{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,30]],"date-time":"2026-07-30T15:57:22Z","timestamp":1785427042228,"version":"3.56.0"},"reference-count":49,"publisher":"MDPI AG","issue":"12","license":[{"start":{"date-parts":[[2026,6,9]],"date-time":"2026-06-09T00:00:00Z","timestamp":1780963200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Deanship of Scientific Research at Imam Mohammad Ibn Saud Islamic University","award":["IMSIU-DDRSP2601"],"award-info":[{"award-number":["IMSIU-DDRSP2601"]}]},{"award":["IMSIU-DDRSP2601"],"award-info":[{"award-number":["IMSIU-DDRSP2601"]}],"id":[{"id":"https:\/\/ror.org\/05gxjyb39","id-type":"ROR","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JCM"],"abstract":"<jats:p>Background: Head and neck paragangliomas (HNPGLs) are rare neuroendocrine neoplasms that may be sporadic or hereditary and may occur as isolated, multifocal, or syndromic disease within the pheochromocytoma-paraganglioma spectrum. Synchronous bilateral carotid body tumors (CBTs) and bilateral vagal paragangliomas (VPGLs) are exceptional, particularly when biochemical activity and metastatic nodal involvement coexist. Case Presentation: We report a 33-year-old man with a 2-year history of enlarging bilateral neck masses, positive family history, and markedly elevated noradrenaline (6430 pg\/mL; reference range &lt;750 pg\/mL). Initial CT angiography suggested bilateral CBTs, and Metaiodobenzylguanidine (MIBG) scintigraphy did not demonstrate abnormal adrenal or distant uptake. After alpha- and beta-blockade, staged surgery was performed. Right CBT excision demonstrated metastatic PGL, with two of four lymph nodes positive. Subsequent MRI and operative reassessment revealed synchronous bilateral CBTs and bilateral VPGLs. Left-sided surgery required partial debulking of a vagal-adherent mass to preserve nerve continuity; pathology confirmed PGL with Ki-67 index approximately 2% and left neck nodes were negative. Postoperatively, profound bradycardia required temporary then permanent pacing, together with bilateral vocal cord paralysis. During follow-up, swallowing and voice improved, the pacemaker was removed, and late imaging showed stable residual cervical disease without visceral metastasis on chest\/abdominal CT. Conclusions: This case highlights the diagnostic and therapeutic complexity of multicentric, biochemically active HNPGLs and supports individualized multidisciplinary management, genetic counseling, biochemical surveillance, and long-term follow-up.<\/jats:p>","DOI":"10.3390\/jcm15124463","type":"journal-article","created":{"date-parts":[[2026,6,10]],"date-time":"2026-06-10T06:33:44Z","timestamp":1781073224000},"page":"4463","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Synchronous Bilateral Carotid Body and Bilateral Vagal Paragangliomas: A Case Report and Literature Review"],"prefix":"10.3390","volume":"15","author":[{"given":"Nahar","family":"Alanezi","sequence":"first","affiliation":[{"name":"Department of Surgery, College of Medicine, Imam Mohammad Ibn Saud Islamic University (IMSIU), Riyadh 11564, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yazeed","family":"Alofisan","sequence":"additional","affiliation":[{"name":"Department of Surgery, Division of Vascular Surgery, Security Forces Hospital, Riyadh 11564, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mohammed","family":"Dahman","sequence":"additional","affiliation":[{"name":"Division of Vascular Surgery and Endovascular Therapy, Department of Surgery, King Faisal Specialist Hospital and Research Center (KFSH&RC), Riyadh 12713, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hassan","family":"Gado","sequence":"additional","affiliation":[{"name":"Department of Surgery, Division of Vascular Surgery, Security Forces Hospital, Riyadh 11564, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Majid","family":"Althobaiti","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology-Head and Neck Surgery, Security Forces Hospital Program, Riyadh 11564, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Omar Tawfiq","family":"Abualnasr","sequence":"additional","affiliation":[{"name":"College of Medicine, Alfaisal University, Riyadh 11533, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tariq","family":"Alanezi","sequence":"additional","affiliation":[{"name":"Division of Vascular Surgery, Department of Surgery, University of Toronto, Toronto, ON M5S 1A1, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2026,6,9]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"R153","DOI":"10.1530\/EO-22-0080","article-title":"Paragangliomas of the head and neck: A contemporary review","volume":"2","author":"Graham","year":"2022","journal-title":"Endocr. Oncol."