Unicystic Castleman disease presenting in the neck: a case report and review of the literature
Georgios Pantelas 1,2, Marios Salloumis 1, Maria Myrto Solomou 2, Yiannis Pistolas 2, Markos Lillis 3
1 Nicosia General Hospital – Department of Oral and Maxillofacial Surgery, 2 European University Cyprus – School of Dentistry, 3 Nicosia General Hospital – Department of Anatomical Pathology – Histopathology
DOI: 10.54936/haoms243151162
SUMMARY: Background: Castleman’s disease (CD) is an uncommon hyperplastic disorder of lymphoid tissue, clinically categorized into unicentric CD (UCD) and multicentric CD (MCD), contingent upon the number of lymph node engagement. Unicentric Castleman disease (UCD) manifests as a solitary lump, predominantly found in the mediastinum, and infrequently in the head and neck region. Unlike multicentric Castleman disease (MCD), which displays peripheral lymph node enlargement and a multitude of systemic indications, UCD usually lacks generalized symptoms. Methods: We present a case of UCD presenting as a neck mass, in a 55-year-old woman as well as a detailed literature review for Castleman’s disease. The patient had undergone surgical excision of the mass. Results: The diagnostic findings following Immunohistochemistry were consistent with Hyaline vascular type Castleman’s disease. The patient remains stable with no signs or symptoms of recurrent disease after the mass removal. Conclusion: It is crucial to include Castleman’s’ disease in the differential diagnoses of lymphadenopathies due to the fact that it can be clinically mistaken for other common causes of lymphadenopathy such as lymphoma, tuberculosis, and nodal metastases.
KEY WORDS: Castleman’s disease; Case report, Unicentric Castleman, Surgical Treatment, Diagnosis
REFERENCES
1. van Rhee F, Oksenhendler E, Srkalovic G, et al. International evidence-based consensus diagnostic and treatment guidelines for unicentric Castleman disease. Blood Advances. 2020;(23):6039-6050. doi:10.1182/bloodadvances.2020003334
2. Zhang L, Dong Y, Peng H, et al. A national, multicenter, retrospective study of Castleman disease in China implementing CDCN criteria. The Lancet Regional Health - Western Pacific. Published online May 2023:100720. doi:10.1016/j.lanwpc.2023.100720
3. Paparella MT, Popolizio T, Grasso D, Serricchio EA, Guglielmi G. Castleman disease: a rare case in a young woman. Acta Biomedica Atenei Parmensis. 2023;(S1):e2023058. doi:10.23750/abm.v94iS1.13795
4. Simpson D. Epidemiology of Castleman Disease. Hematology/Oncology Clinics of North America. 2018;(1):1-10. doi:10.1016/j.hoc.2017.09.001
5. Dispenzieri A, Fajgenbaum DC. Overview of Castleman disease. Blood. 2020;(16):1353-1364. doi:10.1182/blood.2019000931
6. Mitsos S, Stamatopoulos A, Patrini D, George R, Lawrence D, Panagiotopoulos N. The Role of Surgical Resection in Unicentric Castleman’s Disease: A Systematic Review. Advances in Respiratory Medicine. 2018;(1):36-43. doi:10.5603/arm.2018.0008
7. Saadallah MAH. Castleman’s disease: A rare case report and review of literature. International Journal of Surgery Case Reports. Published online June 2022:107282. doi:10.1016/j.ijscr.2022.107282
8. Mremi A, Ndale E, Stephen L, Mkwizu E, Kilonzo K. Castleman’s disease: A report of two cases at a tertiary hospital in Northern Tanzania. SAGE Open Medical Case Reports. Published online January 2023:2050313X2311757. doi:10.1177/2050313x231175720
