Otolaryngology Case Reports最新文献

{"title":"Primary parotid angiosarcoma: A case report and literature review","authors":"Lucía Honrubia ,&nbsp;José Maria Verdaguer ,&nbsp;Antonio Rodríguez-Valiente ,&nbsp;Miguel Ángel Sánchez-Pérez ,&nbsp;José Ramón García-Berrocal ,&nbsp;Ángela Maria Harker-Ladino ,&nbsp;José Luis Rodríguez-Carrillo","doi":"10.1016/j.xocr.2025.100722","DOIUrl":"10.1016/j.xocr.2025.100722","url":null,"abstract":"<div><div>Angiosarcoma is a rare, aggressive vascular malignancy that typically affects older men often following radiotherapy. Primary parotid angiosarcoma is uncommon, particularly in patients without known risk factors. We report the case of an 82-year-old woman who presented a parotid mass and a frontal skin lesion, without prior radiotherapy or relevant medical history. Imaging revealed a 4 cm parotid lesion with mandibular involvement but no nodal disease. Fine needle aspiration suggested a malignant epithelioid neoplasm, and PET confirmed uptake in the parotid gland, mandibular condyle, and frontal lesion. The patient underwent total parotidectomy with en bloc resection of the facial nerve and mandibular condyle, ipsilateral functional neck dissection, and excision of the frontal lesion with deep inferior epigastric perforator (DIEP) flap reconstruction. Histopathology confirmed poorly differentiated angiosarcoma with lymphovascular and perineural invasion. Immunohistochemistry was positive for vimentin, CD31, ERG, and CD34, with loss of H3K27me3 expression but no c-MYC amplification. Despite the adjuvant therapy recommendation, the patient developed pulmonary metastases and died three months postoperatively. Angiosarcoma presents with frequent pulmonary metastases and a dismal prognosis. Surgical resection with negative margins and radiotherapy remains the mainstay of treatment. However, survival outcomes remain poor. This case illustrates its aggressive clinical course and distinctive molecular profile, emphasizing the importance of early diagnosis and multidisciplinary treatment.</div></div>","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100722"},"PeriodicalIF":0.3,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145975902","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Oropharyngeal neuroglial heterotopia/choristoma with intracranial and extra-oral extension: A case report","authors":"Yara A. Akkielah ,&nbsp;Lulwah S. Alturki ,&nbsp;Maraam Al Qout ,&nbsp;Wafaa Alshakweer ,&nbsp;Ali Alzughbi ,&nbsp;Abdulelah A. Alluhaybi ,&nbsp;Maqsood Ahmad ,&nbsp;Dana S. Aljomaah ,&nbsp;Ameen S. Binnasser","doi":"10.1016/j.xocr.2026.100724","DOIUrl":"10.1016/j.xocr.2026.100724","url":null,"abstract":"<div><h3>Background</h3><div>Neuroglial heterotopia, also referred to as glial choristoma, is a rare congenital malformation characterized by ectopic glial tissue outside the central nervous system. While most cases are reported in the nasal cavity, non-nasal head and neck presentations are extremely rare. Intracranial extension is even more uncommon, and extra-oral protrusion has not been documented to date.</div></div><div><h3>Case presentation</h3><div>This case reports a 33-day-old male infant born with a large, non-hemorrhagic mass protruding from the oral cavity, diagnosed antenatally and managed via ex utero intrapartum treatment (EXIT). Imaging revealed a solid and cystic lesion extending from the oral cavity into the right middle cranial fossa through a skull base defect, with remodeling of surrounding bony structures. The mass demonstrated both intracranial and extra-oral components, with prominent vascularity. A two-stage surgical approach was performed: initial resection of the intracranial portion via right pterional craniotomy by neurosurgery, followed by a transpalatal excision of the oral mass by otolaryngology. Reconstruction included an inferiorly based palatal flap and acellular dermal matrix grafting. Histopathology confirmed neuroglial heterotopia, with glial tissue lined by stratified squamous epithelium and choroid plexus structures. Immunohistochemistry showed slight increases in Ki-67 and CD45.</div></div><div><h3>Conclusion</h3><div>This case represents the first reported instance of oropharyngeal neuroglial heterotopia with both intracranial extension and protrusion into the oral cavity. Multidisciplinary care with timely surgical intervention and histopathological confirmation is essential for optimal outcomes in such rare and complex congenital anomalies.