A Case Report

Cai, Yefeng; Wang, Qingxuan; Ni, Chunjue; Dong, Siyang; Lin, Lan; Li, Quan; Chen, Endong; Zhang, Xiaohua

doi:10.1097/md.0000000000001891

Cited by 7 publications

(5 citation statements)

References 12 publications

(11 reference statements)

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“…There is no evidence to prove that adjuvant chemotherapy or radiotherapy after surgical resection improve the outcomes of primary thyroid LCH (2,5,7). Once thyroid LCH is diagnosed, it is necessary to perform systemic inspections, such as lung computed tomography, bone scintigraphy, liver ultrasonography, or positron emission tomography-computed tomography to definitively diagnose multi-system LCH, which requires systemic therapy (17,22). Chemotherapy regimens, such as vinblastine, etoposide, prednisone, and cytarabine have been used to treat aggressive disease.…”

Section: Discussionmentioning

confidence: 99%

Spontaneous Thyroid Hemorrhage Caused by Langerhans Cell Histiocytosis: A Case Report and Literature Review

et al. 2021

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PurposeLangerhans cell histiocytosis (LCH) is a rare clonal disorder of Langerhans antigen-presenting cells. However, thyroid LCH involvement is relatively rare. We present the first case of spontaneous thyroid hemorrhage due to LCH progression and discuss the clinical features, diagnosis, and treatments of thyroid LCH in a literature review.MethodsClinical data were collected. Previously published articles on thyroid LCH involvement were reviewed to assess the clinical features, diagnosis, and treatments for thyroid LCH.ResultsA 54-year-old female presented with a multi-system LCH, affecting the uterus, liver, pituitary gland, and thyroid gland. Clinical stability was achieved after systemic chemotherapy. After 7 years of regular follow up, the patient complained of a sudden painful neck swelling and progressive dyspnea. Computed Tomography revealed bilateral goiter with hematoma, and the patient was diagnosed with spontaneous thyroid bleeding based on her clinical symptoms and radiological findings. The patient was incubated to relieve airway compromise and partial thyroidectomy was performed for definitive treatment. Pathological evaluation further confirmed the diagnosis of thyroid LCH. The patient recovered well after surgery.ConclusionSpontaneous thyroid bleeding due to thyroid LCH progression is extremely rare. Treatments for LCH vary depending on the severity of the disease. We suggest that, for patients with multi-system LCH with thyroid lesion, long-term active surveillance of thyroid hormone concentrations, and thyroid gland volume is required. Physicians should be alert of the potentially life-threatening spontaneous thyroid hemorrhage when aggravated diffuse goiter and hypothyroidism appear. Further investigation is required to establish the guidelines for thyroid LCH treatment.

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Section: Discussionmentioning

confidence: 99%

Spontaneous Thyroid Hemorrhage Caused by Langerhans Cell Histiocytosis: A Case Report and Literature Review

et al. 2021

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“…Therefore, whether the operation should be performed and what is the ideal scope of operation become controversial. [ 1 , 17 ] If LCH is isolated in the thyroid, surgery should be the gold standard treatment whether or not PTC exists. If LCH involves several organs or systems, just as it usually does, cervical compressive symptoms and the circumstances of PTC should be assessed carefully.…”

Section: Discussionmentioning

confidence: 99%

“…It could involve several organs and systems such as the skin, lung, bone, liver, lymph nodes, hypothalamus, and central nervous system. [ 1 – 3 ] The exact etiology is still under investigation. [ 4 , 5 ] Differentiated thyroid cancer, especially papillary thyroid carcinoma (PTC), is the most common subtype of thyroid cancer, having an increasing incidence worldwide, possibly due to the increasing use of diagnostic imaging.…”

Section: Introductionmentioning

confidence: 99%

Langerhans cell histiocytosis of the thyroid complicated by papillary thyroid carcinoma

Chen²,

Zhang³

et al. 2017

Medicine

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Rationale:Langerhans cell histiocytosis (LCH) involves mainly the skin and bone and rarely the thyroid. Meanwhile, papillary thyroid carcinoma (PTC) is the most common subtype of thyroid cancer. Both LCH and PTC could make the thyroid enlarged and hypermetabolic. The coincidence of these 2 events in a patient is rare, and this paper aimed to report such case.Patient concerns:A 40-year-old man presented with polyuria and polydipsia for 5 years. The symptoms had been relieved well by drug therapy for >4 years, until the drugs could not control the symptoms anymore and an extensively enlarged thyroid gland was noticed.Diagnoses:Thyroid ultrasound showed a nodule with microcalcification in the upper right lobe, positron emission tomography/computer tomography scan demonstrated thyroid hypermetabolism, and fine needle aspiration (FNA) revealed PTC. Right lobectomy of the thyroid and cervical lymph node biopsy verified the diagnosis “LCH of the thyroid complicated by PTC.”Interventions:The ultrasound-guided FNA biopsy was performed prior to right lobectomy of the thyroid and cervical lymph node biopsy. Postoperative histopathological examination confirmed the diagnosis, after which the patient received adjuvant chemotherapy.Outcomes:After 5 cycles of adjuvant chemotherapy, the patient had been followed up for 2 years. LCH was controlled satisfactorily and there was no significant sign of recurrence or metastasis of PTC.Lessons:LCH of the thyroid complicated by PTC is rare. Thyroid involvement should always be considered in the differential diagnosis of LCH patients. Surgery for PTC followed by chemotherapy for LCH may be the suitable treatment.

