Langerhans Cell Histiocytosis of the Thyroid Leading to the Diagnosis of a Disseminated Form

Naçef, I. Ben; Mekni, Sabrine; Mhedhebi, C.; Riahi, I.; Rojbi, I.; Mchirgui, N.; Khiari, K.

doi:10.1155/2020/6284764

Cited by 3 publications

(4 citation statements)

References 11 publications

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“…A total of 49 relevant articles were published between 2010 and 2020, but 22 articles were excluded due to incomplete information about the clinical feature and treatment of thyroid LCH. Therefore, in 27 articles, 29 cases of thyroid LCH involvement were reviewed ( Table 1 ) ( 2 – 28 ). LCH involving the thyroid gland is more common in adults compared with children (25 adults vs. 4 children).…”

Section: Resultsmentioning

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: Resultsmentioning

confidence: 99%

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

et al. 2021

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“…Moreover, it should be noted that a thyroid surgical biopsy could also yield a wrong diagnosis. In several cases, the diagnosis of PTC confirmed through surgical biopsy was corrected to LCH due to the identification of disseminated disease (30,31). Overall, we recommend FNA for patients with suspected thyroid LCH.…”

Section: Thyroid Langerhans Cell Histiocytosis Together With Papillar...mentioning

confidence: 95%

Analysis of thyroid involvement in children and adult Langerhans cell histiocytosis: An underestimated endocrine manifestation

Chang²,

Chai³

et al. 2022

Front. Endocrinol.

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BackgroundLangerhans cell histiocytosis (LCH) is a rare disease caused by the clonal expansion of CD1a+/CD207+ LCH cells. The thyroid involvement in LCH has mostly been described in case reports.MethodsWe retrospectively evaluated the clinical characteristics, diagnosis, and treatment of 27 children and adult patients with thyroid LCH in our center between 2010 and 2021.ResultsThe incidence of thyroid LCH was 14.00% (7/50) in children and 10.10% (20/198) in adults, respectively. Among patients with thyroid involvement, 81.5% presented with diabetes insipidus (DI) as the first symptom, and 51.9% complained of neck swelling or mass. Children and adults with thyroid LCH had higher frequencies of the hypothalamic-pituitary axis (HPA) (children: 100% vs. 62.8%, P=0.05; adult: 95% vs. 42.1%, P<0.001), the lung (children: 85.7% vs. 25.6%, P=0.004; adult: 70% vs. 50.6%, P=0.099), and a lower frequency of bone (children: 14.3% vs. 55.8%, P=0.049; adult: 45% vs. 73.6%, P=0.008) involvement than patients without thyroid involvement. Patients with thyroid LCH had a higher frequency of primary hypothyroidism and a lower frequency of euthyroidism than patients without it. The two major types of ultrasound imaging were diffuse (55%) and nodular type (45%). The standardized uptake value of thyroid on 18-F-fluorodeoxyglucose positron emission tomography/computed tomography was 5.3–12.8. The diagnoses were confirmed using thyroid aspiration (54.5%) or surgery (45.5%). In addition, thyroid LCH combined with papillary thyroid carcinoma was not rare (2/27).ConclusionThyroid involvement in LCH is not rare. Furthermore, identifying thyroid involvement can facilitate the pathological diagnosis of LCH. Therefore, the possibility of thyroid LCH should be fully investigated in patients with DI, primary hypothyroidism, abnormal thyroid ultrasound results, and multi-system disease. In addition, thyroid aspiration can confirm suspected thyroid LCH. Finally, special attention should be paid to evaluating HPA and pulmonary involvement in thyroid LCH.

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“…En el caso actual, la PAAF se describe característicamente como un extendido hipercelular, con numerosas células gigantes multinucleadas. Es necesaria una alta sospecha para guiar al diagnóstico final; en algunos pacientes aun luego de una tiroidectomía y una biopsia formal, se confunden los diagnósticos con una tiroiditis subaguda y al presentarse otros síntomas de afectación extratiroidea (diabetes insípida, lesiones líticas oseas), se realiza el diagnostico restrospectivamente tras revisión de placas e inmunohistoquimica (9) . El paciente fue sometido a una tiroidectomía total confirmándose el diagnóstico de HCL con afectación de los ganglios peritiroideos, a través de la Anatomía Patológica y la Inmunohistoquímica en donde se observan las células características LCA+, S100+, CD1a+ CD68+/-.…”

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“…Los niños con afectación del hígado, el bazo o la médula ósea tienen un desenlace fatal, por lo tanto, se clasifican como pacientes con HCL de alto riesgo (6) . Se han descrito casos en que la presentación inicial fue en un solo órgano, como en el caso de la hipófisis o de la tiroides causando una diabetes insípida o un hipotiroidismo respectivamente y con el tiempo evolucionan a afectación sistémica (9) . En los adultos, la HCL pulmonar aislada (PLCH) es la manifestación más común y se considera un tipo específico de HCL por su origen policlonal (10) .…”

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Histiocitosis de células de langerhans. Un hallazgo poco común en la glándula tiroides. A propósito de un caso

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Ferreira

et al. 2021

Rev. cient. cienc. salud

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Langerhans Cell Histiocytosis of the Thyroid Leading to the Diagnosis of a Disseminated Form

Cited by 3 publications

References 11 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

Analysis of thyroid involvement in children and adult Langerhans cell histiocytosis: An underestimated endocrine manifestation

Histiocitosis de células de langerhans. Un hallazgo poco común en la glándula tiroides. A propósito de un caso

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