Oral Lesion as Unusual First Manifestation of Multiple Myeloma: Case Reports and Review of the Literature
Identifieur interne : 002A78 ( Pmc/Corpus ); précédent : 002A77; suivant : 002A79Oral Lesion as Unusual First Manifestation of Multiple Myeloma: Case Reports and Review of the Literature
Auteurs : A. Romano ; M. S. Marescalco ; Chiara Liardo ; L. Villari ; C. Vetro ; C. Conticello ; F. Di Raimondo ; S. FerlitoSource :
- Case Reports in Hematology [ 2090-6560 ] ; 2014.
Abstract
Extramedullary plasmacytoma (EMP) and solitary bone plasmacytoma (SBP) represent a disease continuum through a multistage process of cell differentiation, survival, proliferation, and dissemination, strictly related to multiple myeloma (MM), the second most common hematological malignancy. Herein, we report two cases of recurrent oral plasmacytoma progressed to MM, in which the first clinical sign of a more widespread disease was limited to the mouth. Based on our experience, we recommend a strict workup for the differential diagnosis between EMP, SBP, and MM for patients with oral plasmacytoma, including radiological exam of the skeleton, magnetic resonance imaging (MRI) of the bone, and positive emission tomography (FDG-PET). MRI and possibly PET can all be used to more sensitively detect EM plasmacytoma sites.
Url:
DOI: 10.1155/2014/529452
PubMed: 25506443
PubMed Central: 4260434
Links to Exploration step
PMC:4260434Le document en format XML
<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Oral Lesion as Unusual First Manifestation of Multiple Myeloma: Case Reports and Review of the Literature</title><author><name sortKey="Romano, A" sort="Romano, A" uniqKey="Romano A" first="A." last="Romano">A. Romano</name><affiliation><nlm:aff id="I1">Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="I2">Ospedale Ferrarotto, Divisione di Ematologia, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="I3">Scuola Superiore di Catania, Via San Nullo 5 I, 95125 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Marescalco, M S" sort="Marescalco, M S" uniqKey="Marescalco M" first="M. S." last="Marescalco">M. S. Marescalco</name><affiliation><nlm:aff id="I1">Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="I3">Scuola Superiore di Catania, Via San Nullo 5 I, 95125 Catania, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="I4">1st Section of Dentistry, Department of Surgery, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Liardo, Chiara" sort="Liardo, Chiara" uniqKey="Liardo C" first="Chiara" last="Liardo">Chiara Liardo</name><affiliation><nlm:aff id="I4">1st Section of Dentistry, Department of Surgery, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Villari, L" sort="Villari, L" uniqKey="Villari L" first="L." last="Villari">L. Villari</name><affiliation><nlm:aff id="I5">Division of Pathology, AOU “Policlinico-Vittorio Emanuele”, Via Plebiscito 628, 95100 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Vetro, C" sort="Vetro, C" uniqKey="Vetro C" first="C." last="Vetro">C. Vetro</name><affiliation><nlm:aff id="I1">Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="I3">Scuola Superiore di Catania, Via San Nullo 5 I, 95125 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Conticello, C" sort="Conticello, C" uniqKey="Conticello C" first="C." last="Conticello">C. Conticello</name><affiliation><nlm:aff id="I1">Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Di Raimondo, F" sort="Di Raimondo, F" uniqKey="Di Raimondo F" first="F." last="Di Raimondo">F. Di Raimondo</name><affiliation><nlm:aff id="I1">Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Ferlito, S" sort="Ferlito, S" uniqKey="Ferlito S" first="S." last="Ferlito">S. Ferlito</name><affiliation><nlm:aff id="I4">1st Section of Dentistry, Department of Surgery, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">25506443</idno><idno type="pmc">4260434</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4260434</idno><idno type="RBID">PMC:4260434</idno><idno type="doi">10.1155/2014/529452</idno><date when="2014">2014</date><idno type="wicri:Area/Pmc/Corpus">002A78</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002A78</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Oral Lesion as Unusual First Manifestation of Multiple Myeloma: Case Reports and Review of the Literature</title><author><name sortKey="Romano, A" sort="Romano, A" uniqKey="Romano A" first="A." last="Romano">A. Romano</name><affiliation><nlm:aff id="I1">Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="I2">Ospedale Ferrarotto, Divisione di Ematologia, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="I3">Scuola Superiore di Catania, Via San Nullo 5 I, 95125 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Marescalco, M S" sort="Marescalco, M S" uniqKey="Marescalco M" first="M. S." last="Marescalco">M. S. Marescalco</name><affiliation><nlm:aff id="I1">Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="I3">Scuola Superiore di Catania, Via San Nullo 5 I, 95125 Catania, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="I4">1st Section of Dentistry, Department of Surgery, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Liardo, Chiara" sort="Liardo, Chiara" uniqKey="Liardo C" first="Chiara" last="Liardo">Chiara Liardo</name><affiliation><nlm:aff id="I4">1st Section of Dentistry, Department of Surgery, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Villari, L" sort="Villari, L" uniqKey="Villari L" first="L." last="Villari">L. Villari</name><affiliation><nlm:aff id="I5">Division of Pathology, AOU “Policlinico-Vittorio Emanuele”, Via Plebiscito 628, 95100 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Vetro, C" sort="Vetro, C" uniqKey="Vetro C" first="C." last="Vetro">C. Vetro</name><affiliation><nlm:aff id="I1">Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="I3">Scuola Superiore di Catania, Via San Nullo 5 I, 95125 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Conticello, C" sort="Conticello, C" uniqKey="Conticello C" first="C." last="Conticello">C. Conticello</name><affiliation><nlm:aff id="I1">Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Di Raimondo, F" sort="Di Raimondo, F" uniqKey="Di Raimondo F" first="F." last="Di Raimondo">F. Di Raimondo</name><affiliation><nlm:aff id="I1">Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation></author><author><name sortKey="Ferlito, S" sort="Ferlito, S" uniqKey="Ferlito S" first="S." last="Ferlito">S. Ferlito</name><affiliation><nlm:aff id="I4">1st Section of Dentistry, Department of Surgery, University of Catania, Via Citelli 6, 95124 Catania, Italy</nlm:aff></affiliation></author></analytic><series><title level="j">Case Reports in Hematology</title><idno type="ISSN">2090-6560</idno><idno type="eISSN">2090-6579</idno><imprint><date when="2014">2014</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Extramedullary plasmacytoma (EMP) and solitary bone plasmacytoma (SBP) represent a disease continuum through a multistage process of cell differentiation, survival, proliferation, and dissemination, strictly related to multiple myeloma (MM), the second most common hematological malignancy. Herein, we report two cases of recurrent oral plasmacytoma progressed to MM, in which the first clinical sign of a more widespread disease was limited to the mouth. Based on our experience, we recommend a strict workup for the differential diagnosis between EMP, SBP, and MM for patients with oral plasmacytoma, including radiological exam of the skeleton, magnetic resonance imaging (MRI) of the bone, and positive emission tomography (FDG-PET). MRI and possibly PET can all be used to more sensitively detect EM plasmacytoma sites.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Liebross, R H" uniqKey="Liebross R">R. H. Liebross</name></author><author><name sortKey="Ha, C S" uniqKey="Ha C">C. S. Ha</name></author><author><name sortKey="Cox, J D" uniqKey="Cox J">J. D. Cox</name></author><author><name sortKey="Weber, D" uniqKey="Weber D">D. Weber</name></author><author><name sortKey="Delasalle, K" uniqKey="Delasalle K">K. Delasalle</name></author><author><name sortKey="Alexanian, R" uniqKey="Alexanian R">R. Alexanian</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Alexiou, C" uniqKey="Alexiou C">C. Alexiou</name></author><author><name sortKey="Kau, R J" uniqKey="Kau R">R. J. Kau</name></author><author><name sortKey="Dietzfelbinger, H" uniqKey="Dietzfelbinger H">H. Dietzfelbinger</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Tesei, F" uniqKey="Tesei F">F. Tesei</name></author><author><name sortKey="Caliceti, U" uniqKey="Caliceti U">U. Caliceti</name></author><author><name sortKey="Sorrenti, G" uniqKey="Sorrenti G">G. Sorrenti</name></author><author><name sortKey="Canciullo, A" uniqKey="Canciullo A">A. Canciullo</name></author><author><name sortKey="Sabbatini, E" uniqKey="Sabbatini E">E. Sabbatini</name></author><author><name sortKey="Pileri, S" uniqKey="Pileri S">S. Pileri</name></author><author><name sortKey="Neri, S" uniqKey="Neri S">S. Neri</name></author><author><name sortKey="Frezza, G" uniqKey="Frezza G">G. Frezza</name></author><author><name sortKey="Rinaldi Ceroni, A" uniqKey="Rinaldi Ceroni A">A. Rinaldi Ceroni</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Majumdar, S" uniqKey="Majumdar S">S. Majumdar</name></author><author><name sortKey="Raghavan, U" uniqKey="Raghavan U">U. Raghavan</name></author><author><name sortKey="Jones, N S" uniqKey="Jones N">N. S. Jones</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Seoane, J" uniqKey="Seoane J">J. Seoane</name></author><author><name sortKey="De La Cruz, A" uniqKey="De La Cruz A">A. De La Cruz</name></author><author><name sortKey="Pomareda, M" uniqKey="Pomareda M">M. Pomareda</name></author><author><name sortKey="Varela Centelles, P I" uniqKey="Varela Centelles P">P. I. Varela-Centelles</name></author><author><name sortKey="Pias, L" uniqKey="Pias L">L. Pias</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Dimopoulos, M" uniqKey="Dimopoulos M">M. Dimopoulos</name></author><author><name sortKey="Kyle, R" uniqKey="Kyle R">R. Kyle</name></author><author><name sortKey="Fermand, J P" uniqKey="Fermand J">J.