G Model ANORL-423; No. of Pages 2
ARTICLE IN PRESS European Annals of Otorhinolaryngology, Head and Neck diseases xxx (2015) xxx–xxx
Available online at
ScienceDirect www.sciencedirect.com
Letter to the editor Small cell neuroendocrine carcinoma of the ethmoid sinus revealed by syndrome of inappropriate antidiuretic hormone secretion
1. Case report A 64-year-old man attended the emergency department with delirium and headache. He had a history of former smoking (50 pack-years), hypercholesterolaemia, treated hypertension and treatment with Kardegic (aspirin) 160. Clinical interview revealed a one-month history of repeated episodes of epistaxis. A laboratory work-up demonstrated hyponatremia (119 mmol/L) and normal serum potassium and renal function. Brain CT-scan did not reveal any abnormality, but a left nasal sinus opacity with a neoplastic appearance, the extent of which was subsequently assessed with an MRI of the face (Fig. 1). A left hilar lymph node measuring 2 cm was visualized on chest CT-scan. A syndrome of inappropriate antidiuretic hormone secretion (SIADH) was suspected and water restriction was instituted, allowing resolution of the delirium. Examination of the nasal cavities demonstrated a solid mass in contact with the middle turbinate. Histological examination of biopsies of this mass revealed neuroendocrine carcinoma. The tumour expressed chromogranin A, synaptophysin and CD56 markers. Transbronchial aspiration cytology confirmed the presence of a hilar lymph node metastasis presenting the same immunocytochemical profile as the primary tumour. 18 F-FDG PET-CT confirmed the presence of the sphenoethmoidal lesion (SUVmax = 9), but with no uptake by the hilar lymph node (Fig. 2). The diagnosis of stage T4N0M1 small cell neuroendocrine carcinoma (SNEC) of the ethmoid sinus associated with paraneoplastic SIADH was proposed. Platinum and etoposide combination chemotherapy was initiated with a complete response of the sinus tumour and normal serum sodium after 4 cycles. Radiotherapy to the sinus combined with weekly carboplatin was performed. No recurrence was observed more than 15 months after stopping treatment and the hilar lymph node lesion remained stable. The tumour most commonly responsible for SIADH is small cell lung cancer, present in 10% of patients [1]. Extrapulmonary small cell neuroendocrine tumours are rare. The most common sites are salivary glands, sinuses and larynx [2,3]. In France, 21 cases of SNEC of the nasal cavities and sinuses were reported between 1989 and 2003 in 8 university hospital centres [4]. Cases of SNEC associated with SIADH are exceptional, as only 70 cases have been reported in the international literature, with only 3 cases involving the nasal cavities [1]. SIADH can be associated with other types of head and neck tumours: squamous cell carcinoma (43 patients with SIADH in a series of 1436 patients, i.e. 3%) [5], olfactory neuroblastoma, adenoid cystic carcinoma, undifferentiated carcinoma and sarcoma.
Fig. 1. a and b: MRI of the face, coronal (a) and axial (b) T2-weighted sequences showing numerous dental artifacts, a left ethmoidal tumour extending into the nasal cavity, maxillary sinus, sphenoidal sinus, pterygopalatine fossa and the ipsilateral internal temporal region.
As neuroendocrine tumours are responsible for non-specific nasal symptoms (epistaxis, blocked nose, hyposmia), they are often diagnosed at an advanced stage of the disease. The treatment of SIADH is based on that of the small cell cancer, particularly chemotherapy. Monitoring of serum sodium is a good marker of the course of the tumour.
http://dx.doi.org/10.1016/j.anorl.2015.05.006 1879-7296/© 2015 Elsevier Masson SAS. All rights reserved.
Please cite this article in press as: Bach C-A, et al. Small cell neuroendocrine carcinoma of the ethmoid sinus revealed by syndrome of inappropriate antidiuretic hormone secretion. European Annals of Otorhinolaryngology, Head and Neck diseases (2015), http://dx.doi.org/10.1016/j.anorl.2015.05.006
G Model ANORL-423; No. of Pages 2
ARTICLE IN PRESS Letter to the editor / European Annals of Otorhinolaryngology, Head and Neck diseases xxx (2015) xxx–xxx
2
Fig. 2.
