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Case Study
Inflammatory Myofibroblastic Tumour of the Tonsil: Case Report and Literature Review
Tumor miofibroblástico inflamatorio de amígdala: informe de un caso con revisión de la literatura
Peter Grube-Pagolaa,b,
Corresponding author
grubejr78@gmail.com

Corresponding author.
, Daniel Carrasco-Dazab, Georgia Alderete-Vázquezb, Juan Gutiérrez-Butandac
a Consultorio de Anatomía Patológica y Patología Pediátrica, Veracruz, Veracruz, Mexico
b Laboratorio de Anatomía Patológica, Instituto Nacional de Pediatría (INP), México City, Mexico
c Departamento de Otorrinolaringología, Instituto Nacional de Pediatría (INP), MéxicoCity, Mexico
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with a sudden onset of cervical pain and arthralgia&#46; She was treated with paracetamol&#44; which partially improved her symptoms&#46; Subsequently&#44; she suffered cough and vomiting of gastric contents with traces of blood&#44; as well as foreign body sensation in the throat which limited speech&#44; so she was referred to our hospital&#46; Physical examination revealed a good general condition and the presence of a tumour with irregular edges in the oropharyngeal area&#46; This tumour was dependent on the left tonsil and displaced the uvula&#44; causing pain when touched&#46; The contralateral tonsil showed no abnormalities and the rest of the physical examination was normal&#46; She underwent flexible nasofibrolaryngoscopy&#44; which revealed a grade I adenoid hyperplasia&#46; In addition&#44; we observed that the lesion was in contact with the posterior pharyngeal wall&#46; We performed bilateral tonsillectomy and the pathological findings were as follows&#58; cylindrical left amygdala&#44; yellow-grey in colour&#44; coated with fibrin&#44; measuring 5&#46;2<span class="elsevierStyleHsp" style=""></span>cm&#215;2&#46;4<span class="elsevierStyleHsp" style=""></span>cm&#215;1&#46;5<span class="elsevierStyleHsp" style=""></span>cm and with a firm and smooth consistency &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Histologically&#44; we observed granulation tissue on the surface&#44; with partial absence of epithelial lining&#46; Within the tumour we observed a disorderly proliferation of dense collagenous bands with stellar and tapered myofibroblasts&#44; as well as a polymorphic&#44; inflammatory infiltrate composed of lymphocytes&#44; plasma cells&#44; mast cells&#44; neutrophils and eosinophils &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A and B&#41;&#46; Immunohistochemical stains of the stromal cells were positive for vimentin&#44; CD34 and muscle-specific actin&#44; revealing myofibroblastic differentiation&#44; as well as for CD3&#44; CD20&#44; CD68 and CD21&#44; which confirmed the polyclonal nature of the inflammatory infiltrate &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C and D&#41;&#46; With this evidence&#44; we reported a diagnosis of inflammatory myofibroblastic tumour&#46; We also conducted an ALK1 test&#44; which was negative&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">IMT has been recently classified as a neoplastic process with myofibroblastic differentiation&#44; accompanied by dense and polymorphic inflammatory infiltrate&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The histological image of inflammatory myofibroblastic tumour is variable and because of this it has had several names throughout history&#44; such as inflammatory pseudotumor or plasma cell granuloma&#44; among others&#46; It mainly affects soft tissues and the lungs&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Its location in the head and neck region &#40;extraorbital&#41; is very rare&#46; There have been reports of cases located in the skull base&#44; parapharyngeal space&#44; paranasal sinuses&#44; pterygopalatine fossa&#44; oral cavity and parotid&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4&#44;8</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">At the moment&#44; treatment for IMT is controversial&#46; Throughout history&#44; several authors have proposed therapies based on steroids&#44; excisional biopsy with wide resection&#44; radiotherapy and even chemotherapy&#44; although these treatment models have been described in various locations&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The medical literature only contains 2 reported cases of IMT located in the palatine tonsil&#46; In 1984&#44; Weilbaecher published the case of a 63-year-old man&#44; who was asymptomatic despite presenting an enlarged&#44; right palatine tonsil&#46; This had a firm consistency&#44; measured 3<span class="elsevierStyleHsp" style=""></span>cm&#215;2&#46;5<span class="elsevierStyleHsp" style=""></span>cm&#215;2<span class="elsevierStyleHsp" style=""></span>cm and was reported as a plasma cell granuloma&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> One decade later&#44; in 1995&#44; Newman reported the case of a 62-year-old woman who presented odynophagia as a main symptom&#44; along with an enlarged&#44; left palatine tonsil&#46; This had a firm consistency&#44; an erythematous appearance and measured 3&#46;5<span class="elsevierStyleHsp" style=""></span>cm&#215;2&#46;5<span class="elsevierStyleHsp" style=""></span>cm&#215;2<span class="elsevierStyleHsp" style=""></span>cm&#46; It was reported as an inflammatory pseudotumor&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">In conclusion&#44; we present a case of primary IMT in the palatine tonsil&#44; which represents the third case described in the literature and the first in a paediatric patient&#46; Our patient presented enhanced symptoms compared with previous cases&#44; probably owing to the fact that this tumour was the largest one reported so far&#46; From a clinical standpoint&#44; it is important to be aware that IMT can affect the palatine tonsil&#44; so it should be included in the differential diagnosis of primary lesions which appear with unilateral tonsillar hypertrophy&#46; Unilateral tonsillectomy is controversial in such cases&#44; but we believe that it should be the treatment of choice&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conflict of Interests</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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        "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Inflammatory myofibroblastic tumour &#40;inflammatory pseudotumor&#41; is an idiopathic lesion&#44; rare in the head and neck&#44; of unknown aetiology&#46; It is primarily a soft tissue&#44; lung and orbital condition&#46; In the world literature&#44; only two cases with tonsillar disease have been found&#46;</p><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">We report a case of a 10-year-old girl admitted to our hospital with clinical complaints of pain in the neck region&#44; cough with vomiting&#44; dyspnoea and dyslexia&#46; Clinical examination revealed halitosis and a neoformation dependent on the left tonsil&#46; Bilateral tonsillectomy was performed&#46;</p>"
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        "resumen" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">El tumor miofibrobl&#225;stico inflamatorio &#40;seudotumor inflamatorio&#41; es una lesi&#243;n idiop&#225;tica&#44; poco frecuente en cabeza y cuello&#44; de etiolog&#237;a desconocida&#44; que se presenta m&#225;s com&#250;nmente en tejidos blandos&#44; pulm&#243;n y &#243;rbita&#46; En la literatura mundial solo se tiene conocimiento previo de 2 casos con afecci&#243;n amigdalar&#46;</p><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Informamos del caso de una ni&#241;a de 10 a&#241;os&#44; que ingres&#243; en nuestro hospital con cuadro cl&#237;nico de dolor en la regi&#243;n cervical&#44; tos con v&#243;mito&#44; disnea y dislexia&#44; la exploraci&#243;n f&#237;sica mostr&#243; halitosis y una neoformaci&#243;n dependiente de la am&#237;gdala izquierda&#44; se le realiz&#243; amigdalectom&#237;a bilateral&#46;</p>"
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es en pt

¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?

Você é um profissional de saúde habilitado a prescrever ou dispensar medicamentos