CK Foulsham 2nd

E Schwarz

F Barbaccia

I Heinz

I Loennecken

ID O’Neill

J Jordan

J Lundgen

J Tomik

JE Wal van der

P Engel

R Drut

RA Evans

RM Tiwari

S Pätz

Journal of Otolaryngology : Head and Neck Surgery

Lluís Nisa

Basile Nicolas Landis

Cinzia Salmina

Angeliki Ailianou

Eva Karamitopoulou

Roland Giger

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Nisa et al. Journal of Otolaryngology - Head and Neck Surgery  (2015) 44:16 DOI 10.1186/s40463-015-0067-5CASE REPORT Open AccessWarthin’s tumor of the larynx: a very rarecase and systematic review of the literatureLluís Nisa1,2,3, Basile Nicolas Landis1,4, Cinzia Salmina1, Angeliki Ailianou5, Eva Karamitopoulou6 and Roland Giger1*AbstractBackground: Warthin’s tumor or cystadenolymphoma (CAL) is a benign salivary gland tumor occurring almostexclusively in the parotid gland. CALs of other locations are rare.Case presentation: We report a laryngeal CAL detected in a positron emission tomography/computed tomography(PET/CT) performed for breast cancer follow-up. The tumor was successfully treated by transoral surgery.Discussion: Only 14 cases of laryngeal CAL are reported worldwide. These cases confirmed our experience of anuncomplicated and mostly successful transoral resection.Conclusion: CALs of the larynx are very rare. They are characterized by hypermetabolism in PET/CT. The increasing useof PET/CT investigations in cancer patients could give rise to more incidental findings of CALs at unusual locations suchas the larynx.Keywords: Warthin’s tumor, Cystadenolymphoma, Papillary cystadenoma lymphomatosum, Incidentaloma, PET/CT,LarynxBackgroundLaryngeal tumors are predominantly malignant, withsquamous cell carcinoma being the most frequent histo-pathological entity. Benign tumors represent around 5 %of all laryngeal tumors, and most of these are papillomas(more than 80 %).Cystadenolymphoma (CAL) is a benign tumor of thesalivary glands, also known as Warthin’s tumor or papillarycystadenoma lymphomatosum. CAL affects most often theparotid gland, and is the second most frequent benignsalivary gland tumor following pleomorphic adenoma.CALs represent about 10 % of all salivary gland tumors,are most prevalent after the 6th decade of life, have a malepredominance, and are more common in smokers. Ingeneral, salivary gland tumors of the larynx are rare andmalignant forms prevail. Benign salivary gland tumors ofthe larynx are very rare and therefore poorly documented,despite claims that oncocytic lesions occur more frequentlythan previously thought [1].The aim of the present article is to report a case of supra-glottic CAL and review all cases of laryngeal CALs in the* Correspondence: roland.giger@insel.ch1Department of Otorhinolaryngology—Head and Neck Surgery, Inselspital,Bern University Hospital, 3010 Bern, SwitzerlandFull list of author information is available at the end of the article© 2015 Nisa et al. This is an Open Access artic(http://creativecommons.org/licenses/by/4.0),provided the original work is properly creditedcreativecommons.org/publicdomain/zero/1.0/literature in order to discuss presentation, histologicalfeatures, management and outcome.Case presentationA 61-year-old woman was treated for a lobular carcinomaof the left breast in 2010. She had been disease-freesince the end of treatment, but the 2-year follow-up18 F-fluorodeoxyglucose positron emission tomography/computed tomography (PET/CT) showed increaseduptake on the left supraglottic region (Fig. 1). With theexception of some intermittent episodes of hoarseness inthe recent past, she was without symptoms. The patient isan active smoker and free of professional or environmentalexposures.Transnasal fiberoptic laryngoscopy revealed a 2 x 1 cmexpansive mass involving the left side of the supraglottis.Vocal fold mobility was unaffected. A direct laryngoscopyunder general anesthesia was performed (Fig. 2) for biopsy,which revealed a CAL. Complete endoscopic examinationof the upper aerodigestive tract was otherwise normal. AnMRI showed a 2 × 1 × 3.5 cm multi-cystic lesion ofthe left supraglottic region (Fig. 3). Therapeutic exci-sion was performed endoscopically under suspensionmicrolaryngoscopy using CO2 laser. Histopathologicle distributed under the terms of the Creative Commons Attribution