9. Lomas OC, Streetly M, Pratt G, et al. The management of Castleman disease. British Journal of Haematology. 2021;(3):328-337. doi:10.1111/bjh.17688
10. Oksenhendler E, Boutboul D, Fajgenbaum D, et al. The full spectrum of Castleman disease: 273 patients studied over 20 years. British Journal of Haematology. 2017;(2):206-216. doi:10.1111/bjh.15019
11. Maqbool S, Javed A, Idrees T, Anwar S. Unicentric Castleman Disease: A Rare Diagnosis of Radiological and Histological Correlation. Indian Journal of Otolaryngology and Head & Neck Surgery. 2022;(2):975-978. doi:10.1007/s12070-022-03253-4
12. Marcu RD, Spinu AD, Socea B, et al. Castleman’s Disease - Clinical, Histological and Therapeutic Features. Revista de Chimie. 2018;(4):823-830. doi:10.37358/rc.18.4.6208
13. Wang T, Chen X, Chen W, Shi L, Liu J. A retrospective study of 44 patients with head and neck Castleman’s disease. European Archives of Oto-Rhino-Laryngology. 2021;(5):2625-2630. doi:10.1007/s00405-021-07065-0
14. Gündüz E, Özdemir N, Bakanay ŞM, Karakuş S. A Rare Lymphoproliferative Disease: Castleman Disease. Turkish Journal of Hematology. Published online October 28, 2021. doi:10.4274/tjh.galenos.2021.2021.0440
15. Sun X, Du Y, Zhang Y, Wang R, Hou D. Unicentric Castleman disease: multidetector computed tomography classification with surgical and pathologic correlation. Quantitative Imaging in Medicine and Surgery. 2021;(8):3562-3568. doi:10.21037/qims-20-1033
16. Molacek J, Treska V, Skalicky T, et al. Unicentric form of Castleman´s disease, pitfalls of diagnosis and surgical treatment. Frontiers in Oncology. Published online January 30, 2023. doi:10.3389/fonc.2023.1057683
17. Qian S, Ding M, Hou H, et al. Clinical and Molecular Characteristics of 60 Patients With Human Immunodeficiency Virus-Negative Castleman Disease. Frontiers in Immunology.Published online May 17, 2022. doi:10.3389/fimmu.2022.899073
18. Garcia S, Fan J. Castleman Disease Presenting as a Mediastinal Mass. Cureus. Published online September 30, 2021. doi:10.7759/cureus.18409
1 Nicosia General Hospital – Department of Oral and Maxillofacial Surgery, 2 European University Cyprus – School of Dentistry, 3 Nicosia General Hospital – Department of Anatomical Pathology – Histopathology
DOI: 10.54936/haoms243151162
SUMMARY: Background: Castleman’s disease (CD) is an uncommon hyperplastic disorder of lymphoid tissue, clinically categorized into unicentric CD (UCD) and multicentric CD (MCD), contingent upon the number of lymph node engagement. Unicentric Castleman disease (UCD) manifests as a solitary lump, predominantly found in the mediastinum, and infrequently in the head and neck region. Unlike multicentric Castleman disease (MCD), which displays peripheral lymph node enlargement and a multitude of systemic indications, UCD usually lacks generalized symptoms. Methods: We present a case of UCD presenting as a neck mass, in a 55-year-old woman as well as a detailed literature review for Castleman’s disease. The patient had undergone surgical excision of the mass. Results: The diagnostic findings following Immunohistochemistry were consistent with Hyaline vascular type Castleman’s disease. The patient remains stable with no signs or symptoms of recurrent disease after the mass removal. Conclusion: It is crucial to include Castleman’s’ disease in the differential diagnoses of lymphadenopathies due to the fact that it can be clinically mistaken for other common causes of lymphadenopathy such as lymphoma, tuberculosis, and nodal metastases.
