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A 38-Year-Old Woman With an Osteolytic Rib Lesion FREE TO VIEW

Javi Hartenstine, DO; Hope Jackson, MD; Keith Mortman, MD
Author and Funding Information

CORRESPONDENCE TO: Keith Mortman, MD, 2150 Pennsylvania Ave NW, Ste 6-305, Washington, DC 20037


Copyright 2016, American College of Chest Physicians. All Rights Reserved.


Chest. 2016;149(3):e79-e85. doi:10.1016/j.chest.2015.12.010
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Published online

A 38-year-old black woman with a medical history significant for hypertension and depression presented to the emergency department with a 2-week history of lower back pain. This visit was her second in 1 week with the same symptoms, after attaining minimal pain relief with cyclobenzaprine.

Figures in this Article

To further evaluate the patient’s back pain, the emergency department physicians obtained a CT scan of the abdomen and pelvis, which incidentally revealed a lytic lesion on the posterior portion of the right ninth rib (Fig 1). Outpatient follow-up with her primary care physician included a repeat chest radiograph and CT scan of the thorax 1 month later. These scans confirmed an osteolytic lesion with an underlying pathologic fracture that had expanded in nature (Figs 2, 3). She was then referred for thoracic surgical evaluation.

Figure 1
Figure Jump LinkFigure 1 Initial CT scan of the abdomen and pelvis in the study patient revealing an incidentally found right ninth rib lytic lesion (circle).Grahic Jump Location
Figure 2
Figure Jump LinkFigure 2 Chest radiograph confirming the presence of the lytic lesion (circle).Grahic Jump Location
Figure 3
Figure Jump LinkFigure 3 CT scan of the thorax. (A) Axial and (B) Coronal images revealing an expansile lytic lesion (circles).Grahic Jump Location

At the surgical evaluation, the patient denied recent fevers, chills, dyspnea, orthopnea, chest pain, abdominal pain, or history of trauma or malignancy. She reported a history of intermittent night sweats (which preceded this presentation) and a 4-pound weight loss over the past month. Her physical examination was notable for clear breath sounds bilaterally, mild point tenderness over the right posterior ninth rib, and no evidence of any palpable cervical or supraclavicular lymphadenopathy. Results of laboratory testing revealed a hemoglobin level of 11.1 g/dL and a WBC count of 5.07 × 103/mL (differential, 55% neutrophils, 38% lymphocytes, 4% monocytes, and 2% eosinophils). Her C-reactive protein level was elevated at 16.1 mg/L, and the erythrocyte sedimentation rate was just above the normal level at 34 mm/h. The patient’s 25-hydroxy vitamin D level was significantly reduced at 8.2 ng/mL. Total calcium, ionized calcium, and intact parathyroid hormone levels, as well as results of serum and urine protein electrophoresis, were all within normal limits.

Given the presence of an expansile, lytic rib lesion in the absence of other significant medical findings, as well as to obtain a definitive diagnosis and guide further treatment, an excisional biopsy of the ninth rib was performed. Because the patient’s symptoms were localized to the right ninth rib, additional imaging (eg, bone scan, PET scan) was not obtained. These additional imaging modalities should strongly be considered, however, to rule out other asymptomatic lesions. Although a percutaneous rib biopsy was presented as an alternative, the patient elected to proceed with the excisional biopsy.

The excised rib grossly demonstrated a poorly defined, firm lesion measuring 2 cm in greatest dimension. It appeared to arise from the medullary cavity and involved the cortex in an expansile manner, focally extending through the cortex into the attached intercostal muscle. Located within the lesion was an irregular defect spanning through the medullary cavity. These findings are consistent with the radiographic identification of a lytic lesion with pathologic fracture. At low power, histologic sections exhibited a cellular lesion circumscribed by fibrosis, arising from the medullary cavity. Associated fracture of cortical bone was seen, with no evidence of destruction (Fig 4). Within the lesion were collagen fibers and associated fibroblasts intersecting between and separating areas of mixed inflammatory cells (Fig 4). Some of these areas exhibited diffuse lymphocytic infiltrates with numerous plasma cells and prominent histiocytic cells (Fig 5). At higher power, the histiocytes were bland appearing, with abundant, pale eosinophilic cytoplasm, round to oval nuclei, vesicular chromatin, and conspicuous, central nucleoli. At closer inspection, intact inflammatory cells were identified within histiocytic cells, suggesting emperipolesis (Fig 6). Immunohistochemical staining revealed many histiocytes that stained positive for CD68 and S-100, and negatively for CD1a, including those demonstrating emperipolesis (Figs 7,8,9,10, respectively). A few histiocytes showed positivity for CD68 only (Fig 11).