-P. Fermand</name></author><author><name sortKey="Rajkumar, S V" uniqKey="Rajkumar S">S. V. Rajkumar</name></author><author><name sortKey="San Miguel, J" uniqKey="San Miguel J">J. San Miguel</name></author><author><name sortKey="Chanan Khan, A" uniqKey="Chanan Khan A">A. Chanan-Khan</name></author><author><name sortKey="Ludwig, H" uniqKey="Ludwig H">H. Ludwig</name></author><author><name sortKey="Joshua, D" uniqKey="Joshua D">D. Joshua</name></author><author><name sortKey="Mehta, J" uniqKey="Mehta J">J. Mehta</name></author><author><name sortKey="Gertz, M" uniqKey="Gertz M">M. Gertz</name></author><author><name sortKey="Avet Loiseau, H" uniqKey="Avet Loiseau H">H. Avet-Loiseau</name></author><author><name sortKey="Beksac, M" uniqKey="Beksac M">M. Beksaç</name></author><author><name sortKey="Anderson, K C" uniqKey="Anderson K">K. C. Anderson</name></author><author><name sortKey="Moreau, P" uniqKey="Moreau P">P. Moreau</name></author><author><name sortKey="Singhal, S" uniqKey="Singhal S">S. Singhal</name></author><author><name sortKey="Goldschmidt, H" uniqKey="Goldschmidt H">H. Goldschmidt</name></author><author><name sortKey="Boccadoro, M" uniqKey="Boccadoro M">M. Boccadoro</name></author><author><name sortKey="Kumar, S" uniqKey="Kumar S">S. Kumar</name></author><author><name sortKey="Giralt, S" uniqKey="Giralt S">S. Giralt</name></author><author><name sortKey="Munshi, N C" uniqKey="Munshi N">N. C. Munshi</name></author><author><name sortKey="Jagannath, S" uniqKey="Jagannath S">S. Jagannath</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Van De Donk, N W C J" uniqKey="Van De Donk N">N. W. C. J. van de Donk</name></author><author><name sortKey="Lokhorst, H M" uniqKey="Lokhorst H">H. M. Lokhorst</name></author><author><name sortKey="Anderson, K C" uniqKey="Anderson K">K. C. Anderson</name></author><author><name sortKey="Richardson, P G" uniqKey="Richardson P">P. G. Richardson</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Terpos, E" uniqKey="Terpos E">E. Terpos</name></author><author><name sortKey="Moulopoulos, L A" uniqKey="Moulopoulos L">L. A. Moulopoulos</name></author><author><name sortKey="Dimopoulos, M A" uniqKey="Dimopoulos M">M. A. Dimopoulos</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Rodriguez Caballero, B" uniqKey="Rodriguez Caballero B">B. Rodríguez-Caballero</name></author><author><name sortKey="Sanchez Santolino, S" uniqKey="Sanchez Santolino S">S. Sanchez-Santolino</name></author><author><name sortKey="Garcia Montesinos Perea, B" uniqKey="Garcia Montesinos Perea B">B. García-Montesinos-Perea</name></author><author><name sortKey="Garcia Reija, M F" uniqKey="Garcia Reija M">M.-F. Garcia-Reija</name></author><author><name sortKey="G Mez Roman, J" uniqKey="G Mez Roman J">J. Gómez-Román</name></author><author><name sortKey="Saiz Bustillo, R" uniqKey="Saiz Bustillo R">R. Saiz-Bustillo</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Seoane, J" uniqKey="Seoane J">J. Seoane</name></author><author><name sortKey="Aguirre Urizar, J M" uniqKey="Aguirre Urizar J">J. M. Aguirre Urizar</name></author><author><name sortKey="Esparza G Mez, G" uniqKey="Esparza G Mez G">G. Esparza Gómez</name></author><author><name sortKey="Suarez Cunqueiro, M" uniqKey="Suarez Cunqueiro M">M. Suárez Cunqueiro</name></author><author><name sortKey="Campos Trapero, J" uniqKey="Campos Trapero J">J. Campos Trapero</name></author><author><name sortKey="Pomareda, M" uniqKey="Pomareda M">M. Pomareda</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Pisano, J J" uniqKey="Pisano J">J. J. Pisano</name></author><author><name sortKey="Coupland, R" uniqKey="Coupland R">R. Coupland</name></author><author><name sortKey="Chen, S Y" uniqKey="Chen S">S.-Y. Chen</name></author><author><name sortKey="Miller, A S" uniqKey="Miller A">A. S. Miller</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Lae, M E" uniqKey="Lae M">M. E. Lae</name></author><author><name sortKey="Vencio, E F" uniqKey="Vencio E">E. F. Vencio</name></author><author><name sortKey="Inwards, C Y" uniqKey="Inwards C">C. Y. Inwards</name></author><author><name sortKey="Unni, K K" uniqKey="Unni K">K. K. Unni</name></author><author><name sortKey="Nascimento, A G" uniqKey="Nascimento A">A. G. Nascimento</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Ferreira, S" uniqKey="Ferreira S">S. Ferreira</name></author><author><name sortKey="Baldaia, C" uniqKey="Baldaia C">C. Baldaia</name></author><author><name sortKey="Fatela, N" uniqKey="Fatela N">N. Fatela</name></author><author><name sortKey="Ramalho, F" uniqKey="Ramalho F">F. Ramalho</name></author><author><name sortKey="Costa, M A" uniqKey="Costa M">M. A. Costa</name></author><author><name sortKey="Brito, D" uniqKey="Brito D">D. Brito</name></author><author><name sortKey="Esteves, G" uniqKey="Esteves G">G. Esteves</name></author><author><name sortKey="Monteiro, E" uniqKey="Monteiro E">E. Monteiro</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Pinto, L S S" uniqKey="Pinto L">L. S. S. Pinto</name></author><author><name sortKey="Campagnoli, E B" uniqKey="Campagnoli E">E. B. Campagnoli</name></author><author><name sortKey="Leon, J E" uniqKey="Leon J">J. E. Leon</name></author><author><name sortKey="Lopes, M A" uniqKey="Lopes M">M. A. Lopes</name></author><author><name sortKey="Jorge, J" uniqKey="Jorge J">J. Jorge</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Razaq, S" uniqKey="Razaq S">S. Razaq</name></author><author><name sortKey="Kessler, H P" uniqKey="Kessler H">H. P. Kessler</name></author><author><name sortKey="Phillips, W" uniqKey="Phillips W">W. Phillips</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Anil, S" uniqKey="Anil S">S. Anil</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Lesmes, D" uniqKey="Lesmes D">D. Lesmes</name></author><author><name sortKey="Laster, Z" uniqKey="Laster Z">Z. Laster</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Canger, E M" uniqKey="Canger E">E. M. Canger</name></author><author><name sortKey="Celenk, P" uniqKey="Celenk P">P. Celenk</name></author><author><name sortKey="Alkan, A" uniqKey="Alkan A">A. Alkan</name></author><author><name sortKey="Gunhan, O" uniqKey="Gunhan O">O. Günhan</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Lee, S E" uniqKey="Lee S">S. E. Lee</name></author><author><name sortKey="Kim, J H" uniqKey="Kim J">J. H. Kim</name></author><author><name sortKey="Jeon, Y W" uniqKey="Jeon Y">Y. W. Jeon</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kumar, A K L" uniqKey="Kumar A">A. K. L. Kumar</name></author><author><name sortKey="Dakhil, C" uniqKey="Dakhil C">C. Dakhil</name></author><author><name sortKey="Satyan, M T" uniqKey="Satyan M">M. T. Satyan</name></author><author><name sortKey="Haideri, N" uniqKey="Haideri N">N. Haideri</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Katodritou, E" uniqKey="Katodritou E">E. Katodritou</name></author><author><name sortKey="Terpos, E" uniqKey="Terpos E">E. Terpos</name></author><author><name sortKey="Symeonidis, A S" uniqKey="Symeonidis A">A. S. Symeonidis</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Zhang, J" uniqKey="Zhang J">J. Zhang</name></author><author><name sortKey="Zhong, Y" uniqKey="Zhong Y">Y. Zhong</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Gozzetti, A" uniqKey="Gozzetti A">A. Gozzetti</name></author><author><name sortKey="Cerase, A" uniqKey="Cerase A">A. Cerase</name></author><author><name sortKey="Lotti, F" uniqKey="Lotti F">F. Lotti</name></author><author><name sortKey="Rossi, D" uniqKey="Rossi D">D. Rossi</name></author><author><name sortKey="Palumbo, A" uniqKey="Palumbo A">A. Palumbo</name></author><author><name sortKey="Petrucci, M T" uniqKey="Petrucci M">M. T. Petrucci</name></author><author><name sortKey="Patriarca, F" uniqKey="Patriarca F">F. Patriarca</name></author><author><name sortKey="Nozzoli, C" uniqKey="Nozzoli C">C. Nozzoli</name></author><author><name sortKey="Cavo, M" uniqKey="Cavo M">M. Cavo</name></author><author><name sortKey="Offidani, M" uniqKey="Offidani M">M. Offidani</name></author><author><name sortKey="Floridia, M" uniqKey="Floridia M">M. Floridia</name></author><author><name sortKey="Berretta, S" uniqKey="Berretta S">S. Berretta</name></author><author><name sortKey="Vallone, R" uniqKey="Vallone R">R. Vallone</name></author><author><name sortKey="Musto, P" uniqKey="Musto P">P. Musto</name></author><author><name sortKey="Lauria, F" uniqKey="Lauria F">F. Lauria</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Greipp, P R" uniqKey="Greipp P">P. R. Greipp</name></author><author><name sortKey="Miguel, J S" uniqKey="Miguel J">J. S. Miguel</name></author><author><name sortKey="Dune, B G M" uniqKey="Dune B">B. G. M. Dune</name></author><author><name sortKey="Crowley, J J" uniqKey="Crowley J">J. J. Crowley</name></author><author><name sortKey="Barlogie, B" uniqKey="Barlogie B">B. Barlogie</name></author><author><name sortKey="Blade, J" uniqKey="Blade J">J. Bladé</name></author><author><name sortKey="Boccadoro, M" uniqKey="Boccadoro M">M. Boccadoro</name></author><author><name sortKey="Child, J A" uniqKey="Child J">J. A. Child</name></author><author><name sortKey="Harousseau, J L" uniqKey="Harousseau J">J.-L. Harousseau</name></author><author><name sortKey="Kyle, R A" uniqKey="Kyle R">R. A. Kyle</name></author><author><name sortKey="Lahuerta, J J" uniqKey="Lahuerta J">J. J. Lahuerta</name></author><author><name sortKey="Ludwig, H" uniqKey="Ludwig H">H. Ludwig</name></author><author><name sortKey="Morgan, G" uniqKey="Morgan G">G. Morgan</name></author><author><name sortKey="Powles, R" uniqKey="Powles R">R. Powles</name></author><author><name sortKey="Shimizu, K" uniqKey="Shimizu K">K. Shimizu</name></author><author><name sortKey="Shustik, C" uniqKey="Shustik C">C. Shustik</name></author><author><name sortKey="Sonneveld, P" uniqKey="Sonneveld P">P. Sonneveld</name></author><author><name sortKey="Tosi, P" uniqKey="Tosi P">P. Tosi</name></author><author><name sortKey="Turesson, I" uniqKey="Turesson I">I. Turesson</name></author><author><name sortKey="Westin, J" uniqKey="Westin J">J. Westin</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Lambertenghi Deliliers, G" uniqKey="Lambertenghi Deliliers G">G. Lambertenghi-Deliliers</name></author><author><name sortKey="Bruno, E" uniqKey="Bruno E">E. Bruno</name></author><author><name sortKey="Cortelezzi, A" uniqKey="Cortelezzi A">A. Cortelezzi</name></author><author><name sortKey="Fumagalli, L" uniqKey="Fumagalli L">L. Fumagalli</name></author><author><name sortKey="Morosini, A" uniqKey="Morosini A">A. Morosini</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Garcia, C M" uniqKey="Garcia C">C. M. Garcia</name></author><author><name sortKey="Dominguez, J A" uniqKey="Dominguez J">J. A. Dominguez</name></author><author><name sortKey="Roman, P T" uniqKey="Roman P">P. T. Roman</name></author><author><name sortKey="Pando Pinto, J" uniqKey="Pando Pinto J">J. Pando-Pinto</name></author><author><name sortKey="Huelva, A B" uniqKey="Huelva A">A. B. Huelva</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Shortt, C P" uniqKey="Shortt