Bilateral Mucopyocele of the Torus Tubarius Presenting As Headache

Letters

OBSERVATION ynx and are associated with local trauma, such as surgery, radiation, or neoplastic or infectious processeses.1 Given their Bilateral Mucopyocele of the Torus Tubarius natural history, bilateral acquired lesions are an exceedingly Presenting as Headache uncommon presentation.2 Cystic lesions of the nasopharynx are typically asymptom- Mucoceles are mucus-filled pseudocysts commonly oc- atic and are often discovered incidentally with imaging or en- curring in the oral cavity, including the buccal mucosa, lips, doscopic examination. The etiology of these lesions can be and tongue. The pathophysiologic mechanism relates to trauma either congenital or acquired. Acquired lesions, such as mu- of minor salivary glands allowing extravasation of mucin and coceles, salivary duct cysts, oncocytic (Warthin) cysts, intra- subsequent cyst formation.1 When infected by pathogens, mu- adenoid cysts, and abscesses, occur throughout the nasophar- coceles are referred to as mucopyoceles.3 Untreated mucopyoceles can erode soft tissue and bone Figure 1. Bilateral Nasopharyngeal Lesions Shown on T2-Weighted Axial and extend into adjacent cavities. Critical structures, such as Noncontrast Magnetic Resonance Imaging the brain and orbit, are separated from the sinuses by only a thin layer of mucosa and bone. For this reason, in patients with sinonasal mucopyocele, surgical treatment is indicated to avoid the potentially catastrophic sequelae, including spontaneous cerebrospinal fluid rhinorrhea, orbital, and intracranial infections.4 To our knowledge, there have been no reported cases of bilateral mucopyoceles of the nasopharynx. Herein, we describe the unusual presentation and clinical course of a patient with refractory headaches with inci-

R L dental bilateral nasopharyngeal cysts, appearing to arise from the torus tubarius on brain magnetic resonance imaging (MRI), later confirmed to be mucopyoceles.

Report of a Case | A male veteran in his 40s with a history of post- traumatic stress disorder, traumatic brain injury, and chronic headaches presented to our clinic for evaluation after an MRI ordered for neuropsychiatric evaluation showed bilateral nasopharyngeal lesions (Figure 1) with restricted diffusion on the diffusion weighted sequence. At presentation, the patient reported tension-like headaches for several months, unre- Arrowheads indicate the lesions. lieved by medical treatment. The patient had undergone a ton-

Figure 2. Intraoperative Endoscopic View of the Right Nasopharynx

A Endoscopic view B Suction placed C Draining after biopsy

TT FoR FoR ETO TT

TT NP

RRR

A, Endoscopic view of the right nasopharynx and an enlarged, cystic-appearing torus tubarius (TT). B, Suction is placed in the eustachian tube orifice. C, Draining purulence after biopsy was obtained. ETO indicates eustachian tube orifice; FoR, fossa of Rosenmüeller; NP, nasopharynx.

jamaotolaryngology.com (Reprinted) JAMA Otolaryngology–Head & Neck Surgery January 2016 Volume 142, Number 1 101