},{"key":"ref_2","unstructured":"Prasad, S.C., Paties, C.T., Pantalone, M.R., Mariani-Costantini, R., and Sanna, M. (2019). Carotid Body and Vagal Paragangliomas: Epidemiology, Genetics, Clinicopathological Features, Imaging, and Surgical Management. Paraganglioma: A Multidisciplinary Approach, Exon Publications."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"E210088","DOI":"10.1148\/rycan.210088","article-title":"Head and Neck Paragangliomas: An Update on the Molecular Classification, State-of-the-Art Imaging, and Management Recommendations","volume":"4","author":"Lin","year":"2022","journal-title":"Radiol. Imaging Cancer"},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"T91","DOI":"10.1530\/ERC-15-0268","article-title":"15 YEARS OF PARAGANGLIOMA: Clinical manifestations of paraganglioma syndromes types 1\u20135","volume":"22","author":"Benn","year":"2015","journal-title":"Endocr. Relat. Cancer"},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"19","DOI":"10.6061\/clinics\/2012(Sup01)05","article-title":"Head and neck paragangliomas: Clinical and molecular genetic classification","volume":"67","author":"Offergeld","year":"2012","journal-title":"Clinics"},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"e00256","DOI":"10.1002\/edm2.256","article-title":"Re-evaluating the prevalence and factors characteristic of catecholamine secreting head and neck paragangliomas","volume":"4","author":"Smith","year":"2021","journal-title":"Endocrinol. Diabetes Metab."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"1761","DOI":"10.1002\/lary.23360","article-title":"Genetic and clinical characteristics of head and neck paragangliomas in a Chinese population","volume":"122","author":"Zheng","year":"2012","journal-title":"Laryngoscope"},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"213","DOI":"10.1530\/ERC-21-0359","article-title":"Head\/neck paragangliomas: Focus on tumor location, mutational status and plasma methoxytyramine","volume":"29","author":"Richter","year":"2022","journal-title":"Endocr. Relat. Cancer"},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"1915","DOI":"10.1210\/jc.2014-1498","article-title":"Pheochromocytoma and paraganglioma: An endocrine society clinical practice guideline","volume":"99","author":"Lenders","year":"2014","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"186","DOI":"10.2967\/jnumed.115.161018","article-title":"68Ga-DOTATATE PET\/CT in the Localization of Head and Neck Paragangliomas Compared with Other Functional Imaging Modalities and CT\/MRI","volume":"57","author":"Janssen","year":"2016","journal-title":"J. Nucl. Med."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"1266","DOI":"10.1007\/s00259-012-2119-7","article-title":"A comparison of the performance of 68Ga-DOTATATE PET\/CT and 123I-MIBG SPECT in the diagnosis and follow-up of phaeochromocytoma and paraganglioma","volume":"39","author":"Maurice","year":"2012","journal-title":"Eur. J. Nucl. Med. Mol. Imaging"},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"57","DOI":"10.4081\/rt.2010.e21","article-title":"Synchronous malignant vagal paraganglioma with contralateral carotid body paraganglioma treated by radiation therapy","volume":"2","author":"Kataria","year":"2010","journal-title":"Rare Tumors"},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"171","DOI":"10.1016\/j.anl.2007.05.007","article-title":"Synchronous bilateral carotid body tumor and vagal paraganglioma: A case report and review of literature","volume":"35","author":"Karatas","year":"2008","journal-title":"Auris Nasus Larynx"},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"93","DOI":"10.1002\/lio2.706","article-title":"Contemporary management of paragangliomas of the head and neck","volume":"7","author":"Cleere","year":"2021","journal-title":"Laryngoscope Investig. Otolaryngol."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"467","DOI":"10.1258\/0022215011908180","article-title":"Cervical paragangliomas: Diagnosis, management and complications","volume":"115","author":"Thabet","year":"2001","journal-title":"J. Laryngol. Otol."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"382","DOI":"10.1177\/019459989010200413","article-title":"Familial multiple cervical paragangliomas: Report of a kindred and review of the literature","volume":"102","author":"Sobol","year":"1990","journal-title":"Otolaryngol. Head Neck Surg."