C">C. P. Shortt</name></author><author><name sortKey="Gleeson, T G" uniqKey="Gleeson T">T. G. Gleeson</name></author><author><name sortKey="Breen, K A" uniqKey="Breen K">K. A. Breen</name></author><author><name sortKey="Mchugh, J" uniqKey="Mchugh J">J. McHugh</name></author><author><name sortKey="O Connell, M J" uniqKey="O Connell M">M. J. O'Connell</name></author><author><name sortKey="O Gorman, P J" uniqKey="O Gorman P">P. J. O'Gorman</name></author><author><name sortKey="Eustace, S J" uniqKey="Eustace S">S. J. Eustace</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="D Sa, S" uniqKey="D Sa S">S. D'Sa</name></author><author><name sortKey="Abildgaard, N" uniqKey="Abildgaard N">N. Abildgaard</name></author><author><name sortKey="Tighe, J" uniqKey="Tighe J">J. Tighe</name></author><author><name sortKey="Shaw, P" uniqKey="Shaw P">P. Shaw</name></author><author><name sortKey="Hall Craggs, M" uniqKey="Hall Craggs M">M. Hall-Craggs</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Billecke, L" uniqKey="Billecke L">L. Billecke</name></author><author><name sortKey="Penas, E M M" uniqKey="Penas E">E. M. M. Penas</name></author><author><name sortKey="May, A M" uniqKey="May A">A. M. May</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Billecke, L" uniqKey="Billecke L">L. Billecke</name></author><author><name sortKey="Penas, E M M" uniqKey="Penas E">E. M. M. Penas</name></author><author><name sortKey="May, A M" uniqKey="May A">A. M. May</name></author><author><name sortKey="Engelhardt, M" uniqKey="Engelhardt M">M. Engelhardt</name></author><author><name sortKey="Nagler, A" uniqKey="Nagler A">A. Nagler</name></author><author><name sortKey="Leiba, M" uniqKey="Leiba M">M. Leiba</name></author><author><name sortKey="Schiby, G" uniqKey="Schiby G">G. Schiby</name></author><author><name sortKey="Kroger, N" uniqKey="Kroger N">N. Kröger</name></author><author><name sortKey="Zustin, J" uniqKey="Zustin J">J. Zustin</name></author><author><name sortKey="Marx, A" uniqKey="Marx A">A. Marx</name></author><author><name sortKey="Matschke, J" uniqKey="Matschke J">J. Matschke</name></author><author><name sortKey="Tiemann, M" uniqKey="Tiemann M">M. Tiemann</name></author><author><name sortKey="Goekkurt, E" uniqKey="Goekkurt E">E. Goekkurt</name></author><author><name sortKey="Bokemeyer, C" uniqKey="Bokemeyer C">C. Bokemeyer</name></author><author><name sortKey="Schilling, G" uniqKey="Schilling G">G. Schilling</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Rosi Ol, L" uniqKey="Rosi Ol L">L. Rosiñol</name></author><author><name sortKey="Cibeira, M T" uniqKey="Cibeira M">M. T. Cibeira</name></author><author><name sortKey="Blade, J" uniqKey="Blade J">J. Bladé</name></author><author><name sortKey="Esteve, J" uniqKey="Esteve J">J. Esteve</name></author><author><name sortKey="Aymerich, M" uniqKey="Aymerich M">M. Aymerich</name></author><author><name sortKey="Rozman, M" uniqKey="Rozman M">M. Rozman</name></author><author><name sortKey="Segarra, M" uniqKey="Segarra M">M. Segarra</name></author><author><name sortKey="Cid, M C" uniqKey="Cid M">M. C. Cid</name></author><author><name sortKey="Filella, X" uniqKey="Filella X">X. Filella</name></author><author><name sortKey="Montserrat, E" uniqKey="Montserrat E">E. Montserrat</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Laura, R" uniqKey="Laura R">R. Laura</name></author><author><name sortKey="Cibeira, M T" uniqKey="Cibeira M">M. T. Cibeira</name></author><author><name sortKey="Uriburu, C" uniqKey="Uriburu C">C. Uriburu</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Romano, A" uniqKey="Romano A">A. Romano</name></author><author><name sortKey="Conticello, C" uniqKey="Conticello C">C. Conticello</name></author><author><name sortKey="Di Raimondo, F" uniqKey="Di Raimondo F">F. Di Raimondo</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Blade, J" uniqKey="Blade J">J. Bladé</name></author><author><name sortKey="Fernandez De Larrea, C" uniqKey="Fernandez De Larrea C">C. Fernández de Larrea</name></author><author><name sortKey="Rosi Ol, L" uniqKey="Rosi Ol L">L. Rosiñol</name></author><author><name sortKey="Cibeira, M T" uniqKey="Cibeira M">M. T. Cibeira</name></author><author><name sortKey="Jimenez, R" uniqKey="Jimenez R">R. Jiménez</name></author><author><name sortKey="Powles, R" uniqKey="Powles R">R. Powles</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="research-article"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Case Rep Hematol</journal-id><journal-id journal-id-type="iso-abbrev">Case Rep Hematol</journal-id><journal-id journal-id-type="publisher-id">CRIHEM</journal-id><journal-title-group><journal-title>Case Reports in Hematology</journal-title></journal-title-group><issn pub-type="ppub">2090-6560</issn><issn pub-type="epub">2090-6579</issn><publisher><publisher-name>Hindawi Publishing Corporation</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">25506443</article-id><article-id pub-id-type="pmc">4260434</article-id><article-id pub-id-type="doi">10.1155/2014/529452</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Oral Lesion as Unusual First Manifestation of Multiple Myeloma: Case Reports and Review of the Literature</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Romano</surname><given-names>A.</given-names></name><xref ref-type="aff" rid="I1"><sup>1</sup></xref><xref ref-type="aff" rid="I2"><sup>2</sup></xref><xref ref-type="aff" rid="I3"><sup>3</sup></xref><xref ref-type="corresp" rid="cor1"><sup>*</sup></xref></contrib><contrib contrib-type="author"><name><surname>Marescalco</surname><given-names>M. S.</given-names></name><xref ref-type="aff" rid="I1"><sup>1</sup></xref><xref ref-type="aff" rid="I3"><sup>3</sup></xref><xref ref-type="aff" rid="I4"><sup>4</sup></xref></contrib><contrib contrib-type="author"><name><surname>Liardo</surname><given-names>Chiara</given-names></name><xref ref-type="aff" rid="I4"><sup>4</sup></xref></contrib><contrib contrib-type="author"><name><surname>Villari</surname><given-names>L.</given-names></name><xref ref-type="aff" rid="I5"><sup>5</sup></xref></contrib><contrib contrib-type="author"><name><surname>Vetro</surname><given-names>C.</given-names></name><xref ref-type="aff" rid="I1"><sup>1</sup></xref><xref ref-type="aff" rid="I3"><sup>3</sup></xref></contrib><contrib contrib-type="author"><name><surname>Conticello</surname><given-names>C.</given-names></name><xref ref-type="aff" rid="I1"><sup>1</sup></xref></contrib><contrib contrib-type="author"><name><surname>Di Raimondo</surname><given-names>F.</given-names></name><xref ref-type="aff" rid="I1"><sup>1</sup></xref></contrib><contrib contrib-type="author"><name><surname>Ferlito</surname><given-names>S.</given-names></name><xref ref-type="aff" rid="I4"><sup>4</sup></xref></contrib></contrib-group><aff id="I1"><sup>1</sup>Department of Clinical and Molecular Biomedicine, Section of Haematology, University of Catania, Via Citelli 6, 95124 Catania, Italy</aff><aff id="I2"><sup>2</sup>Ospedale Ferrarotto, Divisione di Ematologia, Via Citelli 6, 95124 Catania, Italy</aff><aff id="I3"><sup>3</sup>Scuola Superiore di Catania, Via San Nullo 5 I, 95125 Catania, Italy</aff><aff id="I4"><sup>4</sup>1st Section of Dentistry, Department of Surgery, University of Catania, Via Citelli 6, 95124 Catania, Italy</aff><aff id="I5"><sup>5</sup>Division of Pathology, AOU “Policlinico-Vittorio Emanuele”, Via Plebiscito 628, 95100 Catania, Italy</aff><author-notes><corresp id="cor1">*A. Romano: <email>sandrina.romano@gmail.com</email></corresp><fn fn-type="other"><p>Academic Editor: Akimichi Ohsaka</p></fn></author-notes><pub-date pub-type="ppub"><year>2014</year></pub-date><pub-date pub-type="epub"><day>25</day><month>11</month><year>2014</year></pub-date><volume>2014</volume><elocation-id>529452</elocation-id><history><date date-type="received"><day>1</day><month>8</month><year>2014</year></date><date date-type="rev-recd"><day>11</day><month>10</month><year>2014</year></date><date date-type="accepted"><day>13</day><month>10</month><year>2014</year></date></history><permissions><copyright-statement>Copyright © 2014 A. Romano et al.</copyright-statement><copyright-year>2014</copyright-year><license xlink:href="https://creativecommons.org/licenses/by/3.0/"><license-p>This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p></license></permissions><abstract><p>Extramedullary plasmacytoma (EMP) and solitary bone plasmacytoma (SBP) represent a disease continuum through a multistage process of cell differentiation, survival, proliferation, and dissemination, strictly related to multiple myeloma (MM), the second most common hematological malignancy. Herein, we report two cases of recurrent oral plasmacytoma progressed to MM, in which the first clinical sign of a more widespread disease was limited to the mouth. Based on our experience, we recommend a strict workup for the differential diagnosis between EMP, SBP, and MM for patients with oral plasmacytoma, including radiological exam of the skeleton, magnetic resonance imaging (MRI) of the bone, and positive emission tomography (FDG-PET). MRI and possibly PET can all be used to more sensitively detect EM plasmacytoma sites.</p></abstract></article-meta></front><body><sec id="sec1"><title>1. Introduction</title><p>Multiple myeloma (MM) is a multifocal plasma cell proliferation in the bone marrow, in which neoplastic cells replace normal marrow cells, produce excess immunoglobulin, and infiltrate the bone. In a few cases, neoplastic plasma cells can be disposed in a localized mass within the bone, in a condition known as solitary bone plasmacytoma [<xref rid="B1" ref-type="bibr">1</xref>] (SBP) or outside thus involving soft tissues, in a condition defined as extramedullary plasmacytoma [<xref rid="B2" ref-type="bibr">2</xref>] (SEP). Clinical manifestations of SEP are generally not specific and may include swelling, pain, numbness, bleeding, mobile teeth, and xerostomia [<xref rid="B3" ref-type="bibr">3</xref>–<xref rid="B5" ref-type="bibr">5</xref>].</p><p>MM is defined by ≥10% of plasma cell infiltration in the bone marrow, ≥30 g/L of monoclonal protein and presence of hypercalcemia, renal insufficiency, anemia, and bone lytic lesions (identified by the acronym of CRAB symptoms) [<xref rid="B6" ref-type="bibr">6</xref>, <xref rid="B7" ref-type="bibr">7</xref>]. Up to 90% MM patients complain of symptoms due to bone disease, defined as multiple destructive lytic lesions of the skeleton, including severe demineralization and osteoporosis through pathological fractures [<xref rid="B8" ref-type="bibr">8</xref>]. Involved areas include skeleton segments at high content of bone marrow such as skull, spine, sternum, vertebrae, pelvis, and hip. Jawbone is MM site in 30% cases [<xref rid="B9" ref-type="bibr">9</xref>, <xref rid="B10" ref-type="bibr">10</xref>]. Oral lesions are seldom the first sign of disease [<xref rid="B11" ref-type="bibr">11</xref>–<xref rid="B14" ref-type="bibr">14</xref>].