Licensewhich permits unrestricted use, distribution, and reproduction in any medium,. The Creative Commons Public Domain Dedication waiver (http://) applies to the data made available in this article, unless otherwise stated.Fig. 1 18F-FDG-PET/CT of the larynx. Intense FDG uptake in the leftsupraglottic region (arrowhead). a) axial, b) coronalNisa et al. Journal of Otolaryngology - Head and Neck Surgery  (2015) 44:16 Page 2 of 5analysis of the resection specimen confirmed thediagnosis of a CAL (Fig. 4). The follow-up was com-plicated by edema of the involved aryepiglottic foldcausing moderate dysphagia three months after sur-gery. A second CO2 laser resection under suspensionmicrolaryngoscopy was performed in order to resectinflammatory and edematous tissue. Further follow-upwas uneventful, without any symptoms or signs of recur-rence after 2 years.Fig. 2 Direct laryngoscopy. A 2 × 1 cm expansive mass on the leftside of the supraglottis (arrowhead; asterisk = anterior commissure)Fig. 3 Fat-suppressed T2 weighted MRI of the larynx. Multi-cystichyperintense lesion occupying the left supraglottic region (arrowhead).a) axial, b) coronalDiscussionWe report a case of CAL of the supraglottic larynxidentified via PET/CT done within the frame of breastcancer follow-up. Additionally, in order to obtain morerelevant information on this seemingly rare entity, we per-formed a literature review without language restriction.Articles were searched in PubMed (up to June 2014) withthe following terms: [(adenolymphoma OR lymphomatouscystadenoma OR cystadenolymphoma OR papillary cysta-denoma lymphomatosum OR Warthin) AND (larynx ORlaryngeal)], and with available cross-linked referencesfrom the articles found. We identified 12 eligible articlesthat reported 14 cases of true laryngeal CAL, histologicallyFig. 4 Histopathology of the laryngeal cystadenolymphoma. H&Estaining of the laryngeal CAL showing oncocytic epithelium (arrowhead)surrounded by lymphoid stroma (asterisk) (magnification 40×)Nisa et al. Journal of Otolaryngology - Head and Neck Surgery  (2015) 44:16 Page 3 of 5defined by the presence of papillary cystic epitheliumand lymphoid stroma (Warthin’s tumor). Availableclinical data were extracted from these articles [2-13]and summarized in Table 1. Our case was added foranalysis purposes.Male to female ratio was 2:13, and patients’ mean agewas of 64.2 ± 10.99 years (range: 45–80). The clinicalpresentation was not disease–but organ-specific, reflectinglaryngeal involvement, with hoarseness as leadingsymptom in 7, dyspnea in 3, and dysphagia in 2 patients.One patient presented with neck swelling. The locationwas exclusively supraglottic, with glottic involvement inonly one of the cases. One case showed a bilateral CAL.Two patients presented CAL within a laryngocele. Tumorsize ranged between 1 and 4.5 cm. In 2 cases, synchronousCALs of the parotid gland were seen. Transoral resectionwas performed in 9 out of 11 patients, and 2 underwentopen transcervical resection. In most cases (9 out of 11reported) one surgical procedure was sufficient to eradicatethe tumor, whereas in one case 4 operations were neededfor recurrent disease and the second case showed tumoralpersistence after 4 months. Our case underwent a secondoperation due to persistent supraglottic edema with noresidual CAL during definitive histopathologic examin-ation. Another 2 cases showed persistent laryngeal edemapostoperatively.In summary, laryngeal CAL is very rare and can mostoften be managed with transoral resection. Recurrenceseems to be an exception in the larynx and only fewcomplications or side effects of the treatment arereported. The unique feature of our reported case isthat the patient’s CAL was detected incidentally dueto regular PET/CT follow-up investigation for a pre-viously treated breast cancer. Only retrospectiveinquiry showed minor laryngeal symptoms consisting ofsome hoarseness episodes. Accurate diagnosis requiresnonetheless histopathologic examination of the resectedspecimens. Malignant degeneration of CALs, originatingfrom both the epithelial and the lymphoid compartments,is a rare but possible event. In the specific case of laryn-geal CALs, no case of malignant degeneration could beidentified.Workup with PET/CT has been shown to indicatefalse