Figure 4
Figure Jump LinkFigure 4 Low-power view of fracture and fibrosis with cellular infiltrate and bone destruction. (Original magnification ×4).Grahic Jump Location
Figure 5
Figure Jump LinkFigure 5 Low-power view of cellular medullary lesion and surrounding reactive fibrosis. (Original magnification ×4).Grahic Jump Location
Figure 6
Figure Jump LinkFigure 6 Medium-power view of cellular infiltrate with intervening fibrous septae. (Original magnification ×10).Grahic Jump Location
Figure 7
Figure Jump LinkFigure 7 Histiocyte demonstrating emperipolesis. (Oil emersion, original magnification ×100).Grahic Jump Location
Figure 8
Figure Jump LinkFigure 8 CD-68 immunohistochemical stain results were positive on cellular membranes of histiocytes. (Original magnification ×20).Grahic Jump Location
Figure 9
Figure Jump LinkFigure 9 S-100 immunohistochemical stain results were positive in nuclei and cytoplasm of histiocytes. (Original magnification ×40).Grahic Jump Location
Figure 10
Figure Jump LinkFigure 10 CD1a immunohistochemical stain results were negative on histiocytes. (Original magnification ×20).Grahic Jump Location
Figure 11
Figure Jump LinkFigure 11 CD68 immunohistochemical stain results were positive on cellular membranes of histiocytes. (Original magnification ×20).Grahic Jump Location

What is the diagnosis?

Diagnosis: Extranodal Rosai-Dorfman disease

Clinical Discussion

Rosai-Dorfman disease (RDD), also known as sinus histiocytosis with massive lymphadenopathy, is believed to be a relatively benign and self-limiting, nonneoplastic polyclonal proliferation of histiocytes, lymphocytes, and plasma cells. However, in patients with immune dysfunction, other predisposing conditions, or systemic involvement, the disease can cause significant morbidity and, rarely, mortality. The pathogenesis of RDD is not fully understood, but studies suggest it may be a result of immune dysfunction with possible implication of infectious agents, chronic inflammation, or autoimmune processes., RDD typically manifests in childhood and early adulthood, with most cases occurring in the second and third decades of life. A slight male predominance has been reported, as well as a higher incidence in those of black ethnicity., Most patients with RDD present with symptoms of fever and extremely large, nonpainful cervical lymphadenopathy in the setting of previously good health.

In a 1990 analysis of a database of patients with RDD, Foucar et al found that of 423 patients, 179 (42.3%) had extranodal involvement and only 33 of those patients (7.8%) had osseous involvement. Nine (2%) of those patients had osseous involvement without lymphadenopathy, as seen in the patient in our study. Since this initial database review, a few other studies have documented patients who presented with solitary or multiple bone lesions as the sole manifestations of RDD.,,,,, Reported locations of lesions isolated to bone include: skull, spine, femur, radius, ulna, metacarpals, talus, and rib., Foucar et al also noted that the most frequently affected upper airway sites were the nasal cavity (50%), pharynx (25%), paranasal sinuses (18.7%), and trachea (6.3%). Involvement of the larynx, lung, and pleura has been reported in only a few cases. Authors of a case report documenting the rare isolated involvement of pleura reported involvement of the lower respiratory tract by RDD to be relatively rare, comprising 3% of extranodal cases. Pulmonary RDD typically involves the tracheobronchial tree, with intraluminal polypoid lesions sometimes associated with airway obstruction. Diffuse interstitial or airspace involvement occurs less frequently; however, when there is extensive parenchymal involvement, the pleura may be involved. Presentation limited to the mediastinum is rare. Only seven cases of tracheal involvement have been reported; of those, five cases were subglottic or laryngeal in location and associated with compromise of the airway.