18
F-FDG PET-CT, fusion images of axial, coronal and sagittal scans: hypermetabolic (SUVmax = 9) left spheno-ethmoidal lesion.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article. References [1] Ma AT, Lei KI. Small cell neuroendocrine carcinoma of the ethmoid sinuses presenting with generalized seizure and syndrome of inappropriate antidiuretic hormone secretion: a case report and review of literature. Am J Otolaryngol 2009;30(1):54–7. [2] Meacham R, Matrka L, Ozer E, Ozer HG, Wakely P, Shah M. Neuroendocrine carcinoma of the head and neck: a 20-year case series. Ear Nose Throat J 2012;91(3):20–4. [3] Renner G. Small cell carcinoma of the head and neck: a review. Semin Oncol 2007;34(1):3–14. [4] Babin E, Rouleau V, Vedrine PO, Toussaint B, de Raucourt D, Malard O, et al. Small cell neuroendocrine carcinoma of the nasal cavity and paranasal sinuses. J Laryngol Otol 2006;120(4):289–97. [5] Talmi YP, Hoffman HT, McCabe BF. Syndrome of inappropriate secretion of arginine vasopressine in patients with cancer of the head and neck. Ann Otol Rhinol Laryngol 1992;101(11):946–9.
C.-A. Bach a,b,∗ L. Guilleré a,b E. Le Stanc c F. Chabolle a,b a Service de chirurgie ORL et cervico-faciale, hôpital Foch, 40, rue Worth, 92150 Suresnes cedex, France b UFR de médecine Paris Ouest Saint-Quentin-en-Yvelines, université de Versailles Saint-Quentin-en-Yvelines, 78280 Guyancourt, France c Service de médecine nucléaire, hôpital Foch, 40, rue Worth, 92150 Suresnes, France ∗ Corresponding author. E-mail address: [email protected] (C.-A. Bach)
Please cite this article in press as: Bach C-A, et al. Small cell neuroendocrine carcinoma of the ethmoid sinus revealed by syndrome of inappropriate antidiuretic hormone secretion. European Annals of Otorhinolaryngology, Head and Neck diseases (2015), http://dx.doi.org/10.1016/j.anorl.2015.05.006
ARTICLE IN PRESS European Annals of Otorhinolaryngology, Head and Neck diseases xxx (2015) xxx–xxx
Available online at
ScienceDirect www.sciencedirect.com
Letter to the editor Small cell neuroendocrine carcinoma of the ethmoid sinus revealed by syndrome of inappropriate antidiuretic hormone secretion
1. Case report A 64-year-old man attended the emergency department with delirium and headache. He had a history of former smoking (50 pack-years), hypercholesterolaemia, treated hypertension and treatment with Kardegic (aspirin) 160. Clinical interview revealed a one-month history of repeated episodes of epistaxis. A laboratory work-up demonstrated hyponatremia (119 mmol/L) and normal serum potassium and renal function. Brain CT-scan did not reveal any abnormality, but a left nasal sinus opacity with a neoplastic appearance, the extent of which was subsequently assessed with an MRI of the face (Fig. 1). A left hilar lymph node measuring 2 cm was visualized on chest CT-scan. A syndrome of inappropriate antidiuretic hormone secretion (SIADH) was suspected and water restriction was instituted, allowing resolution of the delirium. Examination of the nasal cavities demonstrated a solid mass in contact with the middle turbinate. Histological examination of biopsies of this mass revealed neuroendocrine carcinoma. The tumour expressed chromogranin A, synaptophysin and CD56 markers. Transbronchial aspiration cytology confirmed the presence of a hilar lymph node metastasis presenting the same immunocytochemical profile as the primary tumour. 18 F-FDG PET-CT confirmed the presence of the sphenoethmoidal lesion (SUVmax = 9), but with no uptake by the hilar lymph node (Fig. 2). The diagnosis of stage T4N0M1 small cell neuroendocrine carcinoma (SNEC) of the ethmoid sinus associated with paraneoplastic SIADH was proposed. Platinum and etoposide combination chemotherapy was initiated with a complete response of the sinus tumour and normal serum sodium after 4 cycles. Radiotherapy to the sinus combined with weekly carboplatin was performed. No recurrence was observed more than 15 months after stopping treatment and the hilar lymph node lesion remained stable. The tumour most commonly responsible for SIADH is small cell lung cancer, present in 10% of patients [1]. Extrapulmonary small cell neuroendocrine tumours are rare. The most common sites are salivary glands, sinuses and larynx [2,3]. In France, 21 cases of SNEC of the nasal cavities and sinuses were reported between 1989 and 2003 in 8 university hospital centres [4]. Cases of SNEC associated with SIADH are exceptional, as only 70 cases have been reported in the international literature, with only 3 cases involving the nasal cavities [1]. SIADH can be associated with other types of head and neck tumours: squamous cell carcinoma (43 patients with SIADH in a series of 1436 patients, i.e. 3%) [5], olfactory neuroblastoma, adenoid cystic carcinoma, undifferentiated carcinoma and sarcoma.
