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成年乳头状甲状腺癌患者伴发颈部淋巴结朗格汉斯细胞组织细胞增多症:2例报告并文献复习

Concomitant Langerhans cell histiocytosis of cervical lymph nodes in adult patients with papillary thyroid carcinoma: A report of two cases and review of the literature.

作者信息

Maraqa Bayan, Al-Ashhab Maxim, Kamal Nazmi, El Khaldi Mousa, Sughayer Maher

机构信息

King Hussein Cancer Center, Department of Pathology and Laboratory Medicine, Amman, Jordan.

King Hussein Cancer Center, Department of Radiology, Amman, Jordan.

出版信息

Autops Case Rep. 2021 Mar 12;11:e2021253. doi: 10.4322/acr.2021.253. eCollection 2021.

Abstract

OBJECTIVE

: Langerhans cell histiocytosis (LCH) is an uncommon entity of unknown etiology. It contains a wide range of clinical presentations. The discovery of oncogenic mutation in LCH has provided additional evidence that LCH is a neoplasm. Papillary thyroid carcinoma is the most common cancer of the thyroid characterized by a high incidence of mutations. LCH with concomitant PTC is rare, with few cases reported in the literature.

CASES SUMMARY

We identified two cases of LCH with concomitant papillary thyroid carcinoma in adult patients. The first was a 49-year-old female with a thyroid nodule diagnosed with papillary thyroid carcinoma. Later, the patient had a left neck mass; Ultrasound-guided lymph node FNA was diagnosed with Langerhans histiocytosis. Subsequently, a chest CT scan revealed signs of Langerhans cell histiocytosis in the lung. The second case refers to a 69-year-old male who presented with a left thyroid nodule diagnosed on FNA cytology as papillary thyroid carcinoma. The patient was found to have multiple bone lytic lesions. Biopsies revealed Langerhans cell histiocytosis. Later, the patient experienced LCH involvement of the bone marrow with associated secondary myelofibrosis.

CONCLUSIONS

LCH is rare in adults; the association with papillary thyroid carcinoma is reported and should be considered in the presence of Langerhans cell groups along with PTC, whether in the thyroid gland or cervical lymph nodes. Once LCH has been diagnosed, pulmonary involvement should also be investigated. This will direct treatment plans for patients with pulmonary or systemic disease involvement.

摘要

目的

朗格汉斯细胞组织细胞增多症(LCH)是一种病因不明的罕见疾病。它有广泛的临床表现。LCH中致癌突变的发现为LCH是一种肿瘤提供了更多证据。甲状腺乳头状癌是最常见的甲状腺癌,其特征是突变发生率高。LCH合并PTC很罕见,文献中报道的病例很少。

病例总结

我们确定了两例成年患者LCH合并甲状腺乳头状癌的病例。第一例是一名49岁女性,有一个甲状腺结节,诊断为甲状腺乳头状癌。后来,该患者左侧颈部出现肿块;超声引导下淋巴结细针穿刺活检诊断为朗格汉斯组织细胞增多症。随后,胸部CT扫描显示肺部有朗格汉斯细胞组织细胞增多症的迹象。第二例是一名69岁男性,出现左侧甲状腺结节,细针穿刺细胞学检查诊断为甲状腺乳头状癌。该患者被发现有多处骨质溶解病变。活检显示为朗格汉斯细胞组织细胞增多症。后来,该患者出现骨髓LCH累及并伴有继发性骨髓纤维化。

结论

LCH在成人中罕见;报道了其与甲状腺乳头状癌的关联,在存在朗格汉斯细胞群以及PTC时,无论在甲状腺或颈部淋巴结中,都应予以考虑。一旦诊断为LCH,还应调查肺部受累情况。这将指导肺部或全身疾病受累患者的治疗方案。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5d78/8214889/a22b84a7985f/autopsy-11-e2021253-gf01.jpg

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