Extranodal Rosai–Dorfman Disease as Isolated Lesion of the Tibia Diagnosed by Fine-Needle Aspiration Cytology

BACKGROUND We hypothesize that lymphadenopathy and extraosseous manifestation affect the management protocol and our aim is to aid in development of standardized diagnosis and treatment protocols. METHODS PubMed and Google Scholar were searched from January 2012 through December 2022 using keywords Rosai-Dorfman, Rosai and Dorfman, and bone. Out of 98 researches, meta-analysis of 51 full-text studies involving 53 individuals with osseous RDD was conducted. RESULTS A significant association was found between lymphadenopathy with steroid therapy (OR: 20.125, 95%CI: 3.02, 134.09, P=0.0004). Extraosseous RDD showed significant association with steroid therapy (P=0.0145), T1-isointense MRI (OR: 0.173, 95%CI: 0.0304, 0.986, P=0.035), T1-hyperintense MRI (OR: 0.16, 95%CI: 0.0314, 0.815, P=0.019) and T2-hyperintense MRI (OR: 0.115, 95%CI= 0.0206, 0.6456, P=0.007). CONCLUSION Osseous RDD is rare and lacks standardized diagnosis and treatment protocols. Patients with lymphadenopathy were 9 times more likely to receive steroids, and those with extraosseous manifestations were less likely to undergo MRI. RDD is often not initially considered in differential diagnoses, leading to worsened conditions. We propose that further testing is required in affected individuals to develop standardized protocols.

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Osseous Rosai-Dorfman disease of tibia in children: A case report

Vithran¹,

Wang²,

Xiang³

et al. 2021

WJCC

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BACKGROUND Rosai-Dorfman disease (RDD), or sinus histiocytosis with massive lymphadenopathy, is a benign histiocytic disorder. Extranodal involvement is common, occurring in > 40% of patients, but bone involvement occurs in < 10% of cases. In addition, primary bone RDD is extremely rare. The majority of patients are adolescents and young adults, and the mean age at onset is 20-years-old. CASE SUMMARY We report an 8-year-old Chinese girl who presented to our hospital with an insidious onset of swelling and pain in the middle shaft of her right tibia for 4 mo. We performed total surgical resection of the right tibia lesion and allograft transplantation. A good prognosis was confirmed at the 6 mo follow-up. Pain and swelling symptoms were totally relieved, range of motion of her right knee and ankle returned to normal, and there was no clinical evidence of lesion recurrence at last follow up. Our case is the second reported case of osseous RDD without lymphadenopathy in the shaft of the tibia of a child. CONCLUSION Extranodal RDD is a rare disease and can be misdiagnosed easily. Lesion resection and allograft transplantation are an option to treat extranodal RDD in children with good short term result. Pediatric orthopedist should be aware of this rare disease, especially extranodal involvement.

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Extranodal Rosai–Dorfman Disease as Isolated Lesion of the Tibia Diagnosed by Fine-Needle Aspiration Cytology

Cited by 5 publications

References 8 publications

Fine-needle aspiration specimens of 3 cases of intra-abdominal Rosai-Dorfman disease with comprehensive review of the literature

Fine-needle aspiration specimens of 3 cases of intra-abdominal Rosai-Dorfman disease with comprehensive review of the literature

Manifestations, Diagnosis and Treatment of Osseous Rosai-Dorfman Disease: A Systematic Review

Osseous Rosai-Dorfman disease of tibia in children: A case report

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