Radiologic Discussion

CT scans of the neck, chest, abdomen, and pelvis are often performed in adults presenting with diffuse lymphadenopathy, as well as those with lytic bone lesions. Visceral imaging is conducted to identify potential primary and additional metastatic lesions, which are most commonly carcinomas. The lesions of patients with isolated involvement of bone may be more commonly found incidentally as part of the evaluation for other presenting symptoms or conditions. Radiographically, bone lesions in RDD can be small and lytic, with sharply or poorly defined borders, or large with mixed lytic and sclerotic components that have cortical involvement, periosteal reaction, or soft tissue extension. RDD should be considered in the differential diagnosis of an expanding solitary bone lesion (Fig 12). Whole-body 99mTc-methylene diphosphonate bone scans have also been used to identify these sites of bone remodeling. Because extranodal sites of RDD are metabolically active, they will frequently exhibit fluorodeoxyglucose uptake on PET scanning (although one was not performed on this patient). Although the study patient did not have a bone scan or PET scan preoperatively, these imaging modalities should be strongly considered because patients with RDD can have asymptomatic lesions.

Figure 12
Figure Jump LinkFigure 12 Differential diagnoses for solitary lytic bone lesions. ABC = aneurysmal bone cyst; GI = gastrointestinal; GU = genitourinary; MAI/MAC = Mycobacterium avium-intracellulare/Mycobacterium avium complex; UPS = undifferentiated pleomorphic sarcoma (new malignant fibrous histiocytoma).Grahic Jump Location
Pathologic Discussion

Imaging in the study patient showed no evidence of other lesions, and the differential diagnoses considered were primary malignancy of bone, infectious processes, benign fibrous histiocytic lesions of bone, and histiocytic processes. Of note, atypical nuclear features have been documented in sinus histiocytes of RDD. The histologic appearance narrowed the differential to reactive and proliferative processes associated with histiocytic and granulomatous features as well as common mimickers, including: lymphoma, leukemia, plasmacytoma, melanoma, granulomatous autoimmune entities (eg, Wegener granulomatosis), sarcoidosis, juvenile xanthogranuloma, Erdheim-Chester disease, metabolic storage diseases (eg, Gaucher disease), and activated histiocytic processes (eg, Langerhans cell histiocytosis, Langerhans cell sarcoma) (Fig 12). The combination of morphologic features and the results of immunohistochemical and special staining support the diagnosis of RDD.

Clinical presentation can vary greatly, and the histomorphologic characteristics of RDD are only slightly more distinct. As such, RDD is a diagnosis of exclusion requiring thorough investigation of the patient’s medical history, laboratory findings, and imaging studies, along with pertinent negative and confirmatory immunohistochemical and special stain findings; only then can the diagnosis be made.

Because RDD may have various clinical presentations, a high degree of suspicion is needed to make its diagnosis. In the classic presentation, the differential is similar to other causes of lymphadenopathy. With regard to the unusual presentation of localized disease isolated to bone, metastatic malignancy is the most important etiology in adults to be considered in the differential (Table 1).

Table Graphic Jump Location
Table 1 Differential Diagnosis for Rosai-Dorfman Disease

Laboratory findings can involve general and specific markers of hematologic and immunologic abnormalities, including an elevated erythrocyte sedimentation rate, leukocytosis, and polyclonal hypergammaglobulinemia, as well as anemia, thrombocytopenia, and autoantibodies to hematologic, renal, and articular tissues. As reported by the patient in our study, night sweats and weight loss can also occur.

Because RDD is considered a nonmalignant disorder, treatment is typically only indicated in patients who are symptomatic, have involvement of vital organs such as the CNS, or have systemic disease. Up to 20% of cases will exhibit spontaneous regression without therapy. Surgical excision has become the mainstay of treatment for localized forms of RDD, including cutaneous, single lymph nodal areas, and CNS lesions. Most of the morbidities associated with surgical approaches have been reported with CNS involvement.,

In patients with systemic disease, steroids are the first line of treatment and may improve both nodal and extranodal disease. Radiotherapy, cryotherapy, and topical chemotherapy have been shown to have variable success rates. Systemic chemotherapy (eg, clofarabine) has been used with varying degrees of success in cases of RDD refractory to surgery or other modalities.,

The wide variability of the clinical presentation of RDD makes it difficult to diagnose, especially in cases without the typical presenting features (such as the patient in this study) of isolated extranodal involvement. Although the associated clinical laboratory findings of anemia, thrombocytopenia, and elevated erythrocyte sedimentation rate are nonspecific, they may suggest an underlying systemic response. In the case of the current patient, the somewhat subtle, nonspecific history of night sweats and modest weight loss similarly suggested a systemic response. In longer standing lesions of bone, reparative changes may obscure classic histologic findings, to such an extent in some cases that they lead to incorrect diagnosis of a fibrohistiocytic or histiocytic lesion. In such cases, one must remember RDD in the differential to obtain the necessary ancillary testing to make a correct diagnosis. Isolated skeletal involvement by RDD is rare. However, it should be considered in the differential diagnosis for lytic lesions and pathologic fractures of bone. The study patient is doing well 15 months after her surgery.