</div></div>","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100724"},"PeriodicalIF":0.3,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145976490","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"A rare case report of sinonasal mucosal melanoma in the maxillary sinus","authors":"Molham Alahmad ,&nbsp;Venus Hussain Ameen ,&nbsp;Mohammad Jomaa ,&nbsp;Abdulmajeed Yousfan","doi":"10.1016/j.xocr.2025.100712","DOIUrl":"10.1016/j.xocr.2025.100712","url":null,"abstract":"<div><h3>Introduction</h3><div>Sinonasal mucosal melanoma (SNMM) is an aggressive subtype of melanoma that represents &lt;5 % of head and neck melanomas and constitutes less than 1 % of systemic melanoma cases.</div></div><div><h3>Case presentation</h3><div>A 54-year-old female presented with a 5-month history of unilateral recurrent epistaxis, anosmia, and nasal obstruction. Nasal endoscopy showed a bleeding fleshy lesion occupying the right nasal cavity, extending to the nasal floor and nasal septum. Pathological examination and immunohistochemistry (IHC) findings confirmed the diagnosis of SNMM.</div></div><div><h3>Conclusion</h3><div>SNMM in the head and neck region can manifest with nonspecific symptoms. The diagnosis often requires a combination of clinical, pathological, and IHC studies.</div></div>","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100712"},"PeriodicalIF":0.3,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145521194","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Speech outcomes following buccal fat pad interposition in palatal repair: A clinical outcome analysis","authors":"Manu Gupta ,&nbsp;Sanjeev Kumar ,&nbsp;S. Manu Prasad ,&nbsp;Partishtha Sharma ,&nbsp;Harish Kumar Agarwala ,&nbsp;Vivek Kumar Jha","doi":"10.1016/j.xocr.2025.100716","DOIUrl":"10.1016/j.xocr.2025.100716","url":null,"abstract":"<div><h3>Background</h3><div>Cleft palate repair aims not only to achieve anatomical closure but also to restore normal velopharyngeal (VP) function essential for intelligible speech. Despite technical refinements, complications such as fistula formation, wound dehiscence, and persistent velopharyngeal insufficiency (VPI) continue to affect surgical success and speech outcomes. The buccal fat pad (BFP) has emerged as a reliable, well-vascularized graft for intraoral reconstruction, offering potential benefits in enhancing healing and speech function.</div></div><div><h3>Aim</h3><div>To evaluate the effect of BFP interposition during Bardach two-flap palatoplasty on surgical healing, speech outcomes, and velopharyngeal competence in paediatric cleft palate patients.</div></div><div><h3>Materials and methods</h3><div>A prospective case series of 26 patients (17 males, 9 females; mean age 14 ± 2 months) with complete cleft palate underwent Bardach two-flap palatoplasty with BFP interposition between April 2022 and January 2024. Speech evaluations were performed preoperatively and at 3, 6, and 12 months postoperatively using age-appropriate perceptual assessments focusing on resonance, articulation, intelligibility, and VP competence. Postoperative complications, including fistula and donor site morbidity, were recorded. Data were analysed using SPSS v22, with significance set at p &lt; 0.05.</div></div><div><h3>Results</h3><div>BFP provided a consistent, well-vascularized interpositional layer, resulting in satisfactory wound healing. Oronasal fistula occurred in 8 % and wound dehiscence in 4 % of cases. Donor site morbidity was minimal (4 %) and resolved spontaneously. Hypernasality decreased from 76 % preoperatively to 12 % at 12 months, nasal air emission from 64 % to 10 %, and articulation errors from 70 % to 14 %, while speech intelligibility improved from 42 % to 88 % (p &lt; 0.05). Adequate VP closure increased from 23 % preoperatively to 88 % postoperatively.</div></div><div><h3>Conclusion</h3><div>BFP interposition during Bardach two-flap palatoplasty is a safe and effective adjunct for cleft palate repair. It enhances tissue healing, minimizes fistula formation, and significantly improves postoperative speech outcomes and velopharyngeal competence. Given its simplicity, minimal morbidity, and functional advantages, BFP-assisted palatoplasty represents a valuable technique in optimizing both structural and speech outcomes in cleft palate surgery.