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"632","DOI":"10.1002\/hed.21790","article-title":"Head and neck paragangliomas: Report of 175 patients (1989\u20132010)","volume":"34","author":"Papaspyrou","year":"2012","journal-title":"Head. Neck"},{"key":"ref_18","doi-asserted-by":"crossref","unstructured":"Kawanabe, S., Katabami, T., Oshima, R., Yanagisawa, N., Sone, M., and Kimura, N. (2023). A rare case of multiple paragangliomas in the head and neck, retroperitoneum and duodenum: A case report and review of the literature. Front. Endocrinol., 13.","DOI":"10.3389\/fendo.2022.1054468"},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"113","DOI":"10.1046\/j.1365-2796.1999.00519.x","article-title":"Hereditary paraganglioma","volume":"246","author":"Lemaire","year":"1999","journal-title":"J. Intern. Med."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"471","DOI":"10.1007\/s001060170099","article-title":"Bilateral paraganglioma of the vagus nerve","volume":"49","author":"Velegrakis","year":"2001","journal-title":"HNO"},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"334","DOI":"10.1054\/ijom.2001.0196","article-title":"An unusual bilateral cervical paraganglioma: A case report","volume":"31","author":"Pandey","year":"2002","journal-title":"Int. J. Oral. Maxillofac. Surg."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"1","DOI":"10.15761\/OHNS.1000197","article-title":"Multiple paragangliomas involving carotid body and vagal region: A sporadic case","volume":"4","author":"Bakshi","year":"2019","journal-title":"Otorhinolaryngol. Head Neck Surg."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"rjab102","DOI":"10.1093\/jscr\/rjab102","article-title":"Thyroid paraganglioma in a patient with a history of carotid and vagal paraganglioma: Metastatic or primary tumor?","volume":"2021","author":"Yepuri","year":"2021","journal-title":"J. Surg. Case Rep."},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"1021","DOI":"10.1016\/j.radcr.2021.11.009","article-title":"Bilateral carotid body tumor case: A novel preoperative management","volume":"17","author":"Calzada","year":"2022","journal-title":"Radiol. Case Rep."},{"key":"ref_25","doi-asserted-by":"crossref","unstructured":"Duan, H.Y., Guan, Q., Guo, Y.J., and Liang, N. (2022). Case report: Cardiac arrest during carotid body tumor resection indicating carotid sinus hypersensitivity. Front. Cardiovasc. Med., 9.","DOI":"10.3389\/fcvm.2022.996644"},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"rjae011","DOI":"10.1093\/jscr\/rjae011","article-title":"Bilateral carotid body tumours: A case report from surgeon\u2019s perspective","volume":"2024","author":"Sakaran","year":"2024","journal-title":"J. Surg. Case Rep."},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"e175","DOI":"10.1055\/a-2654-2376","article-title":"Bilateral Carotid Body Paraganglioma: A Case Report and Review of Management Strategies","volume":"86","author":"Cidral","year":"2025","journal-title":"J. Neurol. Surg. Rep."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"1135","DOI":"10.1177\/0003489420931540","article-title":"The Changing Paradigm of Head and Neck Paragangliomas: What Every Otolaryngologist Needs to Know","volume":"129","author":"Cass","year":"2020","journal-title":"Ann. Otol. Rhinol. Laryngol."},{"key":"ref_29","doi-asserted-by":"crossref","unstructured":"Palade, D.O., Hainarosie, R., Zamfir, A., Vrinceanu, D., Pertea, M., Tusaliu, M., Mocanu, F., and Voiosu, C. (2024). Paragangliomas of the Head and Neck: A Review of the Latest Diagnostic and Treatment Methods. Medicina, 60.","DOI":"10.3390\/medicina60060914"},{"key":"ref_30","doi-asserted-by":"crossref","unstructured":"Vimawala, S.N., Graboyes, A.Z., Bennett, B., Bonanni, M., Abbasi, A., Oliphant, T., Alonso-Basanta, M., Rassekh, C., Cohen, D., and Brant, J.A. (2024). Head and Neck Paragangliomas: Overview of Institutional Experience. Cancers, 16.","DOI":"10.3390\/cancers16081523"},{"key":"ref_31","unstructured":"Else, T., Greenberg, S., and Fishbein, L. (2026, March 15). Hereditary Paraganglioma-Pheochromocytoma Syndromes. GeneReviews\u00ae, Available online: https:\/\/www.ncbi.nlm.nih.gov\/books\/NBK1548\/."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"943","DOI":"10.1001\/jama.292.8.943","article-title":"Distinct Clinical Features of Paraganglioma Syndromes Associated With SDHB and SDHD Gene Mutations","volume":"292","author":"Neumann","year":"2004","journal-title":"JAMA"},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"415","DOI":"10.1016\/j.beem.2010.04.001","article-title":"SDH-related Pheochromocytoma and paraganglioma","volume":"24","author":"Kantorovich","year":"2010","journal-title":"Best Pract. Res. Clin. Endocrinol. Metab."