</p><p>Solitary bone plasmacytoma (SBP) is an area of lytic bone destruction in an otherwise asymptomatic patient, occurring generally in axial skeleton (70% in spine, seldom in the mouth [<xref rid="B15" ref-type="bibr">15</xref>, <xref rid="B16" ref-type="bibr">16</xref>], including the temporomandibular joint [<xref rid="B17" ref-type="bibr">17</xref>, <xref rid="B18" ref-type="bibr">18</xref>]), in absence of plasma cell infiltration of the bone marrow, without serum/urine M-protein. Patients complain of bone pain, and in critical cases, SBP may cause cord/root compression. High dosage radiotherapy (>5000 Gy) is often curative. However, lesions larger than 5 cm, immunoparesis, and persistence of paraprotein after radiotherapy are adverse prognostic factors for progression to MM [<xref rid="B1" ref-type="bibr">1</xref>].</p><p>Extramedullary plasmacytoma (SEP) occurs rarely (<1% of cases) and preferentially at level of head and neck [<xref rid="B3" ref-type="bibr">3</xref>], upper airways including nose-pharynx, tonsils, and paranasal sinus [<xref rid="B4" ref-type="bibr">4</xref>] and rarely at the mouth [<xref rid="B5" ref-type="bibr">5</xref>]. Like in SBP the M-protein is low or absent; plasma cell infiltration in the bone marrow is less than 5%, in absence of end-organ damage and diffuse osteolytic lesions [<xref rid="B2" ref-type="bibr">2</xref>].</p><p>Here we report two cases of oral plasmacytoma, in which the first clinical sign of a more widespread disease was limited to the mouth.</p></sec><sec id="sec2"><title>2. Case A </title><p>A 72-year-old Caucasian male presented to dental division complaining of pain and difficulty in chewing due to an overgrowth of soft tissue located in the alveolar ridge of the left mandible. He reported the extraction of the residual roots of 3.7 which occurred two weeks before. After two days, the alveolar mucosa grew up over the postextraction site. No pain or alterations of gingival mucosa were reported before the extraction of residual roots.</p><p>Extraoral examination revealed normal facial morphology, in absence of lymph node swelling. The orthopantomography did not show any sign of bone involvement. At intraoral examination, a soft mass was present (1.5 cm) with ill-defined contours being pink in colour and slightly ulcerated on top for the action of antagonist dental elements.</p><p>After the patient provided written informed consent, the soft tissue was biopsied. The histological examination revealed a granulation tissue dissociated by lymphoplasmacytic infiltrate. Seven days after the biopsy the soft mass recurred. Three days later, an excisional biopsy was performed (<xref ref-type="fig" rid="fig1">Figure 1</xref>). Histological analysis showed the presence of a poorly differentiated plasma cell neoplasm. Microscopically the tissue appeared heavily infiltrated by neoplastic proliferation with widespread growth, consisting of elements of plasmablastic/plasmacytic morphology and some of them frankly pleomorphic and anaplastic (<xref ref-type="fig" rid="fig2">Figure 2</xref>), compatible with the diagnosis of extramedullary plasmablastic/anaplastic plasmacytoma CD138+, CD38+, EMA+, vimentin+, CD79a weak, CD20−, and CD3−. The patient was therefore sent to the department of haematology for inquiries regarding the stage of disease and for subsequent treatment.</p><p>According to medical history, the patient was diabetic, asthmatic smoker. A comprehensive metabolic panel and complete blood count (CBC) revealed a total protein of 8.2 g/dL, due to a concomitant low amount of iron, without immunoparesis, in absence of a detectable serum or urine paraprotein by immunofixation. The bone marrow biopsy was negative for clonal plasma cells.</p><p>The patient underwent bone imaging: the skeletal survey was negative for osteolytic lesions; the fluorodeoxyglucose-positron emission tomography (FDG-PET) scan was weakly positive at level of mandible and the second cervical vertebra (SUVmax = 2.5), otherwise normal. This finding was confirmed at magnetic resonance imaging (MRI), otherwise normal; thus we considered these findings without an oncologic meaning.</p><p>In absence of CRAB criteria, SEP was diagnosed and the patient received radiotherapy at level of mandible and vertebra. The treatment was complicated by swelling, weight loss, and worsening asthenia.</p><p>Three months later, FDG-PET scan was repeated showing persistence of positivity, but in absence of symptoms.</p><p>Further three months later, clinical conditions worsened; magnetic resonance imaging (MRI) showed nodular lesions with intense enhancement in vertebrae, in the pelvic bone, and in the sternum. FDG-PET scan showed diffusely positive signal in the left jaw, sternum, and ankle, confirming the systemic nature of the disease. The bone marrow biopsy showed an infiltration of 30% monoclonal plasma cells.</p><p>In absence of circulating monoclonal component in serum and urine, with evidence of immunoparesis, nonsecretory MM was diagnosed, stage II in accord with International Staging System [<xref rid="B19" ref-type="bibr">24</xref>]. Laboratory findings are reported in <xref ref-type="table" rid="tab1">Table 1</xref>.</p><p>Two months later, at eight months from the first occurrence of oral plasmacytoma, the patient started systemic chemotherapy with bortezomib 1.3 g/mq at days 1, 4, 8, and 11, thalidomide 100 mg for 21 days, and dexamethasone 40 mg weekly without benefit. The patient died of pneumonia two months later.</p></sec><sec id="sec3"><title>3. Case B</title><p>A 63-year-old Caucasian man, affected by monoclonal gammopathy of undetermined significance (MGUS) since five years, presented with an evolving swelling in the maxilla, at level of left posterior region with involvement of palate and gingiva. At the intraoral examination, the mass consistency was hard elastic and normal in color and covered with intact oral mucosa (<xref ref-type="fig" rid="fig1">Figure 1(c)</xref>).</p><p>The maxillofacial computerized tomography (CT) scan confirmed the presence of a solid neoplasia, with maximum dimensions 25 × 45 mm, causing a structural rearrangement of the bone. An osteolytic involvement of the upper alveolar process and the maxillary sinus was evident, which appeared partially obliterated in its basal portion. The neoplasm was extended to the homolateral nasal cavity with hyperdense swelling at the nose-maxillary region adjoining. Later enlarged cervical and submandibular lymph nodes were evident; the largest had 13 mm diameter.</p><p>The patient reported that the presence of soft tissue in the same site occurred around six months before and was already biopsied, compatible with diagnosis of mucosal plasmacytoma. The histological revision in our division confirmed the low-grade nature of that lesion (weak plasma cell infiltration, presence of small necrosis foci, low proliferation index, 3-4%, immunophenotype: CD138+, CD38+, EMA+, vimentin+, CD79a weak, CD20−, CD3−, CD45RO−, SMA−, myosin−, and S-100−, <xref ref-type="fig" rid="fig2">Figure 2</xref>). Hematological restaging confirmed persistency of MGUS, with immunofixation positive for IgA, stable M-protein (2200 mg/dL), no end-organ damage signs, including bone evaluation with X-ray scan and MRI, and 6% plasma cell infiltration in the bone marrow. Since the absence of organ damage (see biochemistry <xref ref-type="table" rid="tab1">Table 1</xref>) and CRAB symptoms, the patient remained in follow-up without starting therapy.</p><p>Because of the recurrence of oral lesion two months later, new incisional biopsy was given. Histological examination of the 1.5 cm soft tissue fragment showed a massive component of well-differentiated plasma cells, secreting monotypic immunoglobulins with lambda light chain restriction.</p><p>Diagnosis of MGUS progression to extramedullary myeloma stage II was stated. The patient started systemic chemotherapy with lenalidomide 25 mg and dexamethasone 40 mg weekly, achieving partial remission. He is currently under maintenance therapy with 5 mg lenalidomide daily, in good clinical conditions.</p><p>Whereas thalidomide and lenalidomide might be of help in high-risk and EM-myeloma, most often, proteasome inhibitors, such as bortezomib, have more often been described to induce response and seem the better option to use.</p></sec><sec id="sec4"><title>4. Discussion</title><p>Herein, we report two cases of MM patients with unusual extramedullary sites (mouth) in which the systemic disease was anticipated by localized mouse disease. Both patients received first localized treatment (only radiotherapy) and strict follow-up, thus to shift to systemic chemotherapy later.</p><p>Maxillofacial lesions are very rare as first myeloma manifestation (<0.5%) and are more frequent in advanced disease with extensive skeletal involvement, including the skull [<xref rid="B14" ref-type="bibr">14</xref>]. A Japanese series reported oral manifestations of MM in almost 50% of initially diagnosed mouth plasmocytoma [<xref rid="B18" ref-type="bibr">18</xref>].</p><p>At X-ray scans, osteolytic lesions in maxilla appear well defined, without any sign of bone reaction [<xref rid="B10" ref-type="bibr">10</xref>], more frequent in the jaw than in the maxilla [<xref rid="B14" ref-type="bibr">14</xref>, <xref rid="B20" ref-type="bibr">25</xref>].</p><p>As shown in Case A (<xref ref-type="fig" rid="fig3">Figure 3</xref>), evolution of SEP is unpredictable. Once it is diagnosed and treated, the patient may have no symptoms for months or even years [<xref rid="B21" ref-type="bibr">26</xref>]. However, in unfavourable cases, the tumour may spread, developing regional localizations in 35–50% of cases, or evolve into multiple myeloma [<xref rid="B21" ref-type="bibr">26</xref>].</p><p>In patient B, SBP was the first clinical sign of MGUS progression through MM. Clinical investigations conclude that SBP is more likely to transform in MM than SEP, with a period of progression variable from months to a few years [<xref rid="B9" ref-type="bibr">9</xref>]. In the Mayo Clinic's series [<xref rid="B12" ref-type="bibr">12</xref>] including 33 patients affected by SBP, 43% of them converted to MM after a median of 20.7 months.</p><p>As shown in this report, localized PCN can be the first sign of systemic disease, including nonsecretory MM.