</div></div>","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100716"},"PeriodicalIF":0.3,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145615992","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Surgical planning for paediatric cochlear implantation in Warsaw Breakage Syndrome (WABS) using 3D reconstructed model of the malformed cochlea with a review of the literature","authors":"Fiona Anna Molnar ,&nbsp;Bence Horvath ,&nbsp;Haadi Muhammad Mollabux ,&nbsp;Roland Nagy ,&nbsp;Balint Posta ,&nbsp;Zsolt Geretovszky ,&nbsp;Eva Perenyi-Csathi ,&nbsp;Livia Ivasko ,&nbsp;Miklos Csanady ,&nbsp;Laszlo Rovo ,&nbsp;Adam Perenyi","doi":"10.1016/j.xocr.2026.100725","DOIUrl":"10.1016/j.xocr.2026.100725","url":null,"abstract":"<div><div>Warsaw Breakage Syndrome (WABS) is an ultra-rare autosomal recessive disorder, presenting with multiple abnormalities including sensorineural hearing loss due to cochlear malformation. Cochlear implant is a viable treatment for hearing loss in these patients. We report an implantation in a 2.91 year old girl with WABS who presented 1 year prior with bilateral severe sensorineural hearing loss. Radiological investigations revealed a bilateral Type-I Cochlear Hypoplasia). Using radiological images, we generated a 3D reconstruction of the labyrinth and auditory ossicles using the 3D Slicer image computing platform. The resulting models were 3D printed from a biocompatible resin and insertion of the cochlear implant electrode array was tested using a range of electrode models. Following this pre-surgical planning, an optimal electrode (Med-El Compressed) was selected based on size and positioning within the cochlea. The patient then underwent cochlear implantation and postoperatively has shown signs of hearing assessed through both electrophysiological and subjective approaches. The patient presented with severe bilateral cochlear malformation - hypoplasia of the basal turns, absent modiolus and cyst-like merging of the 2nd and 3rd turns. The preoperative 3D visualization and printed model were instrumental in electrode array insertion, positioning, and selection of the ideal individual electrode. This consequently led to successful cochlear implantation (proper electrode placement, minimal complications, improved hearing thresholds and successful rehabilitation). Our findings, coupled with a review of similar rare cases in the literature, suggest that this approach could be broadly applicable to paediatric cases of cochlear hypoplasia with the future aim of optimizing postoperative outcomes.</div></div>","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100725"},"PeriodicalIF":0.3,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146037158","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Aural myiasis with tympanic membrane perforation and middle ear involvement","authors":"Elena Seiz,&nbsp;Claudia Scherl,&nbsp;Nicole Rotter,&nbsp;Lena Zaubitzer","doi":"10.1016/j.xocr.2026.100726","DOIUrl":"10.1016/j.xocr.2026.100726","url":null,"abstract":"","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100726"},"PeriodicalIF":0.3,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145975904","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Corrigendum to “Bilateral choanal atresia in an adult: a rare case report and a literature review” [Otolaryngol Case Rep,Volume 36, September 2025, 100677]","authors":"Yasser Al-Ghabra,&nbsp;Aliaa Issa,&nbsp;Khaled Ziadah,&nbsp;Reem Ashreefa,&nbsp;Adham bader aldeen Mohsen","doi":"10.1016/j.xocr.2026.100723","DOIUrl":"10.1016/j.xocr.2026.100723","url":null,"abstract":"","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100723"},"PeriodicalIF":0.3,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146187786","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Misdiagnosed NK/T-cell lymphoma of the larynx mimicking acute laryngitis: A case report and literature review","authors":"Yunuo Xiang,&nbsp;Zhaohui Liu,&nbsp;Tianwen Yang,&nbsp;Yilin Long,&nbsp;Yangchun Zhang,&nbsp;Yongxu Wu,&nbsp;Junfang Lei,&nbsp;Xinrui Qian","doi":"10.1016/j.xocr.2025.100719","DOIUrl":"10.1016/j.xocr.2025.100719","url":null,"abstract":"<div><h3>Objective</h3><div>To explore the clinical features and treatment approaches of NK/T-cell lymphoma in the larynx.</div></div><div><h3>Methods</h3><div>We retrospectively analyzed a patient with laryngeal NK/T-cell lymphoma presenting with symptoms mimicking acute laryngitis (including throat pain and hoarseness) and conducted a review of the relevant literature. The clinical manifestations, diagnosis, treatment, and prognosis were summarized.