Fig. 1. a and b: MRI of the face, coronal (a) and axial (b) T2-weighted sequences showing numerous dental artifacts, a left ethmoidal tumour extending into the nasal cavity, maxillary sinus, sphenoidal sinus, pterygopalatine fossa and the ipsilateral internal temporal region.
As neuroendocrine tumours are responsible for non-specific nasal symptoms (epistaxis, blocked nose, hyposmia), they are often diagnosed at an advanced stage of the disease. The treatment of SIADH is based on that of the small cell cancer, particularly chemotherapy. Monitoring of serum sodium is a good marker of the course of the tumour.
http://dx.doi.org/10.1016/j.anorl.2015.05.006 1879-7296/© 2015 Elsevier Masson SAS. All rights reserved.
Please cite this article in press as: Bach C-A, et al. Small cell neuroendocrine carcinoma of the ethmoid sinus revealed by syndrome of inappropriate antidiuretic hormone secretion. European Annals of Otorhinolaryngology, Head and Neck diseases (2015), http://dx.doi.org/10.1016/j.anorl.2015.05.006
G Model ANORL-423; No. of Pages 2
ARTICLE IN PRESS Letter to the editor / European Annals of Otorhinolaryngology, Head and Neck diseases xxx (2015) xxx–xxx
2
Fig. 2.
18
F-FDG PET-CT, fusion images of axial, coronal and sagittal scans: hypermetabolic (SUVmax = 9) left spheno-ethmoidal lesion.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article. References [1] Ma AT, Lei KI. Small cell neuroendocrine carcinoma of the ethmoid sinuses presenting with generalized seizure and syndrome of inappropriate antidiuretic hormone secretion: a case report and review of literature. Am J Otolaryngol 2009;30(1):54–7. [2] Meacham R, Matrka L, Ozer E, Ozer HG, Wakely P, Shah M. Neuroendocrine carcinoma of the head and neck: a 20-year case series. Ear Nose Throat J 2012;91(3):20–4. [3] Renner G. Small cell carcinoma of the head and neck: a review. Semin Oncol 2007;34(1):3–14. [4] Babin E, Rouleau V, Vedrine PO, Toussaint B, de Raucourt D, Malard O, et al. Small cell neuroendocrine carcinoma of the nasal cavity and paranasal sinuses. J Laryngol Otol 2006;120(4):289–97. [5] Talmi YP, Hoffman HT, McCabe BF. Syndrome of inappropriate secretion of arginine vasopressine in patients with cancer of the head and neck. Ann Otol Rhinol Laryngol 1992;101(11):946–9.
C.-A. Bach a,b,∗ L. Guilleré a,b E. Le Stanc c F. Chabolle a,b a Service de chirurgie ORL et cervico-faciale, hôpital Foch, 40, rue Worth, 92150 Suresnes cedex, France b UFR de médecine Paris Ouest Saint-Quentin-en-Yvelines, université de Versailles Saint-Quentin-en-Yvelines, 78280 Guyancourt, France c Service de médecine nucléaire, hôpital Foch, 40, rue Worth, 92150 Suresnes, France ∗ Corresponding author. E-mail address: [email protected] (C.-A. Bach)
Please cite this article in press as: Bach C-A, et al. Small cell neuroendocrine carcinoma of the ethmoid sinus revealed by syndrome of inappropriate antidiuretic hormone secretion. European Annals of Otorhinolaryngology, Head and Neck diseases (2015), http://dx.doi.org/10.1016/j.anorl.2015.05.006
