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Case Report Open Access

Multicentric Castleman disease detected during the evaluation of tongue cancer: A case report

  • Authors:
    • Yuta Yasui
    • Satoshi Shimohama
    • Manabu Yamada
    • Kenichiro Suga
    • Masafumi Nishikawa
    • Seiji Asoda
  • View Affiliations / Copyright

    Affiliations: Department of Dentistry and Oral Surgery, National Hospital Organization, Tochigi Medical Center, Utsunomiya, Tochigi 320‑0057, Japan, Department of Pathology, National Hospital Organization, Tochigi Medical Center, Utsunomiya, Tochigi 320‑0057, Japan
    Copyright: © Yasui et al. This is an open access article distributed under the terms of Creative Commons Attribution License.
  • Article Number: 563
    |
    Published online on: October 1, 2025
       https://doi.org/10.3892/ol.2025.15310
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Abstract

Castleman disease is a non‑neoplastic lymphoproliferative disorder that encompasses a variety of disease types. The current study reports a case of multicentric Castleman disease that was incidentally detected during the workup for tongue cancer. A 73‑year‑old male visited the Tochigi Medical Center (Utsunomiya, Japan) in May 2022, with the main complaint of pain on the left side of his tongue. At his initial visit, a 25x15 mm exophytic tumor with a slightly ambiguous border was observed on the left side of the patient's tongue. Positron emission tomography/computed tomography revealed numerous abnormal accumulations on the left side of his tongue as well as in the cervical and systemic lymph nodes. A tongue biopsy revealed a diagnosis of squamous cell carcinoma. Meanwhile, as a result of fine needle aspiration from the cervical lymph nodes and a biopsy of the inguinal lymph nodes, lymph node lesions were diagnosed to be a plasma cell variant of Castleman disease. Subsequently, a partial glossectomy was performed under general anesthesia, with the final histopathological diagnosis of the lingual lesions being squamous cell carcinoma as well. The progress of the patient was monitored since the symptoms of Castleman disease were mild and asymptomatic. At 30 months postoperatively, none of the lesions had progressed, and the postoperative course remained uneventful.
View Figures

Figure 1

Intraoral findings during the initial
examination. A 25×15 mm exophytic tumor with an indistinct border
was observed on the left side of the patient's tongue.

Figure 2

Histopathological findings of the
tongue biopsy specimen (hematoxylin-eosin stain). (A)
Magnification, ×100 and (B) ×200. The lesion was diagnosed as
well-differentiated squamous cell carcinoma.

Figure 3

Contrast-enhanced computed tomography
images. (A) A neoplastic lesion measuring 25×15×4 mm was observed
on the left side of the tongue. Mild enlargement was observed in
the (B) submental and (C) left submandibular lymph nodes.

Figure 4

Positron emission tomography/computed
tomography images of the neck. (A) Strong FDG accumulation was
observed on the left side of the tongue. Abnormal FDG uptake was
also seen in the (B) submental and (C) left submandibular lymph
nodes.

Figure 5

Positron emission tomography/computed
tomography images of the trunk. (A-C) Strong FDG accumulation was
observed in the bilateral axillary and intraperitoneal lymph nodes.
(A) Right axillary lymph nodes. (B) Left axillary lymph node. (C)
Intraperitoneal lymph node. (D and E) Abnormal FDG uptake was also
seen in the bilateral inguinal lymph nodes. (F) Multiple lymph
nodes showed abnormal FDG accumulation.

Figure 6

Fine needle aspiration cytology
findings of cervical lymph nodes. Red blood cells and mature
lymphocytes were found, but no atypical epithelial cells indicative
of metastasis were observed. Papanicolaou staining, magnification,
×400.

Figure 7

Histopathological findings of the left
inguinal region. (A-C) Hematoxylin-eosin staining and (D)
immunohistochemistry for CD138. (A) Magnification, ×40. Numerous
large and small lymphoid follicles were observed. (B)
Magnification, ×200. Marked plasma cell proliferation was seen
around the follicles. (C) Magnification, ×400. Diffuse
proliferation of plasma cells was observed. (D) Magnification,
×400. Marked CD138 expression indicates plasmacytic
proliferation.

Figure 8

Intraoperative photographs. (A) After
iodine staining of the lesion. (B) The surgical wound was sutured
with 4-0 absorbable sutures.

Figure 9

Histopathological findings of the
resected tongue specimen (hematoxylin-eosin stain). (A)
Magnification, ×20, (B) ×40, (C) ×100 and (D) ×200. The lesion
showed sheet-like proliferation of atypical squamous cells with
keratinization, forming large and small nests infiltrating the
superficial muscle layer.
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Copy and paste a formatted citation
Spandidos Publications style
Yasui Y, Shimohama S, Yamada M, Suga K, Nishikawa M and Asoda S: Multicentric Castleman disease detected during the evaluation of tongue cancer: A case report. Oncol Lett 30: 563, 2025.
APA
Yasui, Y., Shimohama, S., Yamada, M., Suga, K., Nishikawa, M., & Asoda, S. (2025). Multicentric Castleman disease detected during the evaluation of tongue cancer: A case report. Oncology Letters, 30, 563. https://doi.org/10.3892/ol.2025.15310
MLA
Yasui, Y., Shimohama, S., Yamada, M., Suga, K., Nishikawa, M., Asoda, S."Multicentric Castleman disease detected during the evaluation of tongue cancer: A case report". Oncology Letters 30.6 (2025): 563.
Chicago
Yasui, Y., Shimohama, S., Yamada, M., Suga, K., Nishikawa, M., Asoda, S."Multicentric Castleman disease detected during the evaluation of tongue cancer: A case report". Oncology Letters 30, no. 6 (2025): 563. https://doi.org/10.3892/ol.2025.15310
Copy and paste a formatted citation
x
Spandidos Publications style
Yasui Y, Shimohama S, Yamada M, Suga K, Nishikawa M and Asoda S: Multicentric Castleman disease detected during the evaluation of tongue cancer: A case report. Oncol Lett 30: 563, 2025.
APA
Yasui, Y., Shimohama, S., Yamada, M., Suga, K., Nishikawa, M., & Asoda, S. (2025). Multicentric Castleman disease detected during the evaluation of tongue cancer: A case report. Oncology Letters, 30, 563. https://doi.org/10.3892/ol.2025.15310
MLA
Yasui, Y., Shimohama, S., Yamada, M., Suga, K., Nishikawa, M., Asoda, S."Multicentric Castleman disease detected during the evaluation of tongue cancer: A case report". Oncology Letters 30.6 (2025): 563.
Chicago
Yasui, Y., Shimohama, S., Yamada, M., Suga, K., Nishikawa, M., Asoda, S."Multicentric Castleman disease detected during the evaluation of tongue cancer: A case report". Oncology Letters 30, no. 6 (2025): 563. https://doi.org/10.3892/ol.2025.15310
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