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Onodi cell mucocele: Case report and review of the literature

September 19, 2016  |  John M. Lee, MSc, MD; Michael Au, MD


Onodi cell mucoceles are rare entities that can cause devastating ocular complications if not treated promptly. Delays in the diagnosis are possible because of the wide range of differential diagnoses of unilateral retrobulbar optic neuropathy. We describe a new case of Onodi cell mucocele in a 39-year-old woman, and we present a comprehensive review of the literature on this entity. To the best of our knowledge, no review of Onodi cell mucoceles has been previously published. Our review found that 69% of patients with an Onodi cell mucocele experienced an improvement in vision after surgical decompression. The vast majority of these patients underwent endoscopic decompression; the timing of surgical decompression did not appear to affect outcomes in terms of vision. Onodi cell mucocele requires a high degree of clinical suspicion for diagnosis and a multidisciplinary approach to management that involves primary care physicians, ophthalmologists, and otolaryngologists. Early surgical treatment via an endoscopic approach is recommended for most patients, regardless of the duration of their ophthalmologic signs and symptoms.

Isolated epiglottic lymphatic malformation in a 2-year-old: Diagnosis and treatment

September 19, 2016  |  Tate Naylor, BS; Anthony Sheyn, MD; Felicity Lenes-Voit, MD; Eric Berg, MD


Airway obstruction in children has a wide differential diagnosis that includes laryngomalacia, infectious processes, paralysis, extrinsic compression, and other rare anatomic anomalies. Isolated laryngeal lymphatic malformations are rare developments that can manifest with clinically significant airway obstruction. To the best of our knowledge, there have been fewer than 20 reported cases. These laryngeal mucosal lesions are best managed with radiofrequency ablation or laser ablation. We present a case of a 2-year-old child who presented with airway obstruction, initially diagnosed with laryngomalacia, who was subsequently diagnosed and treated for an isolated epiglottic lymphatic malformation.

Chronic invasive fungal sinusitis causing a pathologic Le Fort I fracture in an immunocompetent patient

September 19, 2016  |  Amy L. Richter, MD; K. Kelly Gallagher, MD


We describe the case of a 77-year-old immunocompetent woman with a history of chronic rhinosinusitis who presented with a pathologic Le Fort I fracture after a forceful sneeze. Imaging revealed diffuse sinus opacification and a Le Fort type I complex fracture involving the maxilla, pterygoid plates, clivus, and right nasal bridge. The patient underwent endoscopic debridement of her sinuses, which revealed mucosal dehiscence and otherwise normal healthy bleeding tissue. Anatomic pathology identified necrotic bone with invasive fungal hyphae. Cultures demonstrated Burkholderia cepacia, diphtheroid organisms, and Enterococcus and Serratia spp. The patient was administered an intravenous antibiotic and antifungal for several months, but interval imaging found no significant improvement in bone healing although the stability of her palate had improved on clinical examination. Chronic rhinosinusitis has been found to be a complication of soft-tissue, orbital, and intracranial infections but, to the best of our knowledge, a pathologic facial fracture secondary to chronic invasive fungal and bacterial rhinosinusitis has not been previously reported in the literature.

Decision making in patients with natural myringostapediopexy: A study of the contralateral ear

September 19, 2016  |  Viviane Bom Schmidt, MD; Sady Selaimen da Costa, MD, PhD; Leticia Petersen Schmidt Rosito, MD; Neil Sperling, MD; Rodrigo Goncalves Dias, MD


Naturally occurring myringostapediopexy frequently results in minimal hearing loss and is asymptomatic. Management decisions in such ears, however, often hinge on an appraisal of evolution toward cholesteatoma. The study of the contralateral ear has been used by our research team to infer the progression of chronic otitis media. This cross-sectional, comparative study describes the clinical findings of the contralateral ear in a series of patients with myringostapediopexy. This study included a historical and current sample of 46 patients divided into a pediatric (≤18 years) and an adult group. Patient distribution according to sex was similar (52.2% male), and 56.5% were adults. Mean conductive hearing loss ranged from 14.1 to 21.2 dB in ears with myringostapediopexy and from 16.0 to 26.6 dB in the contralateral ears according to the frequency assessed. The contralateral ear was normal in only 19.6% of the cases of myringostapediopexy. Central tympanic membrane perforation was found in 6.5% of the cases; perforation-retraction, in 17.4%; moderate or severe retraction, in 28.3%; and cholesteatoma, in 28.3%. The prevalence of cholesteatoma in the contralateral ear in the pediatric and adult groups was not significantly different (p = 0.5; χ2 test). The presence of significant abnormalities, particularly cholesteatoma, in the contralateral ears suggests a probable unfavorable progression in cases of myringostapediopexy and may influence management decisions.

