Journal of Prevention and Treatment for Stomatological Diseases ›› 2020, Vol. 28 ›› Issue (2): 97-101.doi: 10.12016/j.issn.2096-1456.2020.02.007

• Prevention and Treatment Practice • Previous Articles     Next Articles

Maxillofacial Rosai-Dorfman′s disease: a case report and literature review

CHEN Yi1,XIE Hongliang1(),TANG Jianming1,WENG Rulian1,FENG Li1,ZUO Min2   

  1. 1. Department of Oral and Maxillofacial Surgery, Shenzhen People′s Hospital,Shenzhen 518000,China
    2. Department of Pathology, Shenzhen People′s Hospital,Shenzhen 518000,China
  • Received:2019-06-02 Revised:2019-08-30 Online:2020-02-20 Published:2020-02-25
  • Contact: Hongliang XIE E-mail:83207882@qq.com

Abstract:

Objective To explore the clinical manifestations, histopathological features, diagnosis, treatment and prognosis of Rosai-Dorfman′s disease (RDD) in the maxillofacial region and to review the relevant literature in order to improve the understanding, diagnosis and treatment of oral and maxillofacial RDD. Methods The clinical manifestations, histopathological features, diagnosis, treatment, and prognosis of a patient with RDD in the maxillofacial region admitted to Shenzhen People′s Hospital were analyzed, and the literature was reviewed for analysis. Results The clinical manifestations were palpable masses of 3.5 cm × 2.0 cm × 1.0 cm in the right cheek and 3.0 cm × 2.0 cm × 1.0 cm in the right submaxillary area, with clear boundaries, good mobility, medium and hard textures, respectively, no tenderness, smooth surfaces, and no obvious nodules. On contrast-enhanced and plain CT scans of the maxilla and neck, a diffuse soft tissue shadow was seen in the right maxillofacial region with an unclear boundary and uniform density, and the contrast-enhanced scan also showed moderate and uniform enhancement. The primary diagnosis was right maxillofacial lesions. The tumor was resected surgically. The pathological report was right buccal and right submaxillary extranodal RDD. Under light microscopy, nodular lesions in the fibrous fat tissue were found, which were composed of light and deep staining areas. The light staining areas consisted of patchy, polygonal cells with large volumes and rich cytoplasm, in which lymphocytes and neutrophils could be seen stretching into the movement; the deep staining areas were composed of lymphocytes and plasma cells. IHC: S-100 (+), CD68 (+), CD163 (+), CD1a (-), CD21FDC (+), langerin (-), IgG (+), IgG4 (+). No recurrence was found 11 months after the operation. RDD is a rare, benign and self-limited tissue and cell disease and consists of multiple lesions in the maxillofacial region. Its imaging features are similar to those of lymphoma. Its pathological features are large volumes, rich cytoplasm and phagocytosis of lymphocytes and plasma cells. Generally, RDD only needs to be observed, and individuals with symptoms or the involvement of important organs need to be treated; the first choice for the extranodal type is drug treatment, with radiotherapy administered if the central nervous system is involved. Surgery is recommended if involvement of important organs and compression of the trachea are observed; chemotherapy should be used for diffuse RDD. Most patients with RDD experienced relapse or remission of the disease; a few patients died because of the involvement of important organs or complications. Conclusion The clinical manifestations of maxillofacial RDD vary and lack specific imaging features, and pathological immunohistochemistry is the gold standard for diagnosis. The etiology is not completely clear, the treatment methods are varied, and the prognosis is related to the involved range of the disease.

Key words: Rosai-Dorfman disease, sinus histiocytosis with giant lymphadenopathy, maxillofacial, pthology, emperipolesis, immunohistochemistry

CLC Number: 

  • R78

Figure 1

CT manifestation in the right maxillofacial region a: diffuse soft tissue shadow in the right maxillofacial region with unclear boundary and uniform density; b:moderate and uniform enhancement of the soft tissue shadow; c: soft tissue shadow up to the pterygopalatine fossa, down to the lower edge of the hyoid bone, inside the pterygoid muscle, and outside the deep parotid"

Figure 2

Pathological and immunohistochemical manifestations a, b: hyperplastic histiocytes interspersed with infiltrating lymphocytes and plasma cells with varying degrees of light and dark staining at low magnification (a HE × 40, b HE × 100), arrow refers to light and dark areas alternate; c: proliferative infiltration of tissue cells and the “extension movement” of lymphocytes at high magnification (HE × 200), arrow refers to emperipolesis; d: immunohistochemical reaction for S-100 protein was strongly positive in the cytoplasm of the histiocytes (IHC × 100), arrow refers to S-100(+)"

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