Carcinoma ex Pleomorphic Adenoma in Nasal Cavity: An Unusual Diagnosis

Pérez-Carbonell, Tomàs; Taleb-Ferroukhi, Chakib; Calvo-Gómez, Jordi; Marco-Algarra, Jaime
doi:10.1016/j.otoeng.2021.07.001
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Clinical Case Study</span><p id="par0005" class="elsevierStylePara elsevierViewall">We present the case of a 42-year-old male patient&#44; with no history of interest&#44; who consulted due to bilateral nasal ventilation insufficiency of some years onset and occasional epistaxis of several months onset&#46; Rhinoscopic and endoscopic examination revealed major obstructive septal deviation to the left nasal cavity and a marked&#44; expansive mass which occupied the entire right nasal cavity&#46; No anatomical structures were located which were able to indicate precedence or origin&#46; A biopsy was performed which initially reported benign fibromixoma&#46; Imaging tests revelaed a solid mass centred in the right nasal cavity with extension to choanae and nasopharynx and with a maximum diameter of 6&#46;5&#160;&#215;&#160;5&#160;&#215;&#160;4&#46;5&#160;cm&#44; well-defined edges and signs of bone remodelling with marked septal deviation&#44; together with distortion of the inferomedial wall of the right orbit and remodelling of the hard palate roof &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Complete endoscopic excision was performed of the sinonasal tumour where a possible origin in the postero-superior third of the nasal septum was indicated&#44; in the joint between the perpendicular plate of the ethmoid sinuses and the vomer&#44; and tumour resection was thus completed with a septectomy of the posterior half of the septum&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Definitive histopathological diagnosis was non-invasive carcinoma ex pleomorphic adenoma &#40;CXPA&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">As a consequence of the bone remodelling&#44; due to expansive growth of the tumour&#44; the patient presented with a destructured anatomy with hypoplasic maxillary sinuses and turbinates and complete disappearance of ethmoid sinuses&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Due to the fragility of the surgical margins the patient presented with the postoperative complication of a oronasal fistula in the middle of the hard palate&#44; which was closed during a second-stage procedure with a palatal mucosal sliding flap&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">At present&#44; after two years of close follow-up&#44; the patient has no signs of recurrence or disease persistence and adheres to strict periodical endoscopic controls&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">CXPA is an extremely rare&#44; malign&#44; epithelial neoplasm which evolves from a pre-existing pleomorphic adenoma &#40;PA&#41;&#46; Only 10&#37; of pleomorphic adenomas are located in minor salivary glands of the upper aerodigestive tract&#46; CXPA accounts for just 3&#46;6&#37; of salivary gland tumours&#44; corresponding to 12&#37; of malign gland tumours&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">CXPA pathogenesis continues under debates&#46; There are basically two hypotheses&#44; the first being that the tumour from onset presents with components of carcinoma and the second more accepted hypothesis is that the adenoma over time and with progressive growth presents with a carcinomatous transformation&#44; and the incidence of this transformation increases by 1&#46;5&#37; at 5 years and by 10&#37; at 15 years&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Histopathologically&#44; pleomorphic adenomas are tumours which present with a mesenchymal and epithelial component&#46; Depending on the degree of invasion of the CXPA they are divided into non-invasive&#44; minimally invasive and invasive&#46; This division will affect the therapeutic approach and follow-up&#44; since it has been described that invasive CXPA has a 30&#37; survival rate of approximately 5 years&#44; whilst the non-invasive or minimally invasive forms present with a more benign disease course&#44; with no recurrences or spread of the disease&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> For CXPA diagnosis&#44; the finding of its must coexist with histological PA evidence&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Sinonasal location of PAs is highly infrequent&#44; and their becoming malignant to CXAP all the more so&#46; Until the year 2018 only 8 cases of CXAP had been reported in paranasal cavities and sinuses&#44;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4&#8211;8</span></a> and described in the literature&#44; but in 2019 Li et al&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> described 7 cases of CXAP in a series of 17 patients with PA in paranasal cavities and sinuses&#46; Their most common location inside the nose and the nasal cavities is the nasal septum&#44; like in our case&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a> Most patients&#44; like in our case&#44; refer to nasal impairment of years onset and congestion&#44; but they may with less frequency refer to oedema&#44; epistaxis&#44; purulent rhinorrhoea&#44; earache&#44; otitis media or hearing loss&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Imaging tests report bone changes depending on the speed and rhythm of tumour growth&#44; with changes being expansive or destructive&#46; In our case&#44; there was a remodelling of the bone margins of the nasal sinuses and cavities&#44; which would indicate a slow and progressive growth with no osteolysis&#44; which is an indirect sign of the degree of malignancy&#46; In the NMR the intensity of the signal of the weighted images in T1 is low to moderate whilst in T2 it is high&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">All the authors agree that surgical treatment is the treatment of choice for this type of tumour&#44; either endoscopic or open surgery depending on the extension and involvement of adjacent structures&#46; However&#44; there is some controversy regarding adjuvant treatment with radiotherapy&#46; Several authors ascertain that this does not improve the overall survival of these patients&#44;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> whilst others state that it leads to significant improvement in local control rates at 5 years and an improvement in survival&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> In our case the surgical specimen was analysed and we observed how the focal points of the carcinoma were at a distance from the peripherical margin and due to this&#44; together with the histological non-invasive subtype and the absence of radiologic findings suggestive of aggression&#44; we decided to only perform surgical resection with periodical endoscopic controls&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conclusion</span><p id="par0065" class="elsevierStylePara elsevierViewall">The histological study of the lesions in paranasal cavities and sinuses which appear to be benign and of slow growth is key to determining possible&#44; but infrequent development of malignant lesions such as CXAP&#44; since it will determine the prognosis and therapeutic approach according to the degree of histological aggression which these lesions present with&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conflict of Interests</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors have no conflict of interests to declare&#46;</p></span></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; P&#233;rez-Carbonell T&#44; Taleb-Ferroukhi C&#44; Calvo-G&#243;mez J&#44; Marco-Algarra J&#46; Carcinoma ex adenoma pleomorfo en fosas nasales&#58; un diagn&#243;stico inusual&#46; Acta Otorrinolaringol Esp&#46; 2021&#59;72&#58;265&#8211;267&#46;</p>"
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