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Case Reports
. 2017 Feb;37(1):76-79.
doi: 10.14639/0392-100X-1141.

Idiopathic SIADH in young patients: don't forget the nose

C Parrilla  1 , D Lucidi  1 , G Petrone  2 , G Bastanza  1 , C Colosimo  3
Affiliations
Case Reports

Idiopathic SIADH in young patients: don't forget the nose

C Parrilla et al. Acta Otorhinolaryngol Ital. 2017 Feb.

Abstract
in English, Italian

Olfactory neuroblastoma (ONB) is an uncommon neuroendocrine sinonasal cancer associated by many authors to ectopic production of several biologically active substances. We report a case of a 31-year-old male patient who presented with idiopathic syndrome of inappropriate secretion of antidiuretic hormone (SIADH). During diagnostic work-up, a CT scan of the head was performed and an ethmoidal ONB was detected. Endoscopical surgery followed by radiotherapy was carried out. Immediately after surgery natraemia levels normalised. Five years later the patient is disease-free. To our knowledge, 17 cases of SIADH associated to ONB have been published. In nine reports, idiopathic SIADH promptly led to the diagnosis of the sinonasal mass as in our clinical case, however, in many reports, correct diagnosis was accomplished months to years later. In young patients with idiopathic inappropriate antidiuretic hormone secretion, a neuroendocrine malignancy of the sinonasal area must be excluded.

Il neuroblastoma olfattivo (ONB) è un raro tumore neuroendocrino dei seni paranasali associato, secondo molti autori, alla produzione di molteplici sostanze biologicamente attive. In questo lavoro descriviamo il caso di un paziente di sesso maschile di 31 anni giunto alla nostra osservazione presentando la sindrome idiopatica da inappropriata secrezione di ormone antidiuretico (SIADH). Durante il work-up diagnostico il paziente è stato sottoposto a TAC del massiccio facciale, che documentava la presenza di un estesioneuroblastoma etmoidale. è stato eseguito un trattamento di chirurgia endoscopica e successiva radioterapia. Subito dopo l’intervento chirurgico i livelli di natremia si sono normalizzati. Cinque anni più tardi il paziente risulta essere libero da malattia. Attualmente in letteratura sono stati pubblicati 17 casi di SIADH associata a ONB. In nove lavori la SIADH idiopatica ha tempestivamente portato alla diagnosi di masse sinusali come nel nostro caso, tuttavia, in molti casi, si è giunti alla diagnosi corretta mesi o anni dopo. Nei giovani pazienti con sindrome idiopatica da inappropriata secrezione di ormone antidiuretico è necessario escludere un tumore maligno neuroendocrino dei seni paranasali.

Keywords: Arginine vasopressin; Olfactory neuroblastoma; Paraneoplastic syndrome; SIADH; Sinonasal.

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Figures

Fig. 1.
Fig. 1.
A. The tumour shows a nodular grow pattern composed by monomorphic round to oval neoplastic cells. (Haematoxylin and eosin, original magnification ×100). B. Tumour cells with finely granular nuclei and scant cytoplasm with neurofibrillary matrix forming Homer-Wright rosettes. (Haematoxylin and eosin, original magnification ×400). C-D-E. Tumour cells exhibiting diffuse cytoplasmatic immunoreactivity for chromogranin A (C), synaptophysin (D) and NSE (E). (Haematoxylin counterstaining, original magnification ×200).
Fig. 2.
Fig. 2.
A-B. Coronal and sagittal T2-weighted images. Pre-operative MRI shows a large, well-defined mass expanding the right ethmoid. The mass lesion exhibits intermediate signal on T2-weighted images, due to its high cellularity; there is no evidence of infiltration of the orbital fat and of the fovea ethmoidalis. Inflammatory/obstructive changes are seen in both right frontal and maxillary sinuses due to mass effect on the ostio-meatal unit. Right lamina papyracea was compressed by the mass, but not interrupted; the orbit, anterior cranial fossa and brain were disease-free. C. Endoscopic view of the right nasal fossa: a fairly well circumscribed, multi-lobulated smooth rounded mass, involving the right ethmoid sinus and the region of middle turbinate is showed. Bony destruction was not recognisable, but the mass effect was prominent. D. Post-operative coronal CT image demonstrates the ethmoido-maxillectomy, without evidence of residual/recurrent disease.
See this image and copyright information in PMC

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