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ANL-1990; No. of Pages 3 Auris Nasus Larynx xxx (2015) xxx–xxx

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Auris Nasus Larynx journal homepage: www.elsevier.com/locate/anl

An uncommon case of dyspnea with unilateral laryngeal paralysis in acromegaly Justine Lerat a,*, Marie Lacoste b, Jean-Marc Prechoux a, Karine Aubry a, Sylvie Nadalon b, Kim Heang Ly b, Jean-Pierre Bessede a a b

Otorhinolaryngology, Head and Neck Surgery, Dupuytren Teaching Hospital, 2 Avenue Martin Luther King, 87100 Limoges cedex, France Internal Medicine, Dupuytren Teaching Hospital, Limoges, France

A R T I C L E I N F O

A B S T R A C T

Article history: Received 10 April 2015 Accepted 8 June 2015 Available online xxx

A 61-year-old man with obstructive sleep apnea syndrome and normal BMI complained of dyspnea. Nasofibroscopy revealed a global and major oedema of the glottis and supraglottis and also a paralysis of the left vocal fold. CT-scan pointed out a spontaneous hyperdensity of the left arytenoid cartilage. A tracheostomy was performed. Clinical examination revealed large hands and macroglossy with high IGF1 rate. MRI confirmed a supracentimetric pituitary adenoma. To our knowledge, this is the first description of a case of acute respiratory distress due to unilateral larynx paralysis leading to acromegaly diagnosis. This is due to submucosal hypertrophy and vocal cord immobility. ß 2015 Elsevier Ireland Ltd. All rights reserved.

Keywords: Vocal cord paralysis Dyspnea Acromegaly Obstructive sleep apnea syndrome

1. Introduction Patients who suffer from acromegaly can develop obstructive sleep apnea syndrome or upper airway obstruction. Dyspnea is exceptional. We describe a rare complication of acromegaly, in the light of a case of obstructive sleep apnea, complicated with acute respiratory distress due to laryngeal paralysis. 2. Case report A 61-year-old man came to the A and E unit for acute respiratory distress. Dyspnea had started a fortnight earlier and had worsened despite steroid treatment (prednisolone 1 mg/kg/d). He had a past medical history of obstructive sleep apnea syndrome with normal BMI and bilateral hip replacement. The patient had self-discontinued the use of positive airway pressure therapy. He had quitted smoking thirty-five years earlier. On clinical examination, the patient suffered from dyspnea and dysphonia. No enlarged lymph node was felt but the thyroid cartilage was tender on palpation. Moderate macroglossy was observed. Nasofibroscopy revealed a global major oedema of the glottis and supraglottis and also an immobility of the left vocal fold in the paramedian position, which led to a significant

* Corresponding author. Tel.: +33 05 55 05 66 59; fax: +33 05 55 05 62 87. E-mail address: [email protected] (J. Lerat).

reduction of the upper respiratory tract. The rest of the examination, mainly neurological and rheumatological and of the oropharynx was normal. No fever was detected. Blood samples did not reveal any abnormality, especially any infectious or inflammatory syndrome. CT-scan pointed out a spontaneous hyperdensity of the left arytenoid cartilage and glottic stenosis due to swelling without any associated thyroid cartilage abnormality (Fig. 1). Direct laryngoscopy and panendoscopy were performed under general anaesthesia and revealed oedema of the whole glottis and supra glottis without any inflammatory sign. No respiratory space remained. Petechiae were observed all along the trachea with a 08 laryngeal optic. A tracheostomy was performed under general anaesthesia after introduction of a Cook airway exchange catheter with difficulties. Biopsies of the supraglottis found parakeratosis without any amyloid deposit, sarcoidosis sign or malignancy (Fig. 2 and Video 1). Supplementary material related to this article can be found, in the online version, at http://dx.doi.org/10.1016/j.anl.2015.06.001. Proteins electrophoresis found moderate hypogammaglobulinemia and no antibody was discovered. Both bacterial and viral samples, as well as serologies, were negative. The medical treatment combined steroids (1 mg/kg/d) and antibiotics (amoxicilline and clavulanic acid) for 7 days. On nasofibroscopy, a decrease of the oedema was observed. Three weeks later a left posterior laser CO2 cordotomy was performed and the inferior chondrocalcification of the left

http://dx.doi.org/10.1016/j.anl.2015.06.001 0385-8146/ß 2015 Elsevier Ireland Ltd. All rights reserved.

Please cite this article in press as: Lerat J, et al. An uncommon case of dyspnea with unilateral laryngeal paralysis in acromegaly. Auris Nasus Larynx (2015), http://dx.doi.org/10.1016/j.anl.2015.06.001

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J. Lerat et al. / Auris Nasus Larynx xxx (2015) xxx–xxx

Fig. 1. Cervical CT-scan with axial and frontal slides: calcification of the left arytenoid cartilage and glottic swelling.

Fig. 2. Direct laryngoscopy under general anaesthesia: oedema of the whole glottis and supra glottis without inflammatory signs and significant reduction of the upper respiratory tract.

arytenoid cartilage was removed. The anatomopathological results were parakeratosis with chronic and congestive inflammatory modifications. Stage I oesophagitis was described on oesophageal fibroscopy. Control CT-scan revealed a circumferential oedema of the glottis and left vocal fold. Decannulation was not possