Spindle Epithelial Tumor with Thymus‐Like Differentiation: A Case Report and Review of the Literature

Bradford, Carol R.; Devaney, Kenneth O.; Lee, Janet I.

doi:10.1053/hn.1999.v120.a85593

Cited by 16 publications

(17 citation statements)

References 8 publications

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“…SETTLE occurs in young patients, with indolent growth and a tendency to develop delayed blood-borne metastases. Since the report of Chan and Rosai (1), a number of case reports have appeared in the literature, but all of them are flawed by short follow-up periods and incomplete compilation of the previously reported cases (7)(8)(9)(10)(11)(12). Consequently, the malignant potential of this tumor may not be fully evident from these data.…”

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confidence: 99%

Spindle Epithelial Tumor with Thymus-Like Differentiation (SETTLE): A Distinctive Malignant Thyroid Neoplasm with Significant Metastatic Potential

2000

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Spindle epithelial tumor with thymus-like differentiation (SETTLE) is a very rare tumor of the thyroid believed to be derived from branchial pouch or thymic remnants and showing primitive thymic differentiation. Although this tumor is prone to develop delayed blood-borne metastases, the metastatic risk is unclear because the case reports in the recent literature had very short follow-up periods. We report one case, the oldest patient reported so far, who had been followed up until death. The 59-year-old man had an enlarged thyroid for all of his adult life, and presented with recent rapid enlargement of the thyroid. Neck exploration revealed a hard tumor in the right lobe, with adhesion to sternothyroid muscle. Histologic examination showed an invasive biphasic neoplasm traversed by sclerotic septa. Tight to loose fascicles of blandlooking spindly cells were intimately intermingled with tubulopapillary structures, diagnostic of the SETTLE. This patient developed left pulmonary metastases at 2 years and subsequently developed bilateral pulmonary and widespread metastases. He died 8 years after initial presentation. This case illustrates the protracted clinical course of the tumor, and survival for many years despite the occurrence of metastases. Review of the literature shows that SETTLE occurs predominantly in young patients with a median age of 15 years and male predominance. There is a significant metastatic rate of 71% for patients with more than 5 years of follow-up in spite of the otherwise indolent nature of the tumor.

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confidence: 99%

Spindle Epithelial Tumor with Thymus-Like Differentiation (SETTLE): A Distinctive Malignant Thyroid Neoplasm with Significant Metastatic Potential

2000

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“…In addition, there is usually strong and diffuse immunostaining for CD99 and BCL-2 protein. The ultrastructural lack of tonofilaments, as well as the specific translocation t(x; 18) (p11.2; q11.2), involving the gene SS18 (SYT or SSXT) in chromosome 18, and the genes SSX1, SSX2 and SSX4 in chromosome X, support this distinguishing (2,3,16,18,23).…”

Section: Immunohistochemistrymentioning

confidence: 83%

“…SETTLE is a distinct low-grade neoplasm, believed to be derived from branchial pouch or thymic remnants, with only 1 case report showing association with epithelium-lined cysts of possible branchial pouch derivation (2). It belongs to a group of cervical lesions that includes ectopic cervical thymoma, ectopic hamartomatous thymoma and carcinoma with thymus-like element (CASTLE) (3). This tumor is composed predominantly of spindle and epithelioid cells with glandular or ductular structures lined by a mucinous or respiratory epithelium (4).…”

Section: Introductionmentioning

confidence: 99%

Thyroid spindle epithelial tumor with thymus-like differentiation (SETTLE): case report and review

Filho

Bordallo

Pessoa

et al. 2010

Arq Bras Endocrinol Metab

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SUMMARYSpindle epithelial tumor with thymus-like element (SETTLE) is a rare malignant neoplasm of the thyroid, occurring predominantly in children, adolescents, and young adults. SETTLE usually presents itself as a thyroid mass, without metastases at diagnosis. It is believed to derive from branchial pouch or thymic remnant tissue showing primitive thymic differentiation. This article reports the clinical, cytological, histological and immunohistochemical features of a SETTLE in a 3-year-old girl. Microscopic exam revealed a nodular, highly cellular neoplasm displayed in the classic biphasic pattern, with mixture of prominent spindle cell component and a minor glandular component lined by mucinous or respiratory-type epithelium. The immunohistochemical study showed strong and diffuse positivity for pan-CK, vimentin and smooth muscle actin. The present case is the first SETTLE case reported in Brazil. To date, the patient described remains without evidence of recurrence or metastasis 5 years after surgery. Arq Bras Endocrinol Metab. 2010;54(7):657-62 SUMÁRIO O tumor epitelial de células fusiformes com elemento thymus-like é uma rara neoplasia maligna da tireoide, ocorrendo predominantemente em crianças, adolescentes e adultos jovens. Habitualmente, esse tumor se apresenta como massa tireoideana, sem metástases ao diagnóstico. Acredita-se derivar de arco branquial ou tecido remanescente tímico, apresentando diferenciação tímica primitiva. Este artigo descreve os aspectos clínicos, citológicos, histológi-cos e imuno-histoquímicos de um caso de SETTLE diagnosticado em uma menina de 3 anos de idade. O aspecto microscópico encontrado no tumor foi de uma lesão nodular, hipercelular, disposta em aspecto bifásico clássico, com componente de células fusiformes, e de tecido glandular acompanhado por epitélio mucinoso e respiratório. O estudo imuno-histoquímico foi positivo para pan-CK, vimentina e actina de músculo liso. Esse caso é o primeiro relato de SETTLE no Brasil. A paciente descrita permanece sem evidência de doença em atividade cinco anos após o tratamento cirúrgico. Arq Bras Endocrinol Metab. 2010;54(7):657-62

