下颌骨缺血性坏死是未确诊的急性髓性白血病的最初表现

IF 1.9 3区医学 Q2 DENTISTRY, ORAL SURGERY & MEDICINE

Oral Surgery Oral Medicine Oral Pathology Oral Radiology Pub Date : 2025-07-21 DOI:10.1016/j.oooo.2025.04.012

Paolo Junior Fantozzi , Zaid Khoury , Gianluca Tenore , Domenico Gaglioti , Federica Bruno , Cira Rosaria Tiziana Di Gioia , Umberto Romeo , Alessandro Villa , Ahmed Sultan

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引用次数: 0

摘要

背景：骨血管性坏死（AVN）是一种罕见但严重的并发症，与代谢性骨疾病、放射治疗以及实体/血液恶性肿瘤的长期治疗相关。白血病患者很少出现AVN，大多数病例发生于急性淋巴细胞白血病（ALL）的皮质类固醇治疗或慢性髓性白血病（CML）的IFN-α/TKI治疗。在此，我们报告一例AVN作为未确诊的急性髓性白血病（AML）的初始表现，在其他方面健康的患者，以前没有放疗或抗吸收治疗。病例报告：一名37岁女性，18 ~ 19颗牙间有疼痛的坏死性牙龈溃疡1周病史。口腔细菌和病毒培养均为阴性。随访1周，溃疡恶化伴骨隔离。重复细菌培养结果推定为坏死性牙周炎（NP）。然后获得带差异的CBC、HIV-RNA测试和下颌骨CBCT。活检，组织病理学检查显示坏死的不能活的骨屑，内衬细菌菌落和炎症细胞。HIV-RNA检测阴性；全血细胞计数显示中性粒细胞减少和贫血，而外周血涂片显示胚细胞占52%的有核细胞。然后将患者转到血液学/肿瘤学进行进一步评估。骨髓抽吸显示母细胞胞质颗粒大，颗粒增生异常的不同阶段。免疫表型分析证实存在CD117、CD34、HLADR、CD15、CD45、CD38和CD4；细胞遗传学分析显示7号染色体长臂（q22-q36）间质性缺失。因此，诊断为急性髓性白血病。结论np和AVN可能是免疫系统损伤导致的潜在未确诊AML的警告信号。虽然罕见，但对于这种口头表现，AML应纳入鉴别诊断。对类似病例的全面检查对于疾病的早期发现至关重要。

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Avascular necrosis of the mandible as the initial presentation of undiagnosed acute myelogenous leukemia

Background

Avascular Necrosis of the Bone (AVN) is a rare, yet a serious complication associated with metabolic bone disorders, radiation therapy as well as long-term treatment of solid/hematologic malignancies. Patients with leukemia rarely present with AVN, with most cases occurring due to corticosteroid treatment in acute lymphoblastic leukemia (ALL) or following IFN-α/TKI therapy in chronic myeloid leukemia (CML). Herein we present a case of AVN as the initial presentation of undiagnosed acute myelogenous leukemia (AML) in an otherwise healthy patient without previous radiation or antiresorptive therapy.

Case Report

A 37-year-old female presented with a 1-week history of painful necrotizing gingival ulceration interdentally between teeth #18 and 19. Oral bacterial and viral cultures were negative. At 1-week follow-up, there was a worsening of the ulceration with bony sequestration. Repeat bacterial culture results were presumptive of necrotizing periodontitis (NP). CBC with differential, HIV-RNA tests, and a mandibular CBCT were then obtained. A biopsy was made, and histopathologic examination revealed necrotic nonviable bone sequestrum lined by bacterial colonies and inflammatory cells. HIV-RNA test was negative; CBC demonstrated neutropenia and anemia whereas a peripheral blood smear showed blasts accounting for 52% of nucleated cells. Patient was then referred to hematology/oncology for further evaluation. Bone marrow aspirate showed blasts with large cytoplasmic granules with different stages of dysgranulopoiesis. Immunophenotyping confirmed the presence of CD117, CD34, HLADR, CD15, CD45, CD38, and CD4; cytogenetic analysis showed interstitial deletion of the long arm of chromosome 7 (q22-q36). Hence, a diagnosis of AML was made.

Conclusion

NP and AVN may represent a warning sign of an underlying undiagnosed AML as a consequence of an immune-system impairment. Although rare, AML should be included in the differential diagnosis for such oral presentations. Comprehensive workup for similar cases is of paramount importance for early detection of the disease.

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来源期刊

Oral Surgery Oral Medicine Oral Pathology Oral Radiology DENTISTRY, ORAL SURGERY & MEDICINE-

CiteScore

3.80

自引率

6.90%

发文量

1217

审稿时长

2-4 weeks

期刊介绍： Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology is required reading for anyone in the fields of oral surgery, oral medicine, oral pathology, oral radiology or advanced general practice dentistry. It is the only major dental journal that provides a practical and complete overview of the medical and surgical techniques of dental practice in four areas. Topics covered include such current issues as dental implants, treatment of HIV-infected patients, and evaluation and treatment of TMJ disorders. The official publication for nine societies, the Journal is recommended for initial purchase in the Brandon Hill study, Selected List of Books and Journals for the Small Medical Library.