</p><p>Thus, whoever is involved in dental care should search for MM signs in each patient with mouth plasmacytomas, including bone scan and if clinically indicated MRI and/or FDG-PET. The integrated use of MRI and FDG-PET can help clinicians to exclude other extramedullary localizations [<xref rid="B22" ref-type="bibr">27</xref>, <xref rid="B23" ref-type="bibr">28</xref>]. Patients affected by extramedullary MM, both primary and progressing from SBP/SEP, have still a poor outcome despite the success of novel agents, due to acquired molecular lesion, including TP53 mutations [<xref rid="B24" ref-type="bibr">29</xref>, <xref rid="B25" ref-type="bibr">30</xref>].</p><p>Preliminary observations in extramedullary MM have shown no efficacy of thalidomide [<xref rid="B26" ref-type="bibr">31</xref>] and controversial results for bortezomib [<xref rid="B27" ref-type="bibr">32</xref>, <xref rid="B28" ref-type="bibr">33</xref>], more often associated with relapse with extramedullary spreading. Encouraging reports arise from lenalidomide based regimens, as our Case B confirms.</p><p>Whereas IMIDs (thalidomide, lenalidomide) might be of help in high-risk and EM-myeloma, most often, proteasome inhibitors, such as bortezomib, have more often been described to induce response and seem better to use.</p><p>The incidence of EMPs is 7% to 18% at MM diagnosis and up to 20% at relapse. The current notion that EMPs are more frequent after treatment with novel agents remains to be proven, especially considering that different patterns of disease recurrence can emerge as patients live longer in the era of novel drugs. Possible mechanisms of extramedullary spread include decreased adhesion molecule expression and downregulation of chemokine receptors. High-dose therapy with autologous stem-cell transplantation (ASCT) can overcome the negative prognostic impact of extramedullary disease in younger selected patients. EMPs do not typically respond to thalidomide alone, but, in contrast, responses to bortezomib have been reported. A potential first-line treatment option seems to be a bortezomib-containing regimen followed by ASCT, whenever possible [<xref rid="B29" ref-type="bibr">34</xref>]. <xref ref-type="table" rid="tab2"> Table 2</xref> summarizes recent reports about outcome of EM-myeloma treated with bortezomib, where overall responses can achieve up to 50% [<xref rid="B30" ref-type="bibr">19</xref>–<xref rid="B34" ref-type="bibr">23</xref>].</p></sec><sec id="sec5"><title>5. Conclusions</title><p>SBP and SEP can evolve to MM. MM signs should be evaluated in each patient with mouth plasmacytomas, including bone scan and if clinically indicated MRI and/or FDG-PET. When SBP and SEP progress to MM, novel agents including lenalidomide and bortezomib are safe and efficacious in disease management.</p></sec></body><back><ack><title>Acknowledgment</title><p>Authors would like to commemorate Dr. E. Salomone, who contributed to this paper and passed away unexpectedly.</p></ack><sec sec-type="conflict"><title>Conflict of Interests</title><p>F. Di Raimondo has received honoraria from Janssen-Cilag and Celgene. All others declare no conflict of interests.</p></sec><ref-list><ref id="B1"><label>1</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Liebross</surname><given-names>R. H.</given-names></name><name><surname>Ha</surname><given-names>C. S.</given-names></name><name><surname>Cox</surname><given-names>J. D.</given-names></name><name><surname>Weber</surname><given-names>D.</given-names></name><name><surname>Delasalle</surname><given-names>K.</given-names></name><name><surname>Alexanian</surname><given-names>R.</given-names></name></person-group><article-title>Solitary bone plasmacytoma: outcome and prognostic factors following radiotherapy</article-title><source><italic>International Journal of Radiation Oncology, Biology, Physics</italic></source><year>1998</year><volume>41</volume><issue>5</issue><fpage>1063</fpage><lpage>1067</lpage><pub-id pub-id-type="other">2-s2.0-0032527981</pub-id><pub-id pub-id-type="doi">10.1016/S0360-3016(98)00186-2</pub-id></element-citation></ref><ref id="B2"><label>2</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Alexiou</surname><given-names>C.</given-names></name><name><surname>Kau</surname><given-names>R. J.</given-names></name><name><surname>Dietzfelbinger</surname><given-names>H.</given-names></name><etal></etal></person-group><article-title>Extramedullary plasmacytoma: tumor occurrence and therapeutic concepts</article-title><source><italic>Cancer</italic></source><year>1999</year><volume>85</volume><issue>11</issue><fpage>2305</fpage><lpage>2314</lpage><pub-id pub-id-type="pmid">10357398</pub-id></element-citation></ref><ref id="B3"><label>3</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Tesei</surname><given-names>F.</given-names></name><name><surname>Caliceti</surname><given-names>U.</given-names></name><name><surname>Sorrenti</surname><given-names>G.</given-names></name><name><surname>Canciullo</surname><given-names>A.</given-names></name><name><surname>Sabbatini</surname><given-names>E.</given-names></name><name><surname>Pileri</surname><given-names>S.</given-names></name><name><surname>Neri</surname><given-names>S.</given-names></name><name><surname>Frezza</surname><given-names>G.</given-names></name><name><surname>Rinaldi Ceroni</surname><given-names>A.</given-names></name></person-group><article-title>Extramedullary plasmocytoma (EMP) of the head and neck: a series of 22 cases</article-title><source><italic>Acta Otorhinolaryngologica Italica</italic></source><year>1995</year><volume>15</volume><issue>6</issue><fpage>437</fpage><lpage>442</lpage><pub-id pub-id-type="other">2-s2.0-0029456304</pub-id><pub-id pub-id-type="pmid">8711997</pub-id></element-citation></ref><ref id="B4"><label>4</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Majumdar</surname><given-names>S.</given-names></name><name><surname>Raghavan</surname><given-names>U.</given-names></name><name><surname>Jones</surname><given-names>N. S.</given-names></name></person-group><article-title>Solitary plasmacytoma and extramedullary plasmacytoma of the paranasal sinuses and soft palate</article-title><source><italic>Journal of Laryngology and Otology</italic></source><year>2002</year><volume>116</volume><issue>11</issue><fpage>962</fpage><lpage>965</lpage><pub-id pub-id-type="other">2-s2.0-0036852432</pub-id><pub-id pub-id-type="doi">10.1258/00222150260369561</pub-id><pub-id pub-id-type="pmid">12487681</pub-id></element-citation></ref><ref id="B5"><label>5</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Seoane</surname><given-names>J.</given-names></name><name><surname>De La Cruz</surname><given-names>A.</given-names></name><name><surname>Pomareda</surname><given-names>M.</given-names></name><name><surname>Varela-Centelles</surname><given-names>P. I.</given-names></name><name><surname>Pias</surname><given-names>L.</given-names></name></person-group><article-title>Primary extramedullary plasmacytoma of the palate</article-title><source><italic>Otolaryngology—Head and Neck Surgery</italic></source><year>1999</year><volume>120</volume><issue>4</issue><fpage>530</fpage><pub-id pub-id-type="other">2-s2.0-13044305856</pub-id><pub-id pub-id-type="doi">10.1053/hn.1999.v120.a87039</pub-id><pub-id pub-id-type="pmid">10187947</pub-id></element-citation></ref><ref id="B6"><label>6</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Dimopoulos</surname><given-names>M.</given-names></name><name><surname>Kyle</surname><given-names>R.</given-names></name><name><surname>Fermand</surname><given-names>J.-P.</given-names></name><name><surname>Rajkumar</surname><given-names>S. V.</given-names></name><name><surname>San Miguel</surname><given-names>J.</given-names></name><name><surname>Chanan-Khan</surname><given-names>A.</given-names></name><name><surname>Ludwig</surname><given-names>H.</given-names></name><name><surname>Joshua</surname><given-names>D.</given-names></name><name><surname>Mehta</surname><given-names>J.</given-names></name><name><surname>Gertz</surname><given-names>M.</given-names></name><name><surname>Avet-Loiseau</surname><given-names>H.</given-names></name><name><surname>Beksaç</surname><given-names>M.</given-names></name><name><surname>Anderson</surname><given-names>K. C.</given-names></name><name><surname>Moreau</surname><given-names>P.</given-names></name><name><surname>Singhal</surname><given-names>S.</given-names></name><name><surname>Goldschmidt</surname><given-names>H.</given-names></name><name><surname>Boccadoro</surname><given-names>M.</given-names></name><name><surname>Kumar</surname><given-names>S.</given-names></name><name><surname>Giralt</surname><given-names>S.</given-names></name><name><surname>Munshi</surname><given-names>N. C.</given-names></name><name><surname>Jagannath</surname><given-names>S.</given-names></name></person-group><article-title>Consensus recommendations for standard investigative workup: report of the International Myeloma Workshop Consensus Panel 3</article-title><source><italic>Blood</italic></source><year>2011</year><volume>117</volume><issue>18</issue><fpage>4701</fpage><lpage>4705</lpage><pub-id pub-id-type="doi">10.1182/blood-2010-10-299529</pub-id><pub-id pub-id-type="other">2-s2.0-79956001465</pub-id><pub-id pub-id-type="pmid">21292778</pub-id></element-citation></ref><ref id="B7"><label>7</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>van de Donk</surname><given-names>N. W. C. J.</given-names></name><name><surname>Lokhorst</surname><given-names>H. M.</given-names></name><name><surname>Anderson</surname><given-names>K. C.</given-names></name><name><surname>Richardson</surname><given-names>P. G.</given-names></name></person-group><article-title>How I treat plasma cell leukemia</article-title><source><italic>Blood</italic></source><year>2012</year><volume>120</volume><issue>12</issue><fpage>2376</fpage><lpage>2389</lpage><pub-id pub-id-type="doi">10.1182/blood-2012-05-408682</pub-id><pub-id pub-id-type="other">2-s2.0-84866605778</pub-id><pub-id pub-id-type="pmid">22837533</pub-id></element-citation></ref><ref id="B8"><label>8</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Terpos</surname><given-names>E.</given-names></name><name><surname>Moulopoulos</surname><given-names>L. A.</given-names></name><name><surname>Dimopoulos</surname><given-names>M. A.