},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"386","DOI":"10.1002\/hed.27923","article-title":"Management of bilateral head and neck paragangliomas at a single-institution across four decades","volume":"47","author":"Bellamkonda","year":"2025","journal-title":"Head Neck"},{"key":"ref_35","doi-asserted-by":"crossref","first-page":"1491","DOI":"10.1002\/lary.27656","article-title":"Is routine genetic testing warranted in head and neck paragangliomas?","volume":"129","author":"Gupta","year":"2019","journal-title":"Laryngoscope"},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"2811","DOI":"10.1002\/1097-0142(20000615)88:12<2811::AID-CNCR21>3.0.CO;2-7","article-title":"Estimation of Growth Rate in Patients with Head and Neck Paragangliomas Influences the Treatment Proposal","volume":"88","author":"Jansen","year":"2000","journal-title":"Cancer"},{"key":"ref_37","doi-asserted-by":"crossref","first-page":"141","DOI":"10.1016\/j.eprac.2022.10.002","article-title":"Paraganglioma of the Head and Neck: A Review","volume":"29","author":"Sandow","year":"2023","journal-title":"Endocr. Pract."},{"key":"ref_38","doi-asserted-by":"crossref","unstructured":"Fang, F., Ding, L., He, Q., and Liu, M. (2020). Preoperative Management of Pheochromocytoma and Paraganglioma. Front. Endocrinol., 11.","DOI":"10.3389\/fendo.2020.586795"},{"key":"ref_39","doi-asserted-by":"crossref","first-page":"732","DOI":"10.1016\/0002-9610(71)90436-3","article-title":"Carotid body tumor (chemodectoma). Clinicopathologic analysis of ninety cases","volume":"122","author":"Shamblin","year":"1971","journal-title":"Am. J. Surg."},{"key":"ref_40","doi-asserted-by":"crossref","first-page":"101951","DOI":"10.1016\/j.beem.2024.101951","article-title":"Head and neck paragangliomas: Recent advances in translational and clinical research and guidelines for patient care","volume":"38","author":"Richter","year":"2024","journal-title":"Best Pract. Res. Clin. Endocrinol. Metab."},{"key":"ref_41","first-page":"31","article-title":"Multiple paragangliomas of the head and neck","volume":"30","author":"Magliulo","year":"2003","journal-title":"Otorrinolaringol. Ibero Am."},{"key":"ref_42","doi-asserted-by":"crossref","first-page":"440","DOI":"10.1111\/cen.13434","article-title":"Outcomes of patients with metastatic phaeochromocytoma and paraganglioma: A systematic review and meta-analysis","volume":"87","author":"Hamidi","year":"2017","journal-title":"Clin. Endocrinol."},{"key":"ref_43","first-page":"11","article-title":"Carotid and Vagal Body Paragangliomas","volume":"6","author":"Narese","year":"2013","journal-title":"Transl. Med. UniSa"},{"key":"ref_44","doi-asserted-by":"crossref","first-page":"187","DOI":"10.1016\/j.avsg.2018.10.019","article-title":"Surgical Management of Bilateral Carotid Body Tumors","volume":"57","author":"Luna","year":"2019","journal-title":"Ann. Vasc. Surg."},{"key":"ref_45","doi-asserted-by":"crossref","first-page":"123","DOI":"10.1097\/MOO.0000000000001035","article-title":"Bilateral carotid body tumor management: Tips, tricks, strategies, and problems","volume":"33","author":"Piazza","year":"2025","journal-title":"Curr. Opin. Otolaryngol. Head Neck Surg."},{"key":"ref_46","doi-asserted-by":"crossref","first-page":"171","DOI":"10.1055\/s-0031-1275251","article-title":"Outcomes after surgical resection of head and neck paragangliomas: A review of 61 patients","volume":"21","author":"Neskey","year":"2011","journal-title":"Skull Base"},{"key":"ref_47","doi-asserted-by":"crossref","first-page":"107997","DOI":"10.1016\/j.ijscr.2023.107997","article-title":"Heart rhythm irregularities during laparoscopy: Description of a novel technique to manage severe intra-operative bradycardia\u2014A case report","volume":"105","author":"Suhail","year":"2023","journal-title":"Int. J. Surg. Case Rep."},{"key":"ref_48","doi-asserted-by":"crossref","first-page":"459","DOI":"10.1001\/jamaoto.2014.144","article-title":"Implications of carotid sinus hypersensitivity following preoperative embolization of a carotid body tumor. An indication for prophylactic intraoperative cardiac pacing","volume":"140","author":"Bauer","year":"2014","journal-title":"JAMA Otolaryngol. Head. Neck Surg."},{"key":"ref_49","doi-asserted-by":"crossref","first-page":"665","DOI":"10.1016\/j.jvs.2024.11.008","article-title":"Preoperative radiological features in predicting complications of carotid body tumor resection","volume":"81","author":"Alanezi","year":"2025","journal-title":"J. Vasc. Surg."}],"container-title":["Journal of Clinical Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2077-0383\/15\/12\/4463\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,10]],"date-time":"2026-06-10T07:16:23Z","timestamp":1781075783000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2077-0383\/15\/12\/4463"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,6,9]]},"references-count":49,"journal-issue":{"issue":"12","published-online":{"date-parts":[[2026,6]]}},"alternative-id":["jcm15124463"],"URL":"https:\/\/doi.org\/10.3390\/jcm15124463","relation":{},"ISSN":["2077-0383"],"issn-type":[{"value":"2077-0383","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,6,9]]}}}