</div></div><div><h3>Results</h3><div>The patient was discharged against medical advice. Post-discharge pathology confirmed laryngeal NK/T-cell lymphoma. Follow-up conducted six months after discharge revealed that the patient was lost to follow-up.</div></div><div><h3>Conclusion</h3><div>Laryngeal NK/T-cell lymphoma is a rare disease. For patients whose symptoms persist or worsen despite anti-infective or anti-tuberculosis treatment, heightened vigilance is necessary.</div></div>","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100719"},"PeriodicalIF":0.3,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145789677","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Recurrent congenital cholesteatoma in autism spectrum disorder: A rare and complex case report and literature review","authors":"Bilal Hasan ,&nbsp;Zulfiqar Hamdan","doi":"10.1016/j.xocr.2026.100727","DOIUrl":"10.1016/j.xocr.2026.100727","url":null,"abstract":"<div><h3>Introduction</h3><div>Congenital cholesteatoma is a rare but locally aggressive middle ear lesion associated with potentially serious complications. In children with autism spectrum disorder (ASD), diagnostic delay is further compounded by communication and behavioral challenges.</div></div><div><h3>Case presentation</h3><div>A 5-year-old Syrian boy with ASD, previously diagnosed at age two, underwent mastoidectomy for right congenital cholesteatoma discovered incidentally during neuroimaging for developmental delay. Despite prior surgical intervention, he presented three years later with recurrent foul-smelling otorrhea. Imaging revealed a recurrent lesion with ossicular erosion, exposure of the sigmoid sinus, and thinning of the middle cranial fossa plate. Revision surgery via a postauricular approach achieved complete excision, with dural and sinus reconstruction using cartilage and temporalis fascia, along with ossicular chain reconstruction.</div></div><div><h3>Clinical discussion</h3><div>The coexistence of ASD posed additional challenges during clinical evaluation and follow-up. Nevertheless, tailored surgical management resulted in disease control, with no recurrence during two years of follow-up.</div></div><div><h3>Conclusion</h3><div>This case highlights the diagnostic and therapeutic challenges of congenital cholesteatoma in children with ASD. Early imaging, vigilant follow-up, and individualized surgical planning are essential to prevent recurrence and life-threatening complications while improving long-term outcomes.</div></div>","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100727"},"PeriodicalIF":0.3,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145975903","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Expect the unexpected: A rare case of bilateral lymphoepithelial carcinoma of the parotid gland","authors":"Umair Hafeez ,&nbsp;Niall James McInerney ,&nbsp;Sri Ganesh Kalimuthu ,&nbsp;Olena Tkachuk","doi":"10.1016/j.xocr.2025.100718","DOIUrl":"10.1016/j.xocr.2025.100718","url":null,"abstract":"<div><div>Lymphoepithelial carcinoma (LEC) of the salivary glands is a rare and aggressive malignancy strongly associated with Epstein–Barr virus (EBV) infection and more frequently reported in endemic regions such as Greenland and Southeast Asia. Bilateral involvement is exceptionally uncommon, particularly in non-endemic populations. We report the case of a 62-year-old male with bilateral, asymptomatic parotid swellings persisting for five years. Initial clinical assessment suggested a benign etiology, with no pain, facial nerve involvement, or other red flag symptoms. However, ultrasound, fine needle aspiration, and contrast-enhanced computed tomography of the left parotid revealed atypical features suspicious for malignancy, and histopathology confirmed LEC. The patient underwent left superficial parotidectomy with clear margins, followed by right superficial parotidectomy, which also confirmed LEC. This case represents a rare presentation of bilateral LEC in a non-endemic region. The indolent course and absence of classic malignant features highlight the diagnostic challenges of this entity. Early multidisciplinary evaluation is essential for persistent or atypical parotid swellings, and further research is warranted into the role of EBV in tumorigenesis outside endemic areas.</div></div>","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100718"},"PeriodicalIF":0.3,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145736647","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}