KEY WORDS: Castleman’s disease; Case report, Unicentric Castleman, Surgical Treatment, Diagnosis
REFERENCES
1. van Rhee F, Oksenhendler E, Srkalovic G, et al. International evidence-based consensus diagnostic and treatment guidelines for unicentric Castleman disease. Blood Advances. 2020;(23):6039-6050. doi:10.1182/bloodadvances.2020003334
2. Zhang L, Dong Y, Peng H, et al. A national, multicenter, retrospective study of Castleman disease in China implementing CDCN criteria. The Lancet Regional Health - Western Pacific. Published online May 2023:100720. doi:10.1016/j.lanwpc.2023.100720
3. Paparella MT, Popolizio T, Grasso D, Serricchio EA, Guglielmi G. Castleman disease: a rare case in a young woman. Acta Biomedica Atenei Parmensis. 2023;(S1):e2023058. doi:10.23750/abm.v94iS1.13795
4. Simpson D. Epidemiology of Castleman Disease. Hematology/Oncology Clinics of North America. 2018;(1):1-10. doi:10.1016/j.hoc.2017.09.001
5. Dispenzieri A, Fajgenbaum DC. Overview of Castleman disease. Blood. 2020;(16):1353-1364. doi:10.1182/blood.2019000931
6. Mitsos S, Stamatopoulos A, Patrini D, George R, Lawrence D, Panagiotopoulos N. The Role of Surgical Resection in Unicentric Castleman’s Disease: A Systematic Review. Advances in Respiratory Medicine. 2018;(1):36-43. doi:10.5603/arm.2018.0008
7. Saadallah MAH. Castleman’s disease: A rare case report and review of literature. International Journal of Surgery Case Reports. Published online June 2022:107282. doi:10.1016/j.ijscr.2022.107282
8. Mremi A, Ndale E, Stephen L, Mkwizu E, Kilonzo K. Castleman’s disease: A report of two cases at a tertiary hospital in Northern Tanzania. SAGE Open Medical Case Reports. Published online January 2023:2050313X2311757. doi:10.1177/2050313x231175720
9. Lomas OC, Streetly M, Pratt G, et al. The management of Castleman disease. British Journal of Haematology. 2021;(3):328-337. doi:10.1111/bjh.17688
10. Oksenhendler E, Boutboul D, Fajgenbaum D, et al. The full spectrum of Castleman disease: 273 patients studied over 20 years. British Journal of Haematology. 2017;(2):206-216. doi:10.1111/bjh.15019
11. Maqbool S, Javed A, Idrees T, Anwar S. Unicentric Castleman Disease: A Rare Diagnosis of Radiological and Histological Correlation. Indian Journal of Otolaryngology and Head & Neck Surgery. 2022;(2):975-978. doi:10.1007/s12070-022-03253-4
12. Marcu RD, Spinu AD, Socea B, et al. Castleman’s Disease - Clinical, Histological and Therapeutic Features. Revista de Chimie. 2018;(4):823-830. doi:10.37358/rc.18.4.6208
13. Wang T, Chen X, Chen W, Shi L, Liu J. A retrospective study of 44 patients with head and neck Castleman’s disease. European Archives of Oto-Rhino-Laryngology. 2021;(5):2625-2630. doi:10.1007/s00405-021-07065-0
14. Gündüz E, Özdemir N, Bakanay ŞM, Karakuş S. A Rare Lymphoproliferative Disease: Castleman Disease. Turkish Journal of Hematology. Published online October 28, 2021. doi:10.4274/tjh.galenos.2021.2021.0440
15. Sun X, Du Y, Zhang Y, Wang R, Hou D. Unicentric Castleman disease: multidetector computed tomography classification with surgical and pathologic correlation. Quantitative Imaging in Medicine and Surgery. 2021;(8):3562-3568. doi:10.21037/qims-20-1033
16. Molacek J, Treska V, Skalicky T, et al. Unicentric form of Castleman´s disease, pitfalls of diagnosis and surgical treatment. Frontiers in Oncology. Published online January 30, 2023. doi:10.3389/fonc.2023.1057683
17. Qian S, Ding M, Hou H, et al. Clinical and Molecular Characteristics of 60 Patients With Human Immunodeficiency Virus-Negative Castleman Disease. Frontiers in Immunology.Published online May 17, 2022. doi:10.3389/fimmu.2022.899073
18. Garcia S, Fan J. Castleman Disease Presenting as a Mediastinal Mass. Cureus. Published online September 30, 2021. doi:10.7759/cureus.18409
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