positive results in head and neck cancer patientsshowing a CAL within neck lymph nodes mimicking ametastasis [14]. There was similarity with our case,where FDG-uptake was found in the larynx, and initiallymistaken for a breast cancer metastasis. Van der Wal et al.described the largest series of laryngeal CALs (3 cases)along with seven other cases of extraparotideal CALs(cheek, oropharynx, palate, buccal fold, lower lip, subman-dibular gland) [11]. As CALs show a hypermetabolism inPET/CT exams, which are increasingly performed, futurePET findings might bring new insight regarding synchron-ous and multiple Warthin’s tumor locations and intopathogenesis of this particular tumor. While some authorshave classically considered CAL to be more similar to areactive process than to a true neoplasia, the viewson pathogenesis have evolved over the last decades.Current techniques show that the epithelial portion ofa substantial subset of CALs is monoclonal andfrequently harbors a translocation between chromo-somes 11 and 19 t (11;19), which in turn results inthe fusion oncogene CRTC1-MAML2 [CREB(cAMPresponse element-binding protein)-regulated transcriptionco-activator 1–mastermind-like protein 2], which is amaster key involved in several signaling pathways thatcontribute to cell survival and proliferation, primarily theNotch pathway [15]. Van der Wal et al. underlinedthe fact that laryngeal CALs are indeed rare tumorsand pathologic diagnosis can be challenging, especiallywhen it comes to distinguishing CAL from oncocytichyperplasia or metaplasia, pointing out that the onlybasic difference between these entities is the lack oflymphoid stroma in oncocytic lesions [11].Another interesting point is the potential multifocalpresentation of CALs. We found 2 cases of concomi-tant involvement of the larynx and the parotid gland(Table 1) [9, 12]. This is not a new finding, as syn-chronous or metachronous development of CALs inboth parotid glands is not a rare event, which raisesthe question if there is a need for long-term follow-up inpatients with CALs in general. Although speculativein nature, we feel that careful workup in incidentalPET/CT findings could reveal a previously underestimatedamount of extra-salivary and multiple synchronousCAL cases. In light of the open questions regardingthe pathogenesis of CALs, more cases as the presentones could bring further insight into the nature of thisspecial benign tumor.Table 1 Summary of published cases of laryngeal cystadenolymphomaAuthors Age (years)/SexPresentation Location/HistologicfeaturesMaximalsize (cm)Other involvedsitesSurgical approach Nbr. ofoperationsOutcome Time of follow-up(months)CommentsHeinz (1951) [2] 80/F NR Supraglottic 2.5 None Transoraldebulking1 Persistence 4 -Barbaccia et al. (1956) [3] NR/M Dyspnea Supraglottic NR None Tracheotomy,transcervicalresection1 FR 1 -Tiwari and Williams (1969) [4] 72/F Neck swelling Supraglottic/withina laryngocele4.5 None Transcervicalresection1 FR NR -Drut et al. (1975) [5] 70/F Hoarseness Supraglottic NR None Transoralresection1 FR NR -Foulsham et al. (1980) [6] 66/F Hoarseness Supraglottic 1 None Transoralresection1 FR 12 Melkerssonsyndrome6 years beforeEngel et al. (1987) [7] 61/F NR NR NR None Transoralresection1 NR NR -Evans et al. (1989) [8] 45/F Hoarseness Supraglottic NR None Transoralresection4 Recurrence, persistentsupraglottic edemaNR -Loennecken (1989) [9] 77/F HoarsenessDyspneaSupraglottic/withina laryngocele2 Parotid gland Transoralresection1 Persistentsupraglottic edemaNR -Tomik et al. (1991) [10] 72/F HoarsenessDyspneaSupraglottic – glottic NR None Transoralresection1 FR 6 -van der Wal et al. (1993) [11] 54/M NR NR NR None NR NR NR NR -50/F NR NR NR NR NR NR NR NR50/F NR NR NR NR NR NR NR NRPätz and Möbius (1997) [12] 74/F Dysphagia Supraglottic NR Parotid gland Transoralresection1 FR NR -Jordan et al. (1999) [13] 67/F HoarsenessDysphagiaSupraglotticbilaterally/withina laryngocele3 / 2 None NR 1 FR NR -Present report (2014) 61/F Hoarseness Supraglottic 3.5 None Transoralresection2 Persistentsupraglottic edema24 -Abbreviations: M – male; F – female; FR – full recovery; NR – not reportedNisaetal.JournalofOtolaryngology-HeadandNeckSurgery (2015) 44:16 Page4of5Nisa et al. Journal of Otolaryngology - Head and Neck Surgery  (2015) 44:16 Page 5 of 5ConclusionWarthin’s tumors of the larynx are very rare. Transoralresection seems to be feasible for most cases, has littleside effects, and is most often curative. As Warthin’stumors show increased FDG-uptake similar to malignanttumors, it is hypothesized that with increasing PET/CTuse, the number of Warthin’s tumor case reports outsidethe salivary glands may augment.ConsentThe patient has given her consent for the Case report tobe published.Competing interestsThe authors declare that they have no competing interests.Authors’ contributionsLN: conception and design, writer, revision and approval of the final version.BNL, CS, EK, RG: conception and design, revision and approval of the finalversion. AA: revision and approval of the final version.AcknowledgementsThe authors wish to thank Mrs. Irina Ciorba-Nisa, MS, and Mr. KrzysztofSzmigiel, scrub nurse, for their skillful assistance during production of thismanuscript and translation of several articles.Author details1Department of Otorhinolaryngology—Head and Neck Surgery, Inselspital,Bern University Hospital, 3010 Bern, Switzerland. 2Department of ClinicalResearch, University of Bern, 3010 Bern, Switzerland. 3Department ofRadiation Oncology, Inselspital, Bern University Hospital, 3010 Bern,Switzerland. 4Department of Otorhinolaryngology—Head and Neck Surgery,Geneva University Hospital, 1211 Geneva, Switzerland. 5Department ofDiagnostic Radiology, Geneva University Hospital, 1211 Geneva, Switzerland.6Clinical Pathology Division, Institute of Pathology, University of Bern, 3010Bern, Switzerland.Received: 3 December 2014 Accepted: 16 April 2015References1. Lundgen J, Olofsson J, Hellquest H. Oncocytic lesions of the larynx.Acta Otolaryngol. 1928;94:335–44.2. Heinz I. The adenolymphomata. Aust N Z J Surg. 1951;2:47–51.3. Barbaccia F, Micelli F, Tosci C. Adenolinfoma della laringe. Arch Ital OtolRhino Laringol. 1956;67:87–96.4. Tiwari RM, Williams HI. Adenolymphoma in a laryngocele. J Laryngol Otol.1969;83:1227–30.5. Drut R, Di Rago C, Di Rago C. Cistoadenoma papilar linfomatoso(tumor de warthin) de laringe. An Otorinolaringol Ibero Am. 1975;2:29–33.6. Foulsham 2nd CK, Snyder 3rd GG, Carpenter 3rd RJ. Papillary cystadenomalymphomatosum of the larynx. Otolaryngol Head Neck Surg. 1981;89:960–4.7. Engel P, Larsen SB, Francis D. Extraparotid occurrence of papillarylymphomatous cystadenoma-Warthin’s tumor. Ugeskr Laeger.1987;149:1244–6.8. Evans RA, Cassidy MT, Russell TS. Adenolymphoma of the larynx. J R CollSurg Edinb. 1989;34:47.9. Loennecken I. Multilokuläres vorkommen eines zystadenolymphoms(warthin-tumor) in parotis und larynx. Laryngorhinootologie. 1989;68:212–5.10. Tomik J, Papla B, Olszewski E. A case of Warthin’s tumor (papillaryadenolymphoma) of the larynx. Otolaryngol Pol. 1991;45:296–8.11. van der Wal JE, Davids JJ, van der Waal I. Extraparotid Warthin’stumours – report of 10 cases. Br J Oral Maxillofac Surg. 1993;31:43–4.12. Pätz S, Möbius H. Simultanes auftreten eines zystadenolymphoms. HNO.1997;45:91–3.13. Jordan J, Babiński D, Sova J. Adenolymphoma (Warthin’s tumor) of thelarynx: coexistence with the bilateral laryngocele. Contribution to differentialdiagnosis with oncocytic papillary cystadenoma. Otolaryngol Pol.1999;53:213–6.14. Schwarz E, Hürlimann S, Soyka JD, Bortoluzzi L, Strobel K. FDG-positiveWarthin’s tumors in cervical lymph nodes mimicking metastases in tonguecancer staging with PET/CT. Otolaryngol Head Neck Surg. 2009;140:134–5.15. O’Neill ID. New insights into the nature of Warthin’s tumour. J Oral PatholMed. 2009;38:145–9.Submit your next manuscript to BioMed Centraland take full advantage of: • Convenient online submission• Thorough peer review• No space constraints or color ﬁgure charges• Immediate publication on acceptance• Inclusion in PubMed, CAS, Scopus and Google Scholar• Research which is freely available for redistributionSubmit your manuscript at www.biomedcentral.com/submit

Journal of Otolaryngology - Head and Neck Surgery

Warthin’s tumor of the larynx: a very rare case and systematic review of the literature

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Warthin’s tumor of the larynx: a very rare case and systematic review of the literature

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