Financial/nonfinancial disclosures: None declared.

Other contributions:CHEST worked with the authors to ensure that the Journal policies on patient consent to report information were met.

Shulman S. .Katzenstein H. .Abramowsky C. .Broecker J. .Wulkan M. .Shehata B. . Unusual presentation of Rosai-Dorfman disease (RDD) in the bone in adolescents. Fetal Pediatr Pathol. 2011;30:442-447 [PubMed]journal. [CrossRef] [PubMed]
 
Foucar E. .Rosai J. .Dorfman R. . Sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease): review of the entity. Semin Diagn Pathol. 1990;7:19-73 [PubMed]journal. [PubMed]
 
Maric I. .Pittaluga S. .Dale J.K. .et al Histologic features of sinus histiocytosis with massive lymphadenopathy in patients with autoimmune lymphoproliferative syndrome. Am J Surg Pathol. 2005;29:903-911 [PubMed]journal. [CrossRef] [PubMed]
 
Rosai J. .Dorfman R.F. . Sinus histiocytosis with massive lymphadenopathy. A newly recognized benign clinicopathological entity. Arch Pathol. 1969;87:63-70 [PubMed]journal. [PubMed]
 
Al-Saad K. .Thorner P. .Ngan B.Y. .et al Extranodal Rosai-Dorfman disease with multifocal bone and epidural involvement causing recurrent spinal cord compression. Pediatr Dev Pathol. 2005;8:593-598 [PubMed]journal. [CrossRef] [PubMed]
 
Bachmann K.R. .Dragoescu E.A. .Foster W.C. . Extranodal rosai-dorfman disease presenting as incidental bone tumor: a case report. Am J Orthop (Belle Mead NJ). 2010;39:E123-E125 [PubMed]journal. [PubMed]
 
Gupta S. .Finzel K.C. .Grubber B.L. . Rosai-Dorfman disease masquerading as chronic ankle arthritis: a case report and review of the literature. Rheumatology. 2004;43:811-812 [PubMed]journal. [CrossRef] [PubMed]
 
Miyake M. .Tateishi U. .Maeda T. .Arai Y. .Sugimura K. .Hasegawa T. . Extranodal Rosai-Dorfman disease: a solitary lesion with soft tissue reaction. Radiat Med. 2005;23:439-442 [PubMed]journal. [PubMed]
 
Sundaram C. .Uppin Shantveer G. .Chandrashekar P. .Prasad V.B. .Umadevi M. . Multifocal osseous involvement as the sole manifestation of Rosai-Dorfman disease. Skeletal Radiol. 2005;34:658-664 [PubMed]journal. [CrossRef] [PubMed]
 
Forest F. .N'Guyen A.T. .Fesselet J. .et al Meningeal Rosai-Dorfman disease mimicking meningioma. Ann Hematol. 2014;93:937-940 [PubMed]journal. [PubMed]
 
Pulsoni A. .Anghel G. .Falcucci P. .et al Treatment of sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease): report of a case and literature review. Am J Hematol. 2002;69:67-71 [PubMed]journal. [CrossRef] [PubMed]
 
Purav P. .Ganapathy K. .Mallikarjuna V.S. .et al Rosai-Dorfman disease of the central nervous system. J Clin Neurosci. 2005;12:656-659 [PubMed]journal. [CrossRef] [PubMed]
 
Ali A. .Mackay D. . Rosai-Dorfman disease of the lung. Thorax. 2009;64:908-909 [PubMed]journal. [CrossRef] [PubMed]
 
Ohori P.N. .Landreneau R.J. .Thaete L. .Kane K. . Rosai-Dorfman disease of the pleura: a rare extranodal presentation. Hum Pathol. 2003;34:1210-1211 [PubMed]journal. [CrossRef] [PubMed]
 
Syed A. .Malhotra R. .Shojaee S. .Shepherd R.W. . Exophytic tracheal mass. A rare presentation of Rosai-Dorfman disease. Ann Am Thorac Soc. 2011;10:518-520 [PubMed]journal
 
