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Rosai-dorfman disease with massive cutaneous nodule on the shoulder and back.

Ma H, Zheng Y, Zhu G, Wu J, Lu C, Lai W - Ann Dermatol (2015)

Bottom Line: Many multinucleated giant cells were found; however, caseating granulomas were not seen.Immunohistochemical staining revealed that the histiocytes were positive for S-100 protein and CD68 but negative for CD1a.The lesion showed no obvious change at the 12-month follow-up.

View Article: PubMed Central - PubMed

Affiliation: Department of Dermatology, Third Affiliated Hospital, Sun Yat-Sen University, Guangzhou, China.

ABSTRACT
Rosai-Dorfman disease is a rare, idiopathic, benign, and self-limiting histiocytic proliferative disorder. A 26-year-old man presented with a single massive cutaneous nodule (reaching 30 cm in diameter) on the left shoulder and back for 15 months. The routine hematological and biochemical tests were normal. Magnetic resonance scanning showed the lesion involved the skin, subcutaneous tissue, and subjacent muscle group, accompanied by obvious lymph node enlargement in the left part of the neck, supraclavicular fossa, and axillary fossa. The histopathology of the left cervical lymph node revealed diffuse effacement of the normal nodal architecture, with patchy chronic inflammatory cell infiltrates comprising lymphocytes and sheets of histiocytes. Some histiocytes contained lymphocytes within their pale cytoplasm. Many multinucleated giant cells were found; however, caseating granulomas were not seen. The skin and muscle biopsy specimen obtained from the back revealed infiltrating lymphocytes and histiocytes diffusely distributed in the dermis, subcutaneous tissue, and crevices of the muscle fibers. The phenomenon of emperipolesis and the presence of multinucleated giant cells were also seen. Immunohistochemical staining revealed that the histiocytes were positive for S-100 protein and CD68 but negative for CD1a. Immunophenotyping of the infiltrating lymphocytes indicated positive reactions to CD3, CD45RO, CD5, CD7, CD4, CD8 (partly), CD79a, CD20 (partly), and Ki-67 (<1%). The final diagnosis was Rosai-Dorfman disease. Owing to the extensive and deep involvement of the subcutaneous tissue and muscles, the patient did not undergo surgery to excise the massive skin nodule. The lesion showed no obvious change at the 12-month follow-up.

No MeSH data available.


Related in: MedlinePlus

(A) Massive cutaneous nodule on the left shoulder and back. (B) Axial T2 weighted magnetic resonance image (MRI) shows a mass lesion (white arrow) with extension into muscle group of the left shoulder and back. (C) Gadolinium-enhanced axial T1 weighted MRI shows an enhancing mass lesion (white arrow). (D) Coronal T2 weighted MRI shows lymph nodes enlargement in the part of supraclavicular fossa and axillary fossa (white arrows).
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Figure 1: (A) Massive cutaneous nodule on the left shoulder and back. (B) Axial T2 weighted magnetic resonance image (MRI) shows a mass lesion (white arrow) with extension into muscle group of the left shoulder and back. (C) Gadolinium-enhanced axial T1 weighted MRI shows an enhancing mass lesion (white arrow). (D) Coronal T2 weighted MRI shows lymph nodes enlargement in the part of supraclavicular fossa and axillary fossa (white arrows).