</given-names></name></person-group><article-title>Advances in imaging and the management of myeloma bone disease</article-title><source><italic>Journal of Clinical Oncology</italic></source><year>2011</year><volume>29</volume><issue>14</issue><fpage>1907</fpage><lpage>1915</lpage><pub-id pub-id-type="doi">10.1200/JCO.2010.32.5449</pub-id><pub-id pub-id-type="other">2-s2.0-79955842002</pub-id><pub-id pub-id-type="pmid">21483016</pub-id></element-citation></ref><ref id="B9"><label>9</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Rodríguez-Caballero</surname><given-names>B.</given-names></name><name><surname>Sanchez-Santolino</surname><given-names>S.</given-names></name><name><surname>García-Montesinos-Perea</surname><given-names>B.</given-names></name><name><surname>Garcia-Reija</surname><given-names>M.-F.</given-names></name><name><surname>Gómez-Román</surname><given-names>J.</given-names></name><name><surname>Saiz-Bustillo</surname><given-names>R.</given-names></name></person-group><article-title>Mandibular solitary plasmocytoma of the jaw: a case report</article-title><source><italic>Medicina Oral, Patología Oral y Cirugía Bucal</italic></source><year>2011</year><volume>16</volume><issue>5</issue><fpage>e647</fpage><lpage>e650</lpage><pub-id pub-id-type="doi">10.4317/medoral.16952</pub-id><pub-id pub-id-type="other">2-s2.0-80051747360</pub-id></element-citation></ref><ref id="B10"><label>10</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Seoane</surname><given-names>J.</given-names></name><name><surname>Aguirre Urizar</surname><given-names>J. M.</given-names></name><name><surname>Esparza Gómez</surname><given-names>G.</given-names></name><name><surname>Suárez Cunqueiro</surname><given-names>M.</given-names></name><name><surname>Campos Trapero</surname><given-names>J.</given-names></name><name><surname>Pomareda</surname><given-names>M.</given-names></name></person-group><article-title>The spectrum of plasma cell neoplasia in oral pathology</article-title><source><italic>Medicina Oral</italic></source><year>2003</year><volume>8</volume><issue>4</issue><fpage>269</fpage><lpage>280</lpage><pub-id pub-id-type="other">2-s2.0-0042520891</pub-id><pub-id pub-id-type="pmid">12937388</pub-id></element-citation></ref><ref id="B11"><label>11</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Pisano</surname><given-names>J. J.</given-names></name><name><surname>Coupland</surname><given-names>R.</given-names></name><name><surname>Chen</surname><given-names>S.-Y.</given-names></name><name><surname>Miller</surname><given-names>A. S.</given-names></name></person-group><article-title>Plasmacytoma of the oral cavity and jaws: a clinicopathologic study of 13 cases</article-title><source><italic>Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontics</italic></source><year>1997</year><volume>83</volume><issue>2</issue><fpage>265</fpage><lpage>271</lpage><pub-id pub-id-type="doi">10.1016/S1079-2104(97)90015-9</pub-id><pub-id pub-id-type="other">2-s2.0-0031065169</pub-id></element-citation></ref><ref id="B12"><label>12</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Lae</surname><given-names>M. E.</given-names></name><name><surname>Vencio</surname><given-names>E. F.</given-names></name><name><surname>Inwards</surname><given-names>C. Y.</given-names></name><name><surname>Unni</surname><given-names>K. K.</given-names></name><name><surname>Nascimento</surname><given-names>A. G.</given-names></name></person-group><article-title>Myeloma of the jaw bones: a clinicopathologic study of 33 cases</article-title><source><italic>Head & Neck</italic></source><year>2003</year><volume>25</volume><issue>5</issue><fpage>373</fpage><lpage>381</lpage><pub-id pub-id-type="doi">10.1002/hed.10210</pub-id><pub-id pub-id-type="other">2-s2.0-0037404540</pub-id><pub-id pub-id-type="pmid">12692874</pub-id></element-citation></ref><ref id="B13"><label>13</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Ferreira</surname><given-names>S.</given-names></name><name><surname>Baldaia</surname><given-names>C.</given-names></name><name><surname>Fatela</surname><given-names>N.</given-names></name><name><surname>Ramalho</surname><given-names>F.</given-names></name><name><surname>Costa</surname><given-names>M. A.</given-names></name><name><surname>Brito</surname><given-names>D.</given-names></name><name><surname>Esteves</surname><given-names>G.</given-names></name><name><surname>Monteiro</surname><given-names>E.</given-names></name></person-group><article-title>Severe intrahepatic cholestasis, erythrocytosis and hypoglycemia: unusual presenting features of systemic AL amyloidosis</article-title><source><italic>Scandinavian Journal of Gastroenterology</italic></source><year>2008</year><volume>43</volume><issue>3</issue><fpage>375</fpage><lpage>379</lpage><pub-id pub-id-type="doi">10.1080/00365520701679298</pub-id><pub-id pub-id-type="other">2-s2.0-39049099410</pub-id><pub-id pub-id-type="pmid">18938770</pub-id></element-citation></ref><ref id="B14"><label>14</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Pinto</surname><given-names>L. S. S.</given-names></name><name><surname>Campagnoli</surname><given-names>E. B.</given-names></name><name><surname>Leon</surname><given-names>J. E.</given-names></name><name><surname>Lopes</surname><given-names>M. A.</given-names></name><name><surname>Jorge</surname><given-names>J.</given-names></name></person-group><article-title>Maxillary lesion presenting as a first sign of multiple myeloma: case report</article-title><source><italic>Medicina Oral, Patologia Oral y Cirugia Bucal</italic></source><year>2007</year><volume>12</volume><issue>5</issue><fpage>E344</fpage><lpage>E347</lpage><pub-id pub-id-type="other">2-s2.0-34848817109</pub-id></element-citation></ref><ref id="B15"><label>15</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Razaq</surname><given-names>S.</given-names></name><name><surname>Kessler</surname><given-names>H. P.</given-names></name><name><surname>Phillips</surname><given-names>W.</given-names></name></person-group><article-title>Oral and maxillofacial pathology case of the month. Solitary plasmacytoma of bone</article-title><source><italic>Texas Dental Journal</italic></source><year>2008</year><volume>125</volume><issue>2</issue><fpage>182</fpage><lpage>190</lpage><pub-id pub-id-type="other">2-s2.0-45149122580</pub-id><pub-id pub-id-type="pmid">18453312</pub-id></element-citation></ref><ref id="B16"><label>16</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Anil</surname><given-names>S.</given-names></name></person-group><article-title>Solitary plasmacytoma of the maxilla—a case report and review of the literature</article-title><source><italic>General Dentistry</italic></source><year>2007</year><volume>55</volume><issue>1</issue><fpage>39</fpage><lpage>43</lpage><pub-id pub-id-type="other">2-s2.0-33846619809</pub-id><pub-id pub-id-type="pmid">17333965</pub-id></element-citation></ref><ref id="B17"><label>17</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Lesmes</surname><given-names>D.</given-names></name><name><surname>Laster</surname><given-names>Z.</given-names></name></person-group><article-title>Plasmacytoma in the temporomandibular joint: a case report</article-title><source><italic>British Journal of Oral and Maxillofacial Surgery</italic></source><year>2008</year><volume>46</volume><issue>4</issue><fpage>322</fpage><lpage>324</lpage><pub-id pub-id-type="doi">10.1016/j.bjoms.2007.06.009</pub-id><pub-id pub-id-type="other">2-s2.0-43049181586</pub-id><pub-id pub-id-type="pmid">17698264</pub-id></element-citation></ref><ref id="B18"><label>18</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Canger</surname><given-names>E. M.</given-names></name><name><surname>Celenk</surname><given-names>P.</given-names></name><name><surname>Alkan</surname><given-names>A.</given-names></name><name><surname>Günhan</surname><given-names>O.</given-names></name></person-group><article-title>Mandibular involvement of solitary plasmocytoma: a case report</article-title><source><italic>Medicina Oral, Patologia Oral y Cirugia Bucal</italic></source><year>2007</year><volume>12</volume><issue>1</issue><fpage>E7</fpage><lpage>E9</lpage><pub-id pub-id-type="other">2-s2.0-34047097302</pub-id></element-citation></ref><ref id="B30"><label>19</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Lee</surname><given-names>S. E.</given-names></name><name><surname>Kim</surname><given-names>J. H.</given-names></name><name><surname>Jeon</surname><given-names>Y. W.</given-names></name><etal></etal></person-group><article-title>Impact of extramedullary plasmacytomas on outcomes according to treatment approach in newly diagnosed symptomatic multiple myeloma</article-title><source><italic>Annals of Hematology</italic></source><year>2014</year><pub-id pub-id-type="doi">10.1007/s00277-014-2216-8</pub-id></element-citation></ref><ref id="B31"><label>20</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Kumar</surname><given-names>A. K. L.</given-names></name><name><surname>Dakhil</surname><given-names>C.</given-names></name><name><surname>Satyan</surname><given-names>M. T.</given-names></name><name><surname>Haideri</surname><given-names>N.</given-names></name></person-group><article-title>Extramedullary progression of multiple myeloma despite concomitant medullary response to multiple combination therapies and autologous transplant: a case report</article-title><source><italic>Journal of Medical Case Reports</italic></source><year>2014</year><volume>8, article 299</volume><pub-id pub-id-type="doi">10.1186/1752-1947-8-299</pub-id></element-citation></ref><ref id="B32"><label>21</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Katodritou</surname><given-names>E.</given-names></name><name><surname>Terpos</surname><given-names>E.</given-names></name><name><surname>Symeonidis</surname><given-names>A. S.</given-names></name><etal></etal></person-group><article-title>Clinical features, outcome, and prognostic factors for survival and evolution to multiple myeloma of solitary plasmacytomas: a report of the Greek myeloma study group in 97 patients</article-title><source><italic>American Journal of Hematology</italic></source><year>2014</year><volume>89</volume><issue>8</issue><fpage>803</fpage><lpage>808</lpage><pub-id pub-id-type="doi">10.1002/ajh.23745</pub-id><pub-id pub-id-type="pmid">24757085</pub-id></element-citation></ref><ref id="B33"><label>22</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Zhang</surname><given-names>J.</given-names></name><name><surname>Zhong</surname><given-names>Y.