫性多腺体综合征。治疗采用综合方法，包括用甲状腺素治疗甲状腺功能减退、低剂量类固醇、免疫抑制剂、补钙、甲状旁腺激素以及输血和血液制品。由于骨折为关节外、不完全且无移位，故采用保守治疗方法。
非典型和病理性骨折的发生，尤其是双侧或复发性骨折，需要进行全面评估以确定根本原因。对每位患者都必须彻底解决潜在病因。忽视这一点可能导致病情复发或病理情况解决不彻底。在涉及两个或更多内分泌器官的情况下，应考虑自身免疫性多腺体综合征作为潜在诊断。