Mentions: A 26-year-old man presented with a single massive cutaneous nodule (reaching 30 cm in diameter) on the left shoulder and back that had been present for 15 months (Fig. 1A). The lesion grew in size progressively, with no pain and pruritus. An irregular, dark red patch was noted on the exterior skin. He had no symptom of fever or weight loss; however, there was little malaise when stretching the shoulder joint. There was no mucosal involvement. He was unresponsive to a 3-month course of itraconazole (0.4 g/day), administered before his initial visit to our hospital's department of dermatology. Physical examination revealed many enlarged superficial lymph nodes in both sides of the neck, axillary fossa, and groin, especially the left part. The routine hematological and biochemical tests were normal. Magnetic resonance scanning showed that the lesion was predominantly located on the shoulder and back, involving the skin, subcutaneous tissue, and subjacent muscle group (Fig. 1B, C), and was accompanied by obvious lymph node enlargement in the left part of the neck, supraclavicular fossa, and axillary fossa (Fig. 1D). Left cervical lymph node biopsy was performed, and microscopic examination revealed diffuse effacement of the normal nodal architecture with patchy chronic inflammatory cell infiltrates comprising lymphocytes and sheets of histiocytes (Fig. 2A). Some histiocytes contained lymphocytes within their pale cytoplasm, suggesting emperipolesis (Fig. 2B). Many multinucleated giant cells were found; however, caseating granulomas were not seen. The skin and muscle biopsy specimen obtained from the back revealed infiltrating lymphocytes and histiocytes diffusely distributed in the dermis (Fig. 2C), subcutaneous tissue, and crevices of the muscle fibers. The phenomenon of emperipolesis and the presence of multinucleated giant cells were also seen (Fig. 2D). Immunohistochemical staining revealed that the histiocytes were positive for S-100 protein (Fig. 2E) and CD68 (Fig. 2F) but negative for CD1a (Fig. 2G). Immunophenotyping of the infiltrating lymphocytes indicated positive reactions to CD3 (Fig. 2H), CD45RO, CD5, CD7, CD4, CD8 (partly), CD79a (Fig. 2I), CD20 (partly), and Ki-67 (<1%) (Fig. 2J). On the basis of clinical data and histopathologic findings, the diagnosis was concluded to be RDD with nodal and extranodal involvement. Owing to the extensive and deep involvement of the subcutaneous tissue and muscles, the patient did not undergo surgery to excise the massive skin nodule. Furthermore, he refused any topical and oral medicine. The massive lesion showed no obvious change at the 12-month follow-up.


Rosai-dorfman disease with massive cutaneous nodule on the shoulder and back.

Ma H, Zheng Y, Zhu G, Wu J, Lu C, Lai W - Ann Dermatol (2015)

(A) Massive cutaneous nodule on the left shoulder and back. (B) Axial T2 weighted magnetic resonance image (MRI) shows a mass lesion (white arrow) with extension into muscle group of the left shoulder and back. (C) Gadolinium-enhanced axial T1 weighted MRI shows an enhancing mass lesion (white arrow). (D) Coronal T2 weighted MRI shows lymph nodes enlargement in the part of supraclavicular fossa and axillary fossa (white arrows).
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4323606&req=5

