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Letter to the Editor
Isolated hypoglossal nerve palsy as an initial manifestation of a lung adenocarcinoma (occipital condyle syndrome)
Parálisis aislada del nervio hipogloso como manifestación inicial de un adenocarcinoma pulmonar (síndrome del cóndilo occipital)
Antonio Bustos-Merlo, Antonio Rosales-Castillo
Corresponding author
anrocas90@hotmail.com

Corresponding author.
, Jessica Ramírez Taboada
Servicio de Medicina Interna, Hospital Universitario Virgen de las Nieves, Granada, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Occipital condyle syndrome&#44; described in 1981&#44; is a rare entity characterised by occipital headache and hypoglossal nerve involvement&#44; usually caused by local erosive lesions of metastatic aetiology&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 55-year-old male who started with headache and left hypoglossal nerve palsy and was finally diagnosed with metastatic lung adenocarcinoma&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">A 55-year-old man with a personal history of type 2 diabetes mellitus with good metabolic control&#44; hypercholesterolemia and smoking &#40;64 packs&#47;year&#41;&#46; The patient came to the emergency department for severe sudden onset&#44; left hemicranial headache&#44; accompanied by nausea and photophobia&#44; refractory to oral analgesia&#46; It associated ipsilateral cervical pain with exacerbation when the head was tilted to the right&#44; intense asthenia and weight loss of 4&#8239;kg over a month&#46; He did not report any other symptoms in the history-taking&#46; On physical examination&#44; the vital signs were&#58; blood pressure &#40;BP&#41; 147&#47;73&#8239;mmHg with heart rate 77&#8239;lpm&#46; Oxygen saturation &#40;SpO2&#41; 95&#37; room air and temperature 36&#46;5 &#8239;&#176;C&#46; Tongue deviation to the left&#44; together with slight atrophy of the ipsilateral hemitongue &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#44; with no other neurological abnormalities&#46; No pathological lymphadenopathies were palpable and auscultation and abdominal examination were normal&#46; Laboratory tests showed hyperglycaemia &#40;200&#8239;mg&#47;dL&#41;&#44; increased lactate dehydrogenase &#40;255&#8239;U&#47;L&#59; NV 10-248&#41; and lymphopenia &#40;white blood cells 7230 with 430&#8239;lymphocytes&#47;&#956; L&#59; NV 1000&#8211;4000&#41;&#46; Renal function&#44; liver profile and coagulation showed no abnormalities&#46; A cranial computed tomography &#40;CT&#41; scan was performed&#44; which identified a 32&#46;5&#8239;mm diameter lesion of irregular margins and soft tissue density with osteolysis of the external margin of the left occipital condyle and foramen magnum &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; Given the resistance of the pain despite opioid treatment&#44; he was finally admitted to the hospital ward for symptomatic control and aetiological study&#46; Tumour markers showed slight elevation of carcinoembryonic antigen &#40;7&#46;3&#8239;ng&#47;mL&#41; with values in the range of Cipher 21&#46;1 and PSA&#46; Interferon gamma release assay &#40;IGRA&#41; was negative&#46; Brain magnetic resonance imaging &#40;MRI&#41; showed a soft tissue mass &#40;35&#8239;&#215;&#8239;27&#8239;mm&#41; in the left occipital condyle of the clivus&#44; causing bone destruction&#44; with intermediate&#47;slightly low signal on T1-weighted sequence&#44; intermediate-slightly high signal on T2-weighted sequence&#44; with weak diffusion restriction and moderate enhancement after contrast&#46; In order to search for the primary neoplasm and a suitable biopsy site&#44; a positron emission tomography &#40;PET&#47;CT&#41; was performed&#44; which revealed an increased metabolic uptake of pathological intensity in the lesion described &#40;SUVmax&#8239;&#61;&#8239;7&#44;27&#41;&#44; together with extensive bone involvement and a hypermetabolic focus in the left pulmonary hilum &#40;SUVmax&#8239;&#61;&#8239;4&#44;12&#41; as a possible primary bronchial lesion&#46; A CT-guided core needle biopsy of the bone lesion in the right iliac crest was performed&#44; and anatomical pathology revealed the presence lung adenocarcinoma with a PDL1&#8239;&#60;&#8239;1&#37; and non-mutated epidermal growth factor receptor &#40;EGFR&#41;&#46; The final diagnosis was occipital condyle syndrome secondary to metastasis of lung adenocarcinoma&#46; After being presented to the tumour committee&#44; chemotherapy with pembrolizumab&#44; carboplatin and pemetrexed was started&#44; together with analgesic radiotherapy of the osteolytic occipital condyle lesion &#40;20&#8239;Gy in 5 consecutive 4&#8239;Gy&#47;fraction&#41;&#44; with neurological symptom improvement&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Occipital condyle syndrome was first described by Greenberg in 1981&#46; It consists of a unilateral occipital headache that is typically exacerbated by rotation to the opposite side and may be associated with scalp hypersensitivity<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> together with involvement of the hypoglossal nerve in the form of paresis or palsy and may be associated with dysarthria and dysphagia&#46; Most cases are secondary to metastatic lesions of a known neoplasm or as an initial manifestation of the same&#44; so early detection and identification are essential&#44;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> with the prostate being the most common origin&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Diagnosis is usually made by cranial MRI&#44; with the characteristic finding of hypointense soft tissue in T1-weighted sequence in the area of the affected condyle&#44; replacing the local fat&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The prognosis is usually quite poor due to the often-advanced stage of the underlying neoplasm&#44; with local radiotherapy being one of the mainstays for symptomatic control&#44;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> together with targeted antineoplastic therapy&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical considerations</span><p id="par0025" class="elsevierStylePara elsevierViewall">Informed consent was obtained for imaging and subsequent publication&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0030" class="elsevierStylePara elsevierViewall">Unfunded&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interest</span><p id="par0035" class="elsevierStylePara elsevierViewall">No conflict of interest&#46;</p></span></span>"
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Original language: English
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