Adam Razvan, Harsovescu Tudor, Tudorache Sorin, Moldovan Cosmin, Pogarasteanu Mark, Dumitru Adrian, Orban Carmen
Department of Orthopedics and Traumatology, Elias Emergency University Hospital, 011461 Bucharest, Romania.
Department of First Aid and Disaster Medicine, Titu Maiorescu University, 040051 Bucharest, Romania.
Diagnostics (Basel). 2022 Mar 23;12(4):783. doi: 10.3390/diagnostics12040783.
Rosai-Dorfman Disease (RDD), also known as sinus histiocytosis, is included in the group of rare diseases, characterized by proliferation and accumulation of histiocytes in the lymph nodes (lymphadenopathy), most often involving the cervical ganglion chains (nodal form). RDD bone involvement is rare, estimated at 10% of cases, but primary bone involvement (extranodal form), is very rare-2-8%. Usually they are solitary lesions, with multifocal primary bone manifestations being extremely rare. Histopathological analysis is of high value for a correct diagnosis. We present the case of a Caucasian woman, 42 years old, initially treated in another clinic, for an osteolytic tumor formation in the right tibial shaft. An excisional biopsy with bone trepanation was performed, the histopathological diagnosis being the chronic inflammatory tissue. The evolution was atypical, with tumor growth, extraosseous, subcutaneous. A needle biopsy was repeated in our clinic, the result being similar to the original one. Evolution of the tumor, and the radiological and imaging aspect (periosteal reaction, eroded and thin bone cortex) suggested a more aggressive disease, these being in inconsistency with the result obtained. The biopsy was repeated, as an excision type this time. The histopathological result and immunohistochemistry indicated an RDD primary bone lesion. Based on this result, and corroborated with the data from the literature, we initiated the surgical treatment, curettage and grafting with bone substitute plus safety osteosynthesis with locked plaque, the patient registering a favorable evolution. RDD primary bone lesions are in fact an atypical manifestation of a rare disease. The correct diagnosis is very difficult due to the non-specific imaging aspect. Histopathological examination errors, especially in the case of needle biopsies can lead to errors in diagnosis and treatment with negative results for the patient.
罗萨伊-多夫曼病(RDD),又称窦性组织细胞增多症,属于罕见病范畴,其特征为组织细胞在淋巴结中增殖和积聚(淋巴结病),最常累及颈淋巴结链(结节型)。RDD累及骨骼的情况罕见,估计占病例的10%,但原发性骨受累(结外型)则极为罕见,为2%-8%。通常为孤立性病变,多灶性原发性骨表现极为罕见。组织病理学分析对正确诊断具有很高价值。我们报告一例42岁白种女性病例,最初在另一家诊所接受治疗,其右胫骨干出现溶骨性肿瘤形成。进行了带骨环钻的切除活检,组织病理学诊断为慢性炎症组织。病情发展不典型,肿瘤向外生长至骨外及皮下。在我们诊所重复进行了针吸活检,结果与最初结果相似。肿瘤的发展以及放射学和影像学表现(骨膜反应、骨皮质侵蚀和变薄)提示病情更具侵袭性,这与所获结果不一致。再次进行活检,此次为切除活检。组织病理学结果和免疫组化显示为RDD原发性骨病变。基于这一结果,并与文献数据相结合,我们启动了手术治疗,即刮除术并用骨替代物植骨,外加锁定钢板安全内固定,患者病情进展良好。RDD原发性骨病变实际上是一种罕见病的非典型表现。由于影像学表现不具特异性,正确诊断非常困难。组织病理学检查错误,尤其是针吸活检的情况下,可能导致诊断和治疗错误,给患者带来不良后果。