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“…Prema podacima američkog Nacionalnog centra za biotehnološka istraživanja, naziv SETTLE spomenut je samo 26 puta u engleskoj literaturi do jula 2010. godine. Naveden je 42 puta do kraja juna 2015. u obliku pojedinačnih izvještaja i nepotpunog zbira prethodno objavljenih slučajeva (7)(8)(9)(10)(11)(12). Donosimo slučaj bolesnika kod koga za razliku od svih dosad prikazanih slučajeva primarni tumor nikada nije bio pronađen.…”

Section: Uvodunclassified

“…U dosadašnjoj literaturi, svi slučajevi SETTLE tumora (1,3,4,5,6,(8)(9)(10)(11)(12)(13)(14) tretirani su hirurškom intervencijom na tumoru. Adjuvantna hemioterapija i radioterapija primijenjene su u nekim slučajevima iako uloga najboljeg protokola nije jasna.…”

Section: Diskusijaunclassified

Clinical, pathological and therapeutic course of disease in patient with initial diagnosis of metastatic settle tumor: A case report

Cicmil-Sarić¹,

Vučković²,

Basovic-Ristic³

et al. 2015

Medicinski casopis

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UVODEpitelijalni tumor vretenastih ćelija sa diferencijacijom sličnoj timusu (SETTLE) ekstremno je rijedak tumor štitaste žlijezde za koji se smatra da se razvija iz škržne brazde ili ostataka timusa i pokazuje primitivnu diferencijaciju grudne žlijezde. Formalno su ga okarakterisali Chan i Rosai 1991. godine (1). Pojedinačni izvještaji o ovome tumorskom entitetu prethodno su objavljivani pod različitim oznakama kao što su: tumor vretenastih ćelija štitaste žlijezde sa mukoznom cistom, maligni teratom, tireoidni timom dječije dobi, neuobičajeni tireoidni tumor kod djece (2-6). Promjena je slabo rastumačena a njen potencijal za kasno metastaziranje postaje opštepriznat: iako tumor ima sklonost da razvija kasne hematogene metastaze, rizik od metastaziranja je nejasan jer prikazi slučajeva u novijoj literaturi imaju veoma kratak period praćenja. Prema podacima američkog Nacionalnog centra za biotehnološka istraživanja, naziv SETTLE spomenut je samo 26 puta u engleskoj literaturi do jula 2010. godine. Naveden je 42 puta do kraja juna 2015. u obliku pojedinačnih izvještaja i nepotpunog zbira prethodno objavljenih slučajeva (7-12). Donosimo slučaj bolesnika kod koga za razliku od svih dosad prikazanih slučajeva primarni tumor nikada nije bio pronađen. Zbog toga pacijent nije operisan. Pacijenta smo pratili dok je postojala samo klinička manifestacija oboljenja i od početka liječenja metastatske bolesti do prve ustanovljene RECIST progresije. Smatramo da bi osnivanje međunarodnog tijela za rijetke maligne tumore bilo veoma značajno. PRIKAZ SLUČAJAPrikazujemo slučaj SETTLE tumora kod dvadesettrogodišnjeg muškarca koji je praćen 24 mjeseca: od trenutka postavljanja dijagnoze dok nije ustanovljena progresija bolesti prema RECIST kriterijumima. Ovo je prvi slučaj SETTLE tumora opisan u Crnoj Gori.Polovinom 2011. godine kod bolesnika koji je imao 19 godina na desnoj strani vrata pojavio se bezbolan limfni čvor. Bolesnik nije imao subjektivnih tegoba. Limfni čvor bi postajao vidljiv prilikom okretanja glave na desnu stranu. Javio se stomatologu koji je objasnio da je pojava limfnog čvora posljedica rasta umnjaka i da će iščeznuti nakon što umnjak bude nikao. Izvjesno vrijeme poslije toga limfni čvor nije bio vidljiv. Kada je promjena ponovo postala uočljiva, u martu 2013, pacijent je zatražio

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Spindle Epithelial Tumor with Thymus‐Like Differentiation: A Case Report and Review of the Literature

Cited by 16 publications

References 8 publications

Spindle Epithelial Tumor with Thymus-Like Differentiation (SETTLE): A Distinctive Malignant Thyroid Neoplasm with Significant Metastatic Potential

Spindle Epithelial Tumor with Thymus-Like Differentiation (SETTLE): A Distinctive Malignant Thyroid Neoplasm with Significant Metastatic Potential

Thyroid spindle epithelial tumor with thymus-like differentiation (SETTLE): case report and review

Clinical, pathological and therapeutic course of disease in patient with initial diagnosis of metastatic settle tumor: A case report

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