Mannelli L. .Monti S. .Love J.E. .et al Primary Rosai-Dorfman disease of the bone in a patient with history of breast cancer: appearance on 99m Tc-MDP scintigraphy, CT, and X-ray. Clin Nucl Med. 2015;40:247-249 [PubMed]journal. [CrossRef] [PubMed]
 
Cooper S.L. .Chavis P.S. .Fortney J.A. .Watkins J.M. .Caplan M.J. .Jenrette J.M. 3rd. A case of orbital Rosai-Dorfman disease responding to radiotherapy. J Pediatr Hematol/Oncol. 2008;30:744-748 [PubMed]journal. [CrossRef]
 
Jabali Y. .Smrcka V. .Pradna J. . Rosai-Dorfman disease: successful long-term results by combination chemotherapy with prednisone, 6-mercaptopurine, methotrexate, and vinblastine: a case report. Int J Surg Pathol. 2005;13:285-289 [PubMed]journal. [CrossRef] [PubMed]
 
Maklad A.M. .Bayoumi Y. .Tunio M. .Alshakweer W. .Dahar M.A. .Akbar S.A. . Steroid-resistant extranodal Rosai-Dorfman disease of cheek mass and ptosis treated with radiation therapy. Case Rep Hematol. 2013;2013:428297- [PubMed]journal. [PubMed]
 

Figures

Figure Jump LinkFigure 1 Initial CT scan of the abdomen and pelvis in the study patient revealing an incidentally found right ninth rib lytic lesion (circle).Grahic Jump Location
Figure Jump LinkFigure 2 Chest radiograph confirming the presence of the lytic lesion (circle).Grahic Jump Location
Figure Jump LinkFigure 3 CT scan of the thorax. (A) Axial and (B) Coronal images revealing an expansile lytic lesion (circles).Grahic Jump Location
Figure Jump LinkFigure 4 Low-power view of fracture and fibrosis with cellular infiltrate and bone destruction. (Original magnification ×4).Grahic Jump Location
Figure Jump LinkFigure 5 Low-power view of cellular medullary lesion and surrounding reactive fibrosis. (Original magnification ×4).Grahic Jump Location
Figure Jump LinkFigure 6 Medium-power view of cellular infiltrate with intervening fibrous septae. (Original magnification ×10).Grahic Jump Location
Figure Jump LinkFigure 7 Histiocyte demonstrating emperipolesis. (Oil emersion, original magnification ×100).Grahic Jump Location
Figure Jump LinkFigure 8 CD-68 immunohistochemical stain results were positive on cellular membranes of histiocytes. (Original magnification ×20).Grahic Jump Location
Figure Jump LinkFigure 9 S-100 immunohistochemical stain results were positive in nuclei and cytoplasm of histiocytes. (Original magnification ×40).Grahic Jump Location
Figure Jump LinkFigure 10 CD1a immunohistochemical stain results were negative on histiocytes. (Original magnification ×20).Grahic Jump Location
Figure Jump LinkFigure 11 CD68 immunohistochemical stain results were positive on cellular membranes of histiocytes. (Original magnification ×20).Grahic Jump Location
Figure Jump LinkFigure 12 Differential diagnoses for solitary lytic bone lesions. ABC = aneurysmal bone cyst; GI = gastrointestinal; GU = genitourinary; MAI/MAC = Mycobacterium avium-intracellulare/Mycobacterium avium complex; UPS = undifferentiated pleomorphic sarcoma (new malignant fibrous histiocytoma).Grahic Jump Location

Tables

Table Graphic Jump Location
Table 1 Differential Diagnosis for Rosai-Dorfman Disease

References

Shulman S. .Katzenstein H. .Abramowsky C. .Broecker J. .Wulkan M. .Shehata B. . Unusual presentation of Rosai-Dorfman disease (RDD) in the bone in adolescents. Fetal Pediatr Pathol. 2011;30:442-447 [PubMed]journal. [CrossRef] [PubMed]
 
Foucar E. .Rosai J. .Dorfman R. . Sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease): review of the entity. Semin Diagn Pathol. 1990;7:19-73 [PubMed]journal. [PubMed]
 
Maric I. .Pittaluga S. .Dale J.K. .et al Histologic features of sinus histiocytosis with massive lymphadenopathy in patients with autoimmune lymphoproliferative syndrome. Am J Surg Pathol. 2005;29:903-911 [PubMed]journal. [CrossRef] [PubMed]
 