Neoplastic causes of nonacute facial paralysis: A review of 221 cases

September 19, 2016  |  John P. Leonetti, MD; Sam J. Marzo, MD; Douglas A. Anderson, MD; Joshua M. Sappington, MD


We conducted a retrospective review to assess the clinical presentation of patients with tumor-related nonacute complete peripheral facial weakness or an incomplete partial facial paresis and to provide an algorithm for the evaluation and management of these patients. Our study population was made up of 221 patients-131 females and 90 males, aged 14 to 79 years (mean: 49.7)-who had been referred to the Facial Nerve Disorders Clinic at our tertiary care academic medical center over a 23-year period with a documented neoplastic cause of facial paralysis. In addition to demographic data, we compiled information on clinical signs and symptoms, radiologic and pathologic findings, and surgical approaches. All patients exhibited gradual-onset facial weakness or facial twitching. Imaging identified an extratemporal tumor in 128 patients (58%), an intratemporal lesion in 55 patients (25%), and an intradural mass in 38 (17%). Almost all of the extratemporal tumors (99%) were malignant, while 91% of the intratemporal and intradural tumors were benign. A transtemporal surgical approach was used in the 93 intratemporal and intradural tumor resections, while the 128 extratemporal lesions required a parotidectomy with partial temporal bone dissection. The vast majority of patients (97%) underwent facial reanimation. We conclude that gradual-onset facial paralysis or twitching may occur as a result of a neoplastic invasion of the facial nerve along its course from the cerebellopontine angle to the parotid gland. We caution readers to beware of a diagnosis of “atypical Bell's palsy.”

A case of radiation-induced mucosal melanoma in an immunohistochemically S-100-negative patient

August 21, 2016  |  Michael Rodriguez, DO; Yash Patil, MD; Arun Gupta, DO, MPH


We report a case of radiation-induced mucosal melanoma in a 41-year-old woman with a history of childhood rhabdomyosarcoma of the nasal cavity that had been treated with radiotherapy. During the workup for the melanoma, the patient was found to be negative for S-100 protein on immunostaining. While many melanotic markers for the histologic confirmation of melanoma exist, they can be negative in some cases, such as ours. To the best of our knowledge, only 1 case of radiation-induced melanoma has been previously reported in the English-language literature, and in that case the patient was S-100-positive. Although our case is rare, it suggests another possible long-term adverse effect of radiotherapy. We also describe the morphologies and histology associated with diagnosing melanoma in an S-100-negative patient.

Blastomycosis of the nose: A case report

August 21, 2016  |  Thomas M. Jetmore, MD; Jennifer Phan, MD; Abideen O. Yekinni, MD, FACS


Blastomyces dermatitidis is a thermally dimorphic fungus endemic to the North American soil near the Mississippi, Ohio, and St. Lawrence river valleys, as well as the Great Lakes Basin. It is responsible for blastomycosis, a systemic pyogranulomatous disease. Blastomycosis of the head and neck is decidedly uncommon and often mistaken for cancer. There are only 3 previous case reports of blastomycosis presenting as an intranasal lesion. We report the unusual case of a 24-year-old man with blastomycosis presenting with a 4-week history of a progressively enlarging intranasal mass who had complete resolution of symptoms after treatment with itraconazole and surgical excision of the lesion.

Surgical management of a high jugular bulb

August 21, 2016  |  Daniel S. Roberts, MD, PhD; Brian Chen, MD; William Slattery, MD

Patient selection for surgical repair of a high dehiscent jugular bulb should include the presence of a codominant sigmoid sinus to avoid elevated postoperative intracranial pressure.

Idiopathic nasal bone osteitis: A preliminary report on a recently recognized entity

August 21, 2016  |  Yotam Shkedy, MD; Ohad Ronen, MD; Sarah Gips, MD; Avi Front†; Tal Marshak, MD; Nechama Uri, MD


Osteitis of the nasal bones is a rare disease; when it occurs, it usually has a known etiology. To the best of our knowledge, idiopathic nasal bone osteitis (NBO) has not been described before. We conducted a study to analyze the behavior of idiopathic NBO and its outcome and to determine a cutoff level for diagnosis using technetium-99m-methylene-diphosphonate (Tc-MDP) bone scanning. This retrospective, controlled analysis involved 9 women (mean age: 44 yr) who had been admitted to an otolaryngology department in Israel with idiopathic NBO over a 5-year period. Clinical evaluation, isotopic evaluation, and computed tomography were done. A lesion-to-nonlesion ratio was calculated between an area of interest on the nasal bone and a reference point (L/R ratio). The same ratio was calculated for a control group of 20 subjects. All the Tc-MDP scans in the study group were suggestive of osteitis. Intravenous antibiotic treatment was effective in all cases. A high degree of accuracy was achieved with cutoff L/R ratios of 2.05 for planar views and 2.34 for single-photon emission computed tomography views.

Balloon dilation of the sphenoid sinus ostium for recurrent sphenoid sinusitis

August 21, 2016  |  Ken Yanagisawa, MD, FACS; Dewey A. Christmas, MD; Joseph P. Mirante, MD, FACS; Eiji Yanagisawa, MD, FACS

The natural sphenoid sinus ostium is often amenable to balloon dilation, which can be performed in the office under local anesthesia or in the operating room under general anesthesia.