Unusual Case of Inflammatory Myofibroblastic Tumor in Maxilla

Abstract

Inflammatory myofibroblastic tumor (IMT) is a rare lesion found mostly in children and young adults and originates from the lung, abdominopelvic region, and retroperitoneum. Clinical manifestations of IMT or imaging are nonspecific and diagnosis is based on histopathological and immunohistochemical findings. Minority of all IMTs will metastasize. IMT in the oral cavity is an extreme rarity and this is a first case report of IMT in maxilla causing delayed tooth eruption and multiple cervical root resorption with an 11-year-old child. The IMT reported here was positive for smooth muscle actin, vimentin, and anaplastic lymphoma kinase (ALK1) with immunohistochemistry. Only three IMTs of the jaws have been reported so far and none of them had delayed root eruption and tooth resorption. This unusual case of IMT in a child was also ALK1- positive supporting neoplastic origin of her tumor. The case presented here underscores the importance of histopathological examination of the tissue found in any root resorption especially in the case of multiple resorptions.

Inflammatory myofibroblastic tumor in the retromolar region of an 8-year-old child

“…The evidence of oral IMT in children was reported by Liston et al [ 5 ] where they described three cases 4 years, 2 years and 6 years, involving the buccal mucosa. Rautava et al [ 6 ] reported the case of an 11-year-old child with IMT involving the gingiva (maxilla buccal mucosa of 23 and 24) and Satomi et al [ 7 ] reported the case of a 14-year-old child with IMT involving the gingiva. To our knowledge, this is the first case report in the child where IMT was observed on the retromolar area.…”

Inflammatory myofibroblastic tumor of the upper alveolus: A rare entity presenting as a jaw swelling

“…IMT of the upper jaw has been reported in an 11-year-old female by Rautava et al . [ 10 ] IMTs of upper aero-digestive tract are common in adults (median age of 59) and more often seen in the males. [ 11 ] In the present case the patient is a female of 36 years old.…”

Recurrent tumor growth in the right maxilla - a rare case of an inflammatory myofibroblastic tumor

“…Aðeins 24 tilfellum af vöðvabandvefsfrumuaexlum með bólgufrumuíferð í munnholi hefur verið lýst og einungis þremur í kinnkjálka. 2,3 Flest birt tilfelli hafa verið í lungum, kviðar-eða grindarholi og aftanskinnubili. Slík aexli utan lungna virðast sýna ágengari ífarandi vöxt í aðlaega vefi þó svo að myndun meinvarpa sé sjaldgaef í öllum tilfellum.…”

“…Við skoðun var sjúklingur aumur við þreifingu yfir haegri kinn, í haegri nös, sem er þrengd af nefskiptaskekkju og að því er virtist miðlaegt hliðraðri nefskel, mátti sjá þrútnar slímhúðir án graftrartauma, en engin einkenni voru frá vinstri andlitshelmingi. Þegar haegri kinnhola var skoluð versnaði verkur til muna, án þess 1 Líffaerafraeði laeknadeildar Háskóla Íslands, 2 rannsóknarstofu í stofnfrumufraeðum við Lífvísindasetur Háskóla Íslands, 3 blóðmeinafraeðideild Landspítala, 4 lyfja-og eiturefnafraeðideild Háskóla Íslands, 5 meinafraeðideild Landspítala, 6 heilbrigðisog taugaverkfraeðisetur Háskólans í Reykjavík, 7 háls-, nef-og eyrnadeild Landspítala.…”

“…Hannes Halldórsson [1][2][3] , Ari jón Arason [1][2][3] , Margrét Sigurðardóttir 5 , Paolo Gargiulo 6 , Magnús Karl Magnússon [2][3][4] , Þórarinn Guðjónsson [1][2][3] , Hannes Petersen 1,7…”