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Case Reports
. 2022 Dec 26;10(36):13364-13372.
doi: 10.12998/wjcc.v10.i36.13364.

Solitary hyoid plasmacytoma with unicentric Castleman disease: A case report and review of literature

Yan-Hui Zhang  1 , Yi-Feng He  2 , Hao Yue  3 , Yue-Ni Zhang  4 , Lei Shi  5 , Bin Jin  1 , Pin Dong  1
Affiliations
Case Reports

Solitary hyoid plasmacytoma with unicentric Castleman disease: A case report and review of literature

Yan-Hui Zhang et al. World J Clin Cases. .

Abstract

Background: Solitary plasmacytoma and unicentric Castleman disease (UCD) are rare lymphoproliferative disorders characterized by monoclonal plasma cells and a single set of locally enlarged lymph nodes, respectively.

Case summary: A 48-year-old Han Chinese man presented to our department with a neck mass and progressive foreign body sensation in his throat. 18F-FDG positron emission tomography revealed focally increased radioactivity centered around the hyoid, and computed tomography (CT) revealed osteolytic lesions. Histopathology revealed Castleman-like features and CD138/CD38-positive mature plasma cells. Systemic work-up ruled out the possibility of POEMS syndrome, lymphoma, and multiple myeloma, leading to a final diagnosis of solitary hyoid plasmacytoma with UCD. The patient underwent partial hyoid resection and selective neck dissection, followed by intensity-modulated radiotherapy. 99mTc-MDP single-photon emission computed tomography/CT reevaluation showed neither local recurrence nor distant bone metastasis at the 40-mo follow-up.

Conclusion: The diagnostic process and differential diagnosis of this rare case provided valuable educational information to clinicians.

Keywords: 18F-FDG; Case report; Castleman disease; Hyoid bone; Plasmacytoma; Positron emission tomography/computed tomography.

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Conflict of interest statement

Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.

Figures

Figure 1
Figure 1
Imaging findings during initial admission. A and C: Thickening of the left epiglottis; B and D: Arytenoid and aryepiglottic fold; E: Osteolytic lesions of the left hyoid; F: Thyroid cartilage plate; G-I: 18F-FDG centered around the hyoid body and its left greater horn (SUVmax = 8.8), and left submandibular and submental soft tissue (SUVmax = 4.8) exhibited on positron emission tomography (PET); J-L: Computed tomography (CT); M-O: PET/CT fusion; and P: PET maximum-intensity projection image demonstrated no other sites with abnormal 18F-FDG uptake.
Figure 2
Figure 2
Histopathological and immunohistochemical analysis. A and B: Hematoxylin and eosin staining revealed angiofollicular lymph node hyperplasia (200 × and 400 ×); C and D: The presence of mature plasma cells (200 × and 400 ×); E-J: Positive immunohistochemical staining for plasma cell markers: CD138, CD38, κ light chains, λ light chains, CD79α, T-cell marker CD43; K: Negative staining for B-cell marker CD20; and L: The Ki-67 positive staining.
Figure 3
Figure 3
Molecular imaging in the evaluation of local recurrence and ectopic metastasis during follow-up. A-C: 99mTc-MDP radioactivity around the hyoid exhibited on single-photon emission computed tomography (SPECT), computed tomography (CT), and SPECT/CT fusion; D: Focal bone destruction and secondary hyperosteogeny exhibited on CT bone window; E and F: No evidence of distant bone metastasis on whole-body SPECT.
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