Downloaded From: https://jamanetwork.com/ on 10/02/2021 Letters

sillectomy and adenoidectomy in childhood and had not Conflict of Interest Disclosures: None reported. experienced any sinonasal symptoms, radiation exposure, or Funding/Support: The project described herein was partially supported by the trauma. National Institutes of Health (NIH), grant 1TL1TR001443. Findings from a head and neck examination, including flex- Role of the Funder/Sponsor: The NIH had no role in the design and conduct of the study; collection, management, analysis, and interpretation of the data; ible nasopharynolaryngoscopy, showed bilateral fullness of the preparation, review, or approval of the manuscript; and decision to submit the nasopharynx. The submucosal lesions appeared to be within manuscript for publication. the torus tubarii (Figure 2). The differential diagnoses in- Disclaimer: The content is solely the responsibility of the authors and does not cluded mucocele, mucopyocele, adenoidal hypertrophy, and necessarily represent the official views of the NIH. a malignant lesion. Previous Presentation: This study was a poster presentation to the American The patient was subsequently taken to the operating room, Rhinologic Society at the annual American Academy of Otolaryngology–Head and Neck Surgery Foundation Meeting; September 25, 2015; Dallas, Texas where endoscopic biopsies revealed cystic lesions filled with (Abstract 1266). purulence (Figure 2). Suspecting bilateral mucopyocele, the le- 1. Chi AC, Lambert PR III, Richardson MS, Neville BW. Oral mucoceles: sions were marsupialized using microdebrider. Results from a clinicopathologic review of 1,824 cases, including unusual variants. JOral cultures, pathologic examination, and flow cytometry stud- Maxillofac Surg. 2011;69(4):1086-1093. ies were sent for evaluation. The pathology report revealed be- 2. Ben Salem D, Duvillard C, Assous D, Ballester M, Krausé D, Ricolfi F. Imaging nign respiratory mucosa with reactive lymphoid hyperplasia of nasopharyngeal cysts and bursae. Eur Radiol. 2006;16(10):2249-2258. and mild chronic inflammation. Cultures grew Staphylococ- 3. Flint PW, Haughey BH, Lund VJ, et al. Cummings Otolaryngology, Head and cus lugdunensis, consistent with a diagnosis of mucopyocele. Neck Surgery. Philadelphia, PA: Elsevier-Saunders; 2014. On discharge, the patient was prescribed nasal saline irriga- 4. Kechagias E, Georgakoulias N, Ioakimidou C, Kyriazi S, Kontogeorgos G, Seretis A. Giant intradural mucocele in a patient with adult onset seizures. Case tion and a 10-day course of sulfamethoxazole and trimetho- Rep Neurol. 2009;1(1):29-32. prim. At the 6-week follow-up, he reported resolution of his 5. Sekiya K, Watanabe M, Nadgir RN, et al. Nasopharyngeal cystic lesions: headache symptoms, and the operative site was noted to be Tornwaldt and mucous retention cysts of the nasopharynx: findings on MR healing well. imaging. J Comput Assist Tomogr. 2014;38(1):9-13. 6. Mehle ME. What do we know about rhinogenic headache? the Discussion | Nasopharyngeal cysts are a relatively common in- otolaryngologist’s challenge. Otolaryngol Clin North Am. 2014;47(2):255-264. cidental finding on MRI. In a recent review of 3000 random- ized MRI scans, 14% of patients showed evidence of nasopha- COMMENT & RESPONSE ryngeal cysts.5 However, the clinical significance and criteria for intervention for incidental nasopharyngeal lesions are not Caution Against Overinvestigation well established. of Small Thyroid Nodules We present a rare case of bilateral nasopharyngeal muco- To the Editor A recent study by Magister et al1 aimed to determine pyoceles discovered on MRI in a patient with chronic head- whether thyroid nodule size affected fine-needle aspiration bi- ache. The patient endorsed resolution of symptoms follow- opsy (FNAB) results and determined malignancy rates in each ing incision, drainage, and marsupialization of the lesions and Bethesda class. The authors1 found that nodules smaller than 2 antibiotic treatment. cm had increased probability of malignancy irrespective of the We suspect it is possible that even subtle infectious patho- Bethesda class and stated, “our data would suggest that smaller logic abnormalities can trigger a similar inflammatory path- thyroid nodules, as opposed to larger thyroid nodules, pose a rela- way as described herein for rhinogenic pain and manifest as tively increased risk of malignant disease and should be viewed chronic refractory headache.6 Therefore, in instances of head- with caution.”We are concerned that these results can be mis- ache refractory to medical treatment, patients should un- leading to readers and fuel the problem of overinvestigation of dergo a complete head and neck examination including en- thyroid nodules and overdiagnosis of thyroid cancer. doscopy and imaging studies, which may indicate an occult By studying a cohort of patients who had both FNAB and process. Incidental sinonasal abnormalities, like mucopyo- thyroidectomy, Magister et al1 obtained results that are bi- cele, may be clinically significant and warrant further evalu- ased to have a higher malignancy rate in smaller nodules. This ation and treatment. is because recommendations for biopsy require smaller nodules to have more suspicious sonographic features than larger Aria Jafari, MD nodules. For example, the Society of Radiologists in Ultra- Joseph Acevedo, BS sound recommends biopsy for solid nodules 1.5 cm or greater, Marc Lebovits, MD but the size cutoff is 1 cm for nodules with additional findings of microcalcifications, and there is no size cutoff for nodules 2 Author Affiliations: Division of Otolaryngology–Head and Neck Surgery, with associated suspicious lymph nodules. The rationale for Department of Surgery, University of California San Diego, San Diego (Jafari); a size cutoff is not because size predicts malignancy, as sug- School of Medicine, University of California, San Diego, La Jolla (Acevedo); gested by Magister et al,1 but because size affects prognosis Division of Head and Neck Surgery, Department of Surgery, Veterans Affairs if the nodule is malignant. The cohort of Magister et al1 in- Hospital, San Diego, La Jolla, California (Lebovits). cluded a biased sample of small thyroid nodules with addi- Corresponding Author: Aria Jafari, MD, University of California, San Diego, Division of Otolaryngology–Head and Neck Surgery, Department of Surgery, tional suspicious features, and the standard group of larger thy- 200 W Arbor Dr, No. 8895, San Diego, CA 92103 ([email protected]). roid nodules. These results cannot be generalized to patients Published Online: November 12, 2015. doi:10.1001/jamaoto.2015.2568. who receive thyroid ultrasonography in clinical practice.

102 JAMA Otolaryngology–Head & Neck Surgery January 2016 Volume 142, Number 1 (Reprinted) jamaotolaryngology.com

Downloaded From: https://jamanetwork.com/ on 10/02/2021