Rosai J. .Dorfman R.F. . Sinus histiocytosis with massive lymphadenopathy. A newly recognized benign clinicopathological entity. Arch Pathol. 1969;87:63-70 [PubMed]journal. [PubMed]
 
Al-Saad K. .Thorner P. .Ngan B.Y. .et al Extranodal Rosai-Dorfman disease with multifocal bone and epidural involvement causing recurrent spinal cord compression. Pediatr Dev Pathol. 2005;8:593-598 [PubMed]journal. [CrossRef] [PubMed]
 
Bachmann K.R. .Dragoescu E.A. .Foster W.C. . Extranodal rosai-dorfman disease presenting as incidental bone tumor: a case report. Am J Orthop (Belle Mead NJ). 2010;39:E123-E125 [PubMed]journal. [PubMed]
 
Gupta S. .Finzel K.C. .Grubber B.L. . Rosai-Dorfman disease masquerading as chronic ankle arthritis: a case report and review of the literature. Rheumatology. 2004;43:811-812 [PubMed]journal. [CrossRef] [PubMed]
 
Miyake M. .Tateishi U. .Maeda T. .Arai Y. .Sugimura K. .Hasegawa T. . Extranodal Rosai-Dorfman disease: a solitary lesion with soft tissue reaction. Radiat Med. 2005;23:439-442 [PubMed]journal. [PubMed]
 
Sundaram C. .Uppin Shantveer G. .Chandrashekar P. .Prasad V.B. .Umadevi M. . Multifocal osseous involvement as the sole manifestation of Rosai-Dorfman disease. Skeletal Radiol. 2005;34:658-664 [PubMed]journal. [CrossRef] [PubMed]
 
Forest F. .N'Guyen A.T. .Fesselet J. .et al Meningeal Rosai-Dorfman disease mimicking meningioma. Ann Hematol. 2014;93:937-940 [PubMed]journal. [PubMed]
 
Pulsoni A. .Anghel G. .Falcucci P. .et al Treatment of sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease): report of a case and literature review. Am J Hematol. 2002;69:67-71 [PubMed]journal. [CrossRef] [PubMed]
 
Purav P. .Ganapathy K. .Mallikarjuna V.S. .et al Rosai-Dorfman disease of the central nervous system. J Clin Neurosci. 2005;12:656-659 [PubMed]journal. [CrossRef] [PubMed]
 
Ali A. .Mackay D. . Rosai-Dorfman disease of the lung. Thorax. 2009;64:908-909 [PubMed]journal. [CrossRef] [PubMed]
 
Ohori P.N. .Landreneau R.J. .Thaete L. .Kane K. . Rosai-Dorfman disease of the pleura: a rare extranodal presentation. Hum Pathol. 2003;34:1210-1211 [PubMed]journal. [CrossRef] [PubMed]
 
Syed A. .Malhotra R. .Shojaee S. .Shepherd R.W. . Exophytic tracheal mass. A rare presentation of Rosai-Dorfman disease. Ann Am Thorac Soc. 2011;10:518-520 [PubMed]journal
 
Mannelli L. .Monti S. .Love J.E. .et al Primary Rosai-Dorfman disease of the bone in a patient with history of breast cancer: appearance on 99m Tc-MDP scintigraphy, CT, and X-ray. Clin Nucl Med. 2015;40:247-249 [PubMed]journal. [CrossRef] [PubMed]
 
Cooper S.L. .Chavis P.S. .Fortney J.A. .Watkins J.M. .Caplan M.J. .Jenrette J.M. 3rd. A case of orbital Rosai-Dorfman disease responding to radiotherapy. J Pediatr Hematol/Oncol. 2008;30:744-748 [PubMed]journal. [CrossRef]
 
Jabali Y. .Smrcka V. .Pradna J. . Rosai-Dorfman disease: successful long-term results by combination chemotherapy with prednisone, 6-mercaptopurine, methotrexate, and vinblastine: a case report. Int J Surg Pathol. 2005;13:285-289 [PubMed]journal. [CrossRef] [PubMed]
 
Maklad A.M. .Bayoumi Y. .Tunio M. .Alshakweer W. .Dahar M.A. .Akbar S.A. . Steroid-resistant extranodal Rosai-Dorfman disease of cheek mass and ptosis treated with radiation therapy. Case Rep Hematol. 2013;2013:428297- [PubMed]journal. [PubMed]
 
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