</given-names></name></person-group><article-title>Clinical analysis of 36 multiple myeloma patients with extramedullary plasmacytoma invasion of the spinal canal</article-title><source><italic>Hematological Oncology</italic></source><year>2014</year><pub-id pub-id-type="doi">10.1002/hon.2126</pub-id><pub-id pub-id-type="other">2-s2.0-84891714857</pub-id></element-citation></ref><ref id="B34"><label>23</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Gozzetti</surname><given-names>A.</given-names></name><name><surname>Cerase</surname><given-names>A.</given-names></name><name><surname>Lotti</surname><given-names>F.</given-names></name><name><surname>Rossi</surname><given-names>D.</given-names></name><name><surname>Palumbo</surname><given-names>A.</given-names></name><name><surname>Petrucci</surname><given-names>M. T.</given-names></name><name><surname>Patriarca</surname><given-names>F.</given-names></name><name><surname>Nozzoli</surname><given-names>C.</given-names></name><name><surname>Cavo</surname><given-names>M.</given-names></name><name><surname>Offidani</surname><given-names>M.</given-names></name><name><surname>Floridia</surname><given-names>M.</given-names></name><name><surname>Berretta</surname><given-names>S.</given-names></name><name><surname>Vallone</surname><given-names>R.</given-names></name><name><surname>Musto</surname><given-names>P.</given-names></name><name><surname>Lauria</surname><given-names>F.</given-names></name></person-group><article-title>Extramedullary intracranial localization of multiple myeloma and treatment with novel agents: a retrospective survey of 50 patients</article-title><source><italic>Cancer</italic></source><year>2012</year><volume>118</volume><issue>6</issue><fpage>1574</fpage><lpage>1584</lpage><pub-id pub-id-type="doi">10.1002/cncr.26447</pub-id><pub-id pub-id-type="other">2-s2.0-84857996397</pub-id><pub-id pub-id-type="pmid">21932386</pub-id></element-citation></ref><ref id="B19"><label>24</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Greipp</surname><given-names>P. R.</given-names></name><name><surname>Miguel</surname><given-names>J. S.</given-names></name><name><surname>Dune</surname><given-names>B. G. M.</given-names></name><name><surname>Crowley</surname><given-names>J. J.</given-names></name><name><surname>Barlogie</surname><given-names>B.</given-names></name><name><surname>Bladé</surname><given-names>J.</given-names></name><name><surname>Boccadoro</surname><given-names>M.</given-names></name><name><surname>Child</surname><given-names>J. A.</given-names></name><name><surname>Harousseau</surname><given-names>J.-L.</given-names></name><name><surname>Kyle</surname><given-names>R. A.</given-names></name><name><surname>Lahuerta</surname><given-names>J. J.</given-names></name><name><surname>Ludwig</surname><given-names>H.</given-names></name><name><surname>Morgan</surname><given-names>G.</given-names></name><name><surname>Powles</surname><given-names>R.</given-names></name><name><surname>Shimizu</surname><given-names>K.</given-names></name><name><surname>Shustik</surname><given-names>C.</given-names></name><name><surname>Sonneveld</surname><given-names>P.</given-names></name><name><surname>Tosi</surname><given-names>P.</given-names></name><name><surname>Turesson</surname><given-names>I.</given-names></name><name><surname>Westin</surname><given-names>J.</given-names></name></person-group><article-title>International staging system for multiple myeloma</article-title><source><italic>Journal of Clinical Oncology</italic></source><year>2005</year><volume>23</volume><issue>15</issue><fpage>3412</fpage><lpage>3420</lpage><pub-id pub-id-type="doi">10.1200/JCO.2005.04.242</pub-id><pub-id pub-id-type="other">2-s2.0-20644460600</pub-id><pub-id pub-id-type="pmid">15809451</pub-id></element-citation></ref><ref id="B20"><label>25</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Lambertenghi-Deliliers</surname><given-names>G.</given-names></name><name><surname>Bruno</surname><given-names>E.</given-names></name><name><surname>Cortelezzi</surname><given-names>A.</given-names></name><name><surname>Fumagalli</surname><given-names>L.</given-names></name><name><surname>Morosini</surname><given-names>A.</given-names></name></person-group><article-title>Incidence of jaw lesions in 193 patients with multiple myeloma</article-title><source><italic>Oral Surgery Oral Medicine and Oral Pathology</italic></source><year>1988</year><volume>65</volume><issue>5</issue><fpage>533</fpage><lpage>537</lpage><pub-id pub-id-type="doi">10.1016/0030-4220(88)90135-1</pub-id><pub-id pub-id-type="other">2-s2.0-0023880875</pub-id></element-citation></ref><ref id="B21"><label>26</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Garcia</surname><given-names>C. M.</given-names></name><name><surname>Dominguez</surname><given-names>J. A.</given-names></name><name><surname>Roman</surname><given-names>P. T.</given-names></name><name><surname>Pando-Pinto</surname><given-names>J.</given-names></name><name><surname>Huelva</surname><given-names>A. B.</given-names></name></person-group><article-title>Primary extramedullary plasmocytoma of the tongue base</article-title><source><italic>Acta Otorrinolaringologica Espanola</italic></source><year>1999</year><volume>50</volume><issue>8</issue><fpage>657</fpage><lpage>659</lpage><pub-id pub-id-type="other">2-s2.0-0033389237</pub-id><pub-id pub-id-type="pmid">10619905</pub-id></element-citation></ref><ref id="B22"><label>27</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Shortt</surname><given-names>C. P.</given-names></name><name><surname>Gleeson</surname><given-names>T. G.</given-names></name><name><surname>Breen</surname><given-names>K. A.</given-names></name><name><surname>McHugh</surname><given-names>J.</given-names></name><name><surname>O'Connell</surname><given-names>M. J.</given-names></name><name><surname>O'Gorman</surname><given-names>P. J.</given-names></name><name><surname>Eustace</surname><given-names>S. J.</given-names></name></person-group><article-title>Whole-body MRI versus PET in assessment of multiple myeloma disease activity</article-title><source><italic>The American Journal of Roentgenology</italic></source><year>2009</year><volume>192</volume><issue>4</issue><fpage>980</fpage><lpage>986</lpage><pub-id pub-id-type="doi">10.2214/AJR.08.1633</pub-id><pub-id pub-id-type="other">2-s2.0-63849194015</pub-id><pub-id pub-id-type="pmid">19304704</pub-id></element-citation></ref><ref id="B23"><label>28</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>D'Sa</surname><given-names>S.</given-names></name><name><surname>Abildgaard</surname><given-names>N.</given-names></name><name><surname>Tighe</surname><given-names>J.</given-names></name><name><surname>Shaw</surname><given-names>P.</given-names></name><name><surname>Hall-Craggs</surname><given-names>M.</given-names></name></person-group><article-title>Guidelines for the use of imaging in the management of myeloma</article-title><source><italic>British Journal of Haematology</italic></source><year>2007</year><volume>137</volume><issue>1</issue><fpage>49</fpage><lpage>63</lpage><pub-id pub-id-type="doi">10.1111/j.1365-2141.2007.06491.x</pub-id><pub-id pub-id-type="other">2-s2.0-33947268684</pub-id><pub-id pub-id-type="pmid">17359371</pub-id></element-citation></ref><ref id="B24"><label>29</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Billecke</surname><given-names>L.</given-names></name><name><surname>Penas</surname><given-names>E. M. M.</given-names></name><name><surname>May</surname><given-names>A. M.</given-names></name><etal></etal></person-group><article-title>Cytogenetics of extramedullary manifestations in multiple myeloma</article-title><source><italic>British Journal of Haematology</italic></source><year>2013</year><volume>161</volume><issue>1</issue><fpage>87</fpage><lpage>94</lpage><pub-id pub-id-type="doi">10.1111/bjh.12223</pub-id><pub-id pub-id-type="other">2-s2.0-84875054416</pub-id><pub-id pub-id-type="pmid">23368088</pub-id></element-citation></ref><ref id="B25"><label>30</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Billecke</surname><given-names>L.</given-names></name><name><surname>Penas</surname><given-names>E. M. M.</given-names></name><name><surname>May</surname><given-names>A. M.</given-names></name><name><surname>Engelhardt</surname><given-names>M.</given-names></name><name><surname>Nagler</surname><given-names>A.</given-names></name><name><surname>Leiba</surname><given-names>M.</given-names></name><name><surname>Schiby</surname><given-names>G.</given-names></name><name><surname>Kröger</surname><given-names>N.</given-names></name><name><surname>Zustin</surname><given-names>J.</given-names></name><name><surname>Marx</surname><given-names>A.</given-names></name><name><surname>Matschke</surname><given-names>J.</given-names></name><name><surname>Tiemann</surname><given-names>M.</given-names></name><name><surname>Goekkurt</surname><given-names>E.</given-names></name><name><surname>Bokemeyer</surname><given-names>C.</given-names></name><name><surname>Schilling</surname><given-names>G.</given-names></name></person-group><article-title>Similar incidences of TP53 deletions in extramedullary organ infiltrations, soft tissue and osteolyses of patients with multiple myeloma</article-title><source><italic>Anticancer Research</italic></source><year>2012</year><volume>32</volume><issue>5</issue><fpage>2031</fpage><lpage>2034</lpage><pub-id pub-id-type="other">2-s2.0-84861496164</pub-id><pub-id pub-id-type="pmid">22593484</pub-id></element-citation></ref><ref id="B26"><label>31</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Rosiñol</surname><given-names>L.</given-names></name><name><surname>Cibeira</surname><given-names>M. T.</given-names></name><name><surname>Bladé</surname><given-names>J.</given-names></name><name><surname>Esteve</surname><given-names>J.</given-names></name><name><surname>Aymerich</surname><given-names>M.</given-names></name><name><surname>Rozman</surname><given-names>M.</given-names></name><name><surname>Segarra</surname><given-names>M.</given-names></name><name><surname>Cid</surname><given-names>M. C.</given-names></name><name><surname>Filella</surname><given-names>X.</given-names></name><name><surname>Montserrat</surname><given-names>E.</given-names></name></person-group><article-title>Extramedullary multiple myeloma escapes the effect of thalidomide</article-title><source><italic>Haematologica</italic></source><year>2004</year><volume>89</volume><issue>7</issue><fpage>832</fpage><lpage>836</lpage><pub-id pub-id-type="other">2-s2.0-3242763046</pub-id><pub-id pub-id-type="pmid">15257935</pub-id></element-citation></ref><ref id="B27"><label>32</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Laura</surname><given-names>R.</given-names></name><name><surname>Cibeira</surname><given-names>M. T.