Figure 1: (A) Massive cutaneous nodule on the left shoulder and back. (B) Axial T2 weighted magnetic resonance image (MRI) shows a mass lesion (white arrow) with extension into muscle group of the left shoulder and back. (C) Gadolinium-enhanced axial T1 weighted MRI shows an enhancing mass lesion (white arrow). (D) Coronal T2 weighted MRI shows lymph nodes enlargement in the part of supraclavicular fossa and axillary fossa (white arrows).
Mentions: A 26-year-old man presented with a single massive cutaneous nodule (reaching 30 cm in diameter) on the left shoulder and back that had been present for 15 months (Fig. 1A). The lesion grew in size progressively, with no pain and pruritus. An irregular, dark red patch was noted on the exterior skin. He had no symptom of fever or weight loss; however, there was little malaise when stretching the shoulder joint. There was no mucosal involvement. He was unresponsive to a 3-month course of itraconazole (0.4 g/day), administered before his initial visit to our hospital's department of dermatology. Physical examination revealed many enlarged superficial lymph nodes in both sides of the neck, axillary fossa, and groin, especially the left part. The routine hematological and biochemical tests were normal. Magnetic resonance scanning showed that the lesion was predominantly located on the shoulder and back, involving the skin, subcutaneous tissue, and subjacent muscle group (Fig. 1B, C), and was accompanied by obvious lymph node enlargement in the left part of the neck, supraclavicular fossa, and axillary fossa (Fig. 1D). Left cervical lymph node biopsy was performed, and microscopic examination revealed diffuse effacement of the normal nodal architecture with patchy chronic inflammatory cell infiltrates comprising lymphocytes and sheets of histiocytes (Fig. 2A). Some histiocytes contained lymphocytes within their pale cytoplasm, suggesting emperipolesis (Fig. 2B). Many multinucleated giant cells were found; however, caseating granulomas were not seen. The skin and muscle biopsy specimen obtained from the back revealed infiltrating lymphocytes and histiocytes diffusely distributed in the dermis (Fig. 2C), subcutaneous tissue, and crevices of the muscle fibers. The phenomenon of emperipolesis and the presence of multinucleated giant cells were also seen (Fig. 2D). Immunohistochemical staining revealed that the histiocytes were positive for S-100 protein (Fig. 2E) and CD68 (Fig. 2F) but negative for CD1a (Fig. 2G). Immunophenotyping of the infiltrating lymphocytes indicated positive reactions to CD3 (Fig. 2H), CD45RO, CD5, CD7, CD4, CD8 (partly), CD79a (Fig. 2I), CD20 (partly), and Ki-67 (<1%) (Fig. 2J). On the basis of clinical data and histopathologic findings, the diagnosis was concluded to be RDD with nodal and extranodal involvement. Owing to the extensive and deep involvement of the subcutaneous tissue and muscles, the patient did not undergo surgery to excise the massive skin nodule. Furthermore, he refused any topical and oral medicine. The massive lesion showed no obvious change at the 12-month follow-up.

Bottom Line: Many multinucleated giant cells were found; however, caseating granulomas were not seen.Immunohistochemical staining revealed that the histiocytes were positive for S-100 protein and CD68 but negative for CD1a.The lesion showed no obvious change at the 12-month follow-up.

View Article: PubMed Central - PubMed

Affiliation: Department of Dermatology, Third Affiliated Hospital, Sun Yat-Sen University, Guangzhou, China.

ABSTRACT
Rosai-Dorfman disease is a rare, idiopathic, benign, and self-limiting histiocytic proliferative disorder. A 26-year-old man presented with a single massive cutaneous nodule (reaching 30 cm in diameter) on the left shoulder and back for 15 months. The routine hematological and biochemical tests were normal. Magnetic resonance scanning showed the lesion involved the skin, subcutaneous tissue, and subjacent muscle group, accompanied by obvious lymph node enlargement in the left part of the neck, supraclavicular fossa, and axillary fossa. The histopathology of the left cervical lymph node revealed diffuse effacement of the normal nodal architecture, with patchy chronic inflammatory cell infiltrates comprising lymphocytes and sheets of histiocytes. Some histiocytes contained lymphocytes within their pale cytoplasm. Many multinucleated giant cells were found; however, caseating granulomas were not seen. The skin and muscle biopsy specimen obtained from the back revealed infiltrating lymphocytes and histiocytes diffusely distributed in the dermis, subcutaneous tissue, and crevices of the muscle fibers. The phenomenon of emperipolesis and the presence of multinucleated giant cells were also seen. Immunohistochemical staining revealed that the histiocytes were positive for S-100 protein and CD68 but negative for CD1a. Immunophenotyping of the infiltrating lymphocytes indicated positive reactions to CD3, CD45RO, CD5, CD7, CD4, CD8 (partly), CD79a, CD20 (partly), and Ki-67 (<1%). The final diagnosis was Rosai-Dorfman disease. Owing to the extensive and deep involvement of the subcutaneous tissue and muscles, the patient did not undergo surgery to excise the massive skin nodule. The lesion showed no obvious change at the 12-month follow-up.

No MeSH data available.


Related in: MedlinePlus