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“…Given the likelihood of systemic involvement among LCH cases, close follow-up with periodic systemic PET/CT scans after the initial diagnosis and treatment of perianal LCH in our patient seems notable. PET/CT scan assessment enabled the incidental detection of thyroid gland involvement in our patient only after 9 months of single-system disease and long before the appearance of the clinically overt symptoms such as enlargement of neck mass or a goiter ( 9 ). Hence, our findings support the statement that most patients with LCH were PET positive at the time of initial diagnosis, while also emphasize the inclusion of this imaging modality as a part of the diagnostic workflow as well as in the setting of treatment response evaluation among adult LCH patients ( 10 ).…”

Section: Discussionmentioning

confidence: 87%

“…Our findings support that, albeit inconclusive, thyroid FNAB is instrumental in initiating diagnostic work up in LCH, while histology remains the most sensitive diagnostic modality ( 6 ). Nonetheless, given the likelihood of failure to get ideal biopsy specimens in thyroid nodules for the identification of LCH, it should also be noted that PET/CT is considered a competent diagnostic method for thyroid nodules with inconclusive cytologic results such as LCH involving thyroid ( 9 ).…”

Section: Discussionmentioning

confidence: 99%

An unusual case of adult-onset multi-systemic Langerhans cell histiocytosis with perianal and incident thyroid involvement

Gül¹,

Sisman²,

Cander³

et al. 2017

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryLangerhans cell histiocytosis (LCH) is a rare sporadic disease characterized by histiocytic neoplastic infiltration of various organ systems and a wide spectrum of clinical manifestations, ranging from benign and self-limiting to lethal. Herein, we report a rare case of adult-onset multi-systemic LCH in a 36-year-old male patient with an initial perianal presentation and incidental finding of subsequent thyroid gland involvement in the follow-up period. The patient with a history of perianal LCH treated with surgical excision and local radiotherapy was referred to our Endocrinology Department upon detection of hypermetabolic nodular lesions in the left lateral lobe of thyroid gland on positron emission tomography–computed tomography (PET/CT) scan in the nineth month of follow-up. Current evaluation revealed euthyroid status, a hypoechoic solid lesion of 13 × 9 mm in size with irregular borders in the left thyroid lobe on thyroid USG and cytologic assessment of thyroid nodule. The patient was diagnosed with suspected, oncocytic lesion, Hashimoto thyroiditis or LCH. The patient underwent total thyroidectomy and pathological assessment confirmed the diagnosis of Langerhans cell histiocytosis. Assessments in the sixth month of postoperative follow-up revealed euthyroid status with no thyroid tissue remnants or pathological lymph node on thyroid USG. In view of the multifocal lesions indicating multi-system disease, a systemic chemotherapy protocol with combination of prednisone (PRED) and vinblastine (VBL) has been planned by the hematology department.Learning points:Langerhans cell histiocytosis (LCH) shows a wide clinical spectrum and prognosis that ranges from benign and self-limiting single-system disease (with single or multifocal lesions) to a potentially lethal multi-system disease with severe organ dysfunction and death in some cases.It has been stated that the diagnosis is often delayed in perianal LCH unless LCH is specifically considered in the etiology, despite the fact that mucosal involvement may precede systemic involvement.Our findings support the statement that most of patients with LCH were PET positive at the time of initial diagnosis, while also emphasize the inclusion of this imaging modality as a part of the diagnostic workflow as well as in the setting of treatment response evaluation among adult LCH patients.

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A Case Report

Cited by 7 publications

References 12 publications

Spontaneous Thyroid Hemorrhage Caused by Langerhans Cell Histiocytosis: A Case Report and Literature Review

Spontaneous Thyroid Hemorrhage Caused by Langerhans Cell Histiocytosis: A Case Report and Literature Review

Langerhans cell histiocytosis of the thyroid complicated by papillary thyroid carcinoma

An unusual case of adult-onset multi-systemic Langerhans cell histiocytosis with perianal and incident thyroid involvement

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