WANG Qian, SUN Quanya, HE Min, ZHANG Shuo, XIANG Boni, LI Qiufan, WANG Yong, ZHANG Xialing, DING Tianling, YE Hongying. Clinical Characteristics and Diagnostic Experience of Adult Thyroid Langerhans Cell Histiocytosis with Diabetes Insipidus[J]. Journal of Rare Diseases, 2023, 2(3): 346-352. DOI: 10.12376/j.issn.2097-0501.2023.03.005
Citation: WANG Qian, SUN Quanya, HE Min, ZHANG Shuo, XIANG Boni, LI Qiufan, WANG Yong, ZHANG Xialing, DING Tianling, YE Hongying. Clinical Characteristics and Diagnostic Experience of Adult Thyroid Langerhans Cell Histiocytosis with Diabetes Insipidus[J]. Journal of Rare Diseases, 2023, 2(3): 346-352. DOI: 10.12376/j.issn.2097-0501.2023.03.005
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Clinical Characteristics and Diagnostic Experience of Adult Thyroid Langerhans Cell Histiocytosis with Diabetes Insipidus

  • 1.

    Department of Endocrinology, Huashan Hospital Affiliated to Fudan University, Shanghai 200040, China

  • 2.

    Department of Ultrasound, Huashan Hospital Affiliated to Fudan University, Shanghai 200040, China

  • 3.

    Department of Pathology, Huashan Hospital Affiliated to Fudan University, Shanghai 200040, China

  • 4.

    Department of Hematology, Huashan Hospital Affiliated to Fudan University, Shanghai 200040, China

Funds: 

National Project in Promoting the Diagnosis and Treatment of Major Diseases by MDT 

More Information
  • Corresponding author:

    YE Hongying, E-mail: yehongying@huashan.org.cn

  • Received Date: May 07, 2023
  • Accepted Date: June 01, 2023
2097-0501/©2023 Editorial Office of Journal of Rare Diseases This is an open access article under the CC BY-NC-ND license (https://creativecommons.org/licenses/by-nc-nd/4.0/)
    Graphical Abstract
    • Abstract
  • Objective 

    To analyze the clinical characteristics of thyroid LCH to enhance understanding of the disease.

    Methods 

    We retrospectively studied the clinical data from six thyroid LCH patients who hospitalized in Huashan Hospital Affiliated to Fudan University from January 2015 to January 2022.We analyzed the ultrasound and 18F FDG-PET/CT imaging characteristics of thyroid LCH.

    Results 

    The six patients diagnosed (2 males and 4 females) were between 18 and 58 years old.All patients had diabetes insipidus.MRI revealed thickened pituitary stalk.Two cases had central hypothyroidism, while four cases euthyroidism.Three cases tested positive for thyroid antibodies.Ultrasound showed thyroid nodules of TI-RADS 3 in three cases, TI-RADS 4 in two cases, and 1 with nodular goiter.Ultrasound showed that all sic cases indicated low echogenicity, 5 of which clear boundaries, 4 of which uneven echo distribution, 5 of which irregular shape, and noen has calcification.18F FDG-PET/CT indicated high uptake nodules with SUVmax values all above 10.4 cases were diagnosed by surgical excision and the other 2 by coarse-needle aspiration biopsy.When diagnosed, two cases had liver and thymus involvement respectively.One case had lung and bone involvement respectively.After treatment, 4 cases showed that nodular goiter shrank, while the other two with liver involvement progressed fast and no assessment made.

    Conclusions 

    Thyroid LCH presented low echogenicity, clear boundaries, irregular shape, without calcification, and high uptake in 18F FDG-PET/CT.A definite diagnosis of pituitary stalk thicking accompanied by thyroid nodules, especially those with hypoechoic and irregular nodules, can be achieved by coarse-needle aspiration biopsy and langerin-specific pathological staining.

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    • References (17)
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