Referred Orofacial Pain as an Initial Symptom of Distant, Nonmetastatic Cancer: Report of a Case and Review of the Literature

Published:August 12, 2021DOI:


      Referred orofacial pain as the first symptom of an otherwise silent distant, nonmetastatic cancer has been reported, but there is sparse literature on the subject. Referred pain may not be considered in the orofacial pain differential diagnosis because of its rarity; however, this may delay a cancer diagnosis. The authors present a case report and a review of the English literature. Peer-reviewed publications were identified through a systematic search of MEDLINE, Embase, and Cochrane CENTRAL. Historic cases were reviewed, and available data regarding demographics, pain characteristics, treatment, cancer diagnosis, and outcome were extracted. Thirty-seven cases were identified. All cancers were intrathoracic. The average age was 54.1 years with a slight female predominance (3:2). Common pain characteristics were (1) diffuse location affecting the ear (76%), jaw (46%), and temple (30%); (2) constant duration (65%); (3) aching quality (74%); (4) severe intensity (94%); and (5) associated systemic symptoms (68%) such as weight loss and digital clubbing. The average time from the onset of facial pain to seeking medical attention was 9 months, and the average time from seeking medication attention to cancer diagnosis was 8 months. Orofacial pain was often attributed to odontogenic (35%) or neuropathic (25%) causes, and treatments for these conditions were also common. The impact of referred orofacial pain on the cancer prognosis was not possible because of the nature of the reviewed studies (case reports with no comparison group). Cancer-associated referred orofacial pain as a first symptom is rare but should be considered in cases with intractable pain and associated systemic symptoms.

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        • Macfarlane T.V.
        • Beasley M.
        • Macfarlane G.J.
        Self-reported facial pain in UK biobank study: prevalence and associated factors.
        J Oral Maxillofac Res. 2014; 5: e2
        • Ghurye S.
        • McMillan R.
        Orofacial pain - an update on diagnosis and management.
        Br Dent J. 2017; 223: 639-647
        • Okeson J.P.
        Nonodontogenic toothache.
        Tex Dent J. 2000; 117: 64-74
        • Renton T.
        Tooth-related pain or not?.
        Headache. 2020; 60: 235-246
        • Romero-Reyes M.
        • Salvemini D.
        Cancer and orofacial pain.
        Med Oral Patol Oral Cir Bucal. 2016; 21: e665-e671
        • Kikuta S.
        • Dalip D.
        • Loukas M.
        • et al.
        Jaw pain and myocardial ischemia: a review of potential neuroanatomical pathways.
        Clin Anat. 2019; 32: 476-479
        • Palmieri A.
        Lung cancer presenting with unilateral facial pain: remission after laryngeal nerve palsy.
        Headache. 2006; 46: 813-815
        • Eross E.
        • Dodick D.
        • Swanson J.
        • Capobianco D.
        A review of intractable facial pain secondary to underlying lung neoplasms.
        Cephalalgia. 2003; 23: 2-5
        • Sarlani E.
        • Schwartz A.
        • Greenspan J.
        • Grace E.
        Facial pain as first manifestation of lung cancer: a case of lung cancer-related cluster headache and a review of the literature.
        J Orofac Pain. 2003; 17: 262-267
        • Abraham P.
        • Capobianco D.
        • Cheshire W.
        Facial pain as the presenting symptom of lung carcinoma with normal chest radiograph..
        Headache. 2003; 43: 499-504
        • Schoenen J.
        • Broux R.
        • Moonen G.
        Unilateral facial pain as the first symptom of lung cancer: are there diagnostic clues?.
        Cephalalgia. 1992; 12: 178-179
        • Pembroke C.A.
        • Byrne A.
        • Lester J.F.
        • Button M.
        Persistent unilateral facial pain in lung cancer patients with mediastinal nodal involvement.
        Lung Cancer. 2013; 82: 173-175
        • Bindoff L.A.
        • Heseltine D.
        Unilateral facial pain in patients with lung cancer: a referred pain via the vagus?.
        Lancet. 1988; 1: 812-815
        • Paparella M.M.
        in: Paparella M.M. Shumrick D.A. Otolaryngology. 2nd ed. WB Saunders, Philadelphia1980: 1354-1357
        • Capobianco D.J.
        Facial pain as a symptom of nonmetastatic lung cancer.
        Headache. 1995; 35: 581-585
        • Des Prez R.
        • Freemon F.
        Facial pain associated with lung cancer: a case report.
        Headache. 2005; 23: 43-44
        • Nestor J.J.
        Unilateral facial pain in lung cancer.
        Lancet. 1991; 338: 1149
        • Bongers K.
        • Willigers H.
        • Koehler P.
        Referred facial pain from lung carcinoma.
        Neurology. 1992; 42: 1840
        • Nestor J.J.
        • Ngo L.K.
        Incidence of facial pain caused by lung cancer.
        Otolaryngol Head Neck Surg. 1994; 111: 155-156
        • Shakespeare T.
        • Stevens M.
        Unilateral facial pain and lung cancer.
        Australas Radiol. 1996; 40: 45-46
        • Goldberg H.L.
        Chest cancer refers pain to face and jaw: a case review.
        Cranio. 1997; 15: 167-169
        • Jones M.T.
        • Lawson R.A.
        Unilateral facial pain as a rare presentation of bronchial carcinoma.
        Br J Clin Pract. 1987; 41: 1025-1026
        • Epstein J.B.
        • Miaskowski C.
        Oral pain in the cancer patient.
        J Natl Cancer Inst Monogr. 2019; 2019: lgz003
        • Klieb H.B.
        • Avon S.L.
        • Gilbert J.
        • et al.
        Florid cutaneous and mucosal papillomatosis: mucocutaneous markers of an underlying gastric malignancy.
        J Clin Oncol. 2013; 31: e218-e219
        • Klieb H.B.
        • Freeman B.V.
        Trigeminal neuralgia caused by intracranial epidermoid tumour: report of a case.
        J Can Dent Assoc. 2008; 74: 63-65
        • Romero-Reyes M.
        • Teruel A.
        • Ye Y.
        Cancer and referred facial pain.
        Curr Pain Headache Rep. 2015; 19: 37
        • Portenoy R.
        Treatment of cancer pain.
        Lancet. 2011; 377: 2236-2247
        • Khan M.
        • Nishi S.E.
        • Hassan S.N.
        • et al.
        Trigeminal neuralgia, glossopharyngeal neuralgia, and myofascial pain dysfunction syndrome: an update.
        Pain Res Manag. 2017; 2017: 7438326
        • Weiss A.L.
        • Ehrhardt K.P.
        • Tolba R.
        Atypical facial pain: a comprehensive, evidence-based review.
        Curr Pain Headache Rep. 2017; 21: 8
        • Dabuleanu M.
        Pulpitis (reversible/irreversible).
        J Can Dent Assoc. 2013; 79: d90
        • Fukuda K.I.
        Diagnosis and treatment of abnormal dental pain.
        J Dent Anesth Pain Med. 2016; 16: 1-8