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Comprehensive analysis of 225 Castleman’s diseases in the oral maxillofacial and neck region: a rare disease revisited

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Abstract

Objectives

The aim of the present study was to comprehensively summarize the epidemiological, clinicopathological characteristics, treatments as well as prognosis of Castleman’s disease (CD) identified in the oral maxillofacial and neck region.

Materials and methods

Patients with CD in the oral maxillofacial and neck were retrieved from disease registry at our institution from Jan. 1990 to Dec. 2015. Systematic reviews from both English and Chinese literature were performed to collect the detailed information about the oral maxillofacial and neck CD. The epidemiological, clinicopathological data and treatment outcomes were further statistically analyzed.

Results

Four patients with the oral maxillofacial and neck CD were identified and histologically confirmed as hyaline-vascular type. They underwent surgical excision without recurrence during the follow-up. Systematic literature reviews identified 221 cases from 123 eligible articles which satisfied the inclusion criteria. In 225 patients, most patients were diagnosed as unicentric (207) or hyaline-vascular type (205) of CD and identified in the neck, and treated by surgical resection with good prognosis. In contrast, the minority of patients was multicentric or plasma-cell/mixed type and treated by chemotherapy with inferior outcomes. Kaplan-Meir analyses revealed that both clinical and pathological types were significantly associated with patients’ overall survival.

Conclusions

Although rare, most cases of the oral maxillofacial neck CD are found in adults and classified as unicentric and hyaline-vascular type of CD. Complete surgical excision is preferred with favorable prognosis for unicentric disease, whereas chemotherapy is usually exploited for multicentric disease with inferior outcomes.

Clinical relevance

These data provide comprehensive information about the epidemiology, clinicopathological features, treatments, and outcomes of the oral maxillofacial and neck CD.

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Funding

This work is financially supported, in whole or in part, by the National Natural Science Foundation of China (81572669), Natural Science Foundation of Jiangsu Province (BK20161564), a Project Funded by the Priority Academic Program Development of Jiangsu Higher Education Institutions (2014-37), China Postdoctoral Science Foundation (2014M560436), Jiangsu Creative Training Project for Graduates in Colleges (SJZZ_0119), and Jiangsu Creative Training Project for College Student (201510312053X).

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Authors and Affiliations

  1. Jiangsu Key Institute of Stomatology, Nanjing Medical University, 136, Hanzhong Road, Nanjing, 210029, People’s Republic of China

    Xiaotong He, Qiong Wang, Yanling Wang & Jie Cheng

  2. Department of Oral and Maxillofacial Surgery, Nanjing Medical University, Nanjing, 210029, People’s Republic of China

    Yaping Wu, Jiaan Hu, Dongmiao Wang & Jie Cheng

  3. Department of Oral Pathology, Affiliated Stomatological Hospital, Nanjing Medical University, Nanjing, 210029, People’s Republic of China

    Bin Qi & Wei Zhang

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  1. Xiaotong He
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  2. Qiong Wang
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  3. Yaping Wu
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  4. Jiaan Hu
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  5. Dongmiao Wang
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  6. Bin Qi
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  7. Wei Zhang
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  8. Yanling Wang
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  9. Jie Cheng
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Correspondence to Jie Cheng.

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The authors declare that they have no competing interests.

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All procedures performed in studies involving human participants were in accordance with the ethical standards of our institutional research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

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Informed consent was obtained from all individual participants included in the study.

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Dr. Xiaotong He and Dr. Qiong Wang contributed equally to this work as co-first authors.

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He, X., Wang, Q., Wu, Y. et al. Comprehensive analysis of 225 Castleman’s diseases in the oral maxillofacial and neck region: a rare disease revisited. Clin Oral Invest 22, 1285–1295 (2018). https://doi.org/10.1007/s00784-017-2232-x

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  • DOI: https://doi.org/10.1007/s00784-017-2232-x

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