</given-names></name><name><surname>Uriburu</surname><given-names>C.</given-names></name><etal></etal></person-group><article-title>Bortezomib: an effective agent in extramedullary disease in multiple myeloma</article-title><source><italic>European Journal of Haematology</italic></source><year>2006</year><volume>76</volume><issue>5</issue><fpage>405</fpage><lpage>408</lpage><pub-id pub-id-type="doi">10.1111/j.0902-4441.2005.t01-1-EJH2462.x</pub-id><pub-id pub-id-type="other">2-s2.0-33645722203</pub-id><pub-id pub-id-type="pmid">16529604</pub-id></element-citation></ref><ref id="B28"><label>33</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Romano</surname><given-names>A.</given-names></name><name><surname>Conticello</surname><given-names>C.</given-names></name><name><surname>Di Raimondo</surname><given-names>F.</given-names></name></person-group><article-title>Bortezomib for the treatment of previously untreated multiple myeloma</article-title><source><italic>Immunotherapy</italic></source><year>2013</year><volume>5</volume><issue>4</issue><fpage>327</fpage><lpage>352</lpage><pub-id pub-id-type="doi">10.2217/imt.13.14</pub-id><pub-id pub-id-type="other">2-s2.0-84875959497</pub-id><pub-id pub-id-type="pmid">23557417</pub-id></element-citation></ref><ref id="B29"><label>34</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Bladé</surname><given-names>J.</given-names></name><name><surname>Fernández de Larrea</surname><given-names>C.</given-names></name><name><surname>Rosiñol</surname><given-names>L.</given-names></name><name><surname>Cibeira</surname><given-names>M. T.</given-names></name><name><surname>Jiménez</surname><given-names>R.</given-names></name><name><surname>Powles</surname><given-names>R.</given-names></name></person-group><article-title>Soft-tissue plasmacytomas in multiple myeloma: incidence, mechanisms of extramedullary spread, and treatment approach</article-title><source><italic>Journal of Clinical Oncology</italic></source><year>2011</year><volume>29</volume><issue>28</issue><fpage>3805</fpage><lpage>3812</lpage><pub-id pub-id-type="doi">10.1200/JCO.2011.34.9290</pub-id><pub-id pub-id-type="other">2-s2.0-80053621178</pub-id><pub-id pub-id-type="pmid">21900099</pub-id></element-citation></ref></ref-list></back><floats-group><fig id="fig1" orientation="portrait" position="float"><label>Figure 1</label><caption><p>Extramedullary plasmacytoma, macroscopic features. (a) The soft mass neoplasm appears pink in colour and has floating consistency with a diameter of about 1.5 cm, occupying the area of dental elements 37 and 38. It has ill-defined contours and is slightly ulcerated on the top for the effects of antagonist dental elements, causing algia and difficulty in chewing for the patient. No sign of bone involvement at the orthopantomography (Case A). (b) The patient presented a relapse of the neoplasm seven days after the biopsy, with the enlargement of the lesion to the entire edentulous left side of the jaw, until the first premolar. The mucosa covering the lesion was of irregular colour and the traumatism due to the antagonist dental elements was accentuated (Case A). (c) The mass presented as a swelling in the left posterior region of the maxilla, with maximum dimensions 25 × 45 mm, involving also palate and gingiva. The mass consistency was hard elastic and normal in colour and covered with intact oral mucosa (Case B). (d) The maxillofacial CT scan showed an important osteolytic involvement of the upper alveolar process and the partial obliteration of the basal portion of the left maxillary sinus. The lesion extended also to the homolateral nasal cavity. Concomitant enlargement of lateral cervical and submandibular lymph nodes, with the biggest having 13 mm diameter (Case B).</p></caption><graphic xlink:href="CRIHEM2014-529452.001"></graphic></fig><fig id="fig2" orientation="portrait" position="float"><label>Figure 2</label><caption><p>Extramedullary plasmacytoma, microscopic features. (a) The submucosa was heavily infiltrated by atypical plasma cells (hematoxylin eosine stain × 20, Case A). (b) MUM-1 + atypical plasma cells (nuclear staining pattern) (×40). (c) Some of the neoplastic plasma cells (pleomorphic or showing atypical mitosis stain × 63, Case B). (d) CD138 + atypical plasma cells (membranous staining pattern, ×40, Case B).</p></caption><graphic xlink:href="CRIHEM2014-529452.002"></graphic></fig><fig id="fig3" orientation="portrait" position="float"><label>Figure 3</label><graphic xlink:href="CRIHEM2014-529452.003"></graphic></fig><table-wrap id="tab1" orientation="portrait" position="float"><label>Table 1</label><caption><p>Clinical variables at diagnosis of patients A and B. </p></caption><table frame="hsides" rules="groups"><thead><tr><th align="left" rowspan="1" colspan="1"> </th><th align="center" rowspan="1" colspan="1">Case A</th><th align="center" rowspan="1" colspan="1">Case B</th><th align="center" rowspan="1" colspan="1">Normal range</th></tr></thead><tbody><tr><td align="left" rowspan="1" colspan="1">Hemoglobin, g/L</td><td align="center" rowspan="1" colspan="1">10.5</td><td align="center" rowspan="1" colspan="1">10.6</td><td align="center" rowspan="1" colspan="1">12.1–15.1</td></tr><tr><td align="left" rowspan="1" colspan="1">Serum lactate dehydrogenase, U/L </td><td align="center" rowspan="1" colspan="1">377</td><td align="center" rowspan="1" colspan="1">256</td><td align="center" rowspan="1" colspan="1">256–450</td></tr><tr><td align="left" rowspan="1" colspan="1">Serum beta-2 microglobulin, mg/L </td><td align="center" rowspan="1" colspan="1">2.8</td><td align="center" rowspan="1" colspan="1">2.2</td><td align="center" rowspan="1" colspan="1">1.2–2.4</td></tr><tr><td align="left" rowspan="1" colspan="1">Serum albumin (g/dL)</td><td align="center" rowspan="1" colspan="1">4.0</td><td align="center" rowspan="1" colspan="1">4.1</td><td align="center" rowspan="1" colspan="1">3.5–5.0</td></tr><tr><td align="left" rowspan="1" colspan="1">Serum AST (U/L)</td><td align="center" rowspan="1" colspan="1">14</td><td align="center" rowspan="1" colspan="1">15</td><td align="center" rowspan="1" colspan="1">5–42</td></tr><tr><td align="left" rowspan="1" colspan="1">Serum ALT (U/L)</td><td align="center" rowspan="1" colspan="1">42</td><td align="center" rowspan="1" colspan="1">31</td><td align="center" rowspan="1" colspan="1">5–42</td></tr><tr><td align="left" rowspan="1" colspan="1">Serum calcium (mmol/L)</td><td align="center" rowspan="1" colspan="1">2.22</td><td align="center" rowspan="1" colspan="1">2.40</td><td align="center" rowspan="1" colspan="1">2.15–2.55</td></tr><tr><td align="left" rowspan="1" colspan="1">Serum creatinine (micromol/L)</td><td align="center" rowspan="1" colspan="1">108</td><td align="center" rowspan="1" colspan="1">89</td><td align="center" rowspan="1" colspan="1">60–125</td></tr><tr><td align="left" rowspan="1" colspan="1">Blood urea nitrogen (mmol/L)</td><td align="center" rowspan="1" colspan="1">5.2</td><td align="center" rowspan="1" colspan="1">4.8</td><td align="center" rowspan="1" colspan="1">2.5–8.0</td></tr><tr><td align="left" rowspan="1" colspan="1">ISS stage</td><td align="center" rowspan="1" colspan="1">I</td><td align="center" rowspan="1" colspan="1">I</td><td align="center" rowspan="1" colspan="1">—</td></tr><tr><td align="left" rowspan="1" colspan="1">Durie and Salmon Stage</td><td align="center" rowspan="1" colspan="1">1</td><td align="center" rowspan="1" colspan="1">1</td><td align="center" rowspan="1" colspan="1">—</td></tr></tbody></table></table-wrap><table-wrap id="tab2" orientation="portrait" position="float"><label>Table 2</label><table frame="hsides" rules="groups"><thead><tr><th align="left" rowspan="1" colspan="1">Number of patients</th><th align="center" rowspan="1" colspan="1">Regimen description</th><th align="center" rowspan="1" colspan="1">Response rate</th><th align="center" rowspan="1" colspan="1">Notes </th><th align="center" rowspan="1" colspan="1">Reference</th></tr></thead><tbody><tr><td align="left" rowspan="1" colspan="1">275<break></break>54 w EMP</td><td align="center" rowspan="1" colspan="1">Bortezomib based + ASCT</td><td align="center" rowspan="1" colspan="1">Not reported</td><td align="center" rowspan="1" colspan="1">Retrospective series<break></break>EMP is an independent factor for PFS and OS only in transplant-ineligible patients </td><td align="center" rowspan="1" colspan="1">Lee et al. [<xref rid="B30" ref-type="bibr">19</xref>]</td></tr><tr><td align="center" colspan="5" rowspan="1"><hr></hr></td></tr><tr><td align="left" rowspan="1" colspan="1">1</td><td align="center" rowspan="1" colspan="1">(1) VTD + ASCT <break></break>(2) Bortezomib + tanespimycin</td><td align="center" rowspan="1" colspan="1">Not reported</td><td align="center" rowspan="1" colspan="1">Case report</td><td align="center" rowspan="1" colspan="1">Kumar et al. [<xref rid="B31" ref-type="bibr">20</xref>]</td></tr><tr><td align="center" colspan="5" rowspan="1"><hr></hr></td></tr><tr><td align="left" rowspan="1" colspan="1">97 (plasmacytoma evolved to MM)</td><td align="center" rowspan="1" colspan="1">Radiotherapy versus novel agents/chemotherapy</td><td align="center" rowspan="1" colspan="1">91.8%</td><td align="center" rowspan="1" colspan="1">Retrospective series<break></break>Immunoparesis was the only negative predictor of progression to MM</td><td align="center" rowspan="1" colspan="1">Katodritou et al. [<xref rid="B32" ref-type="bibr">21</xref>]</td></tr><tr><td align="center" colspan="5" rowspan="1"><hr></hr></td></tr><tr><td align="left" rowspan="1" colspan="1">36<break></break>(EMP with invasion of the spinal cord)</td><td align="center" rowspan="1" colspan="1">Bortezomib based</td><td align="center" rowspan="1" colspan="1">13/36 (including complete remission)</td><td align="center" rowspan="1" colspan="1"> </td><td align="center" rowspan="1" colspan="1">Zhang and Zhong [<xref rid="B33" ref-type="bibr">22</xref>]</td></tr><tr><td align="center" colspan="5" rowspan="1"><hr></hr></td></tr><tr><td align="left" rowspan="1" colspan="1">50<break></break>(intracranial MM)</td><td align="center" rowspan="1" colspan="1">Bortezomib based (<italic>n</italic> = 29)</td><td align="center" rowspan="1" colspan="1">18/29 (CR + VGPR)</td><td align="center" rowspan="1" colspan="1">Retrospective series</td><td align="center" rowspan="1" colspan="1">Gozzetti et al. [<xref rid="B34" ref-type="bibr">23</xref>]</td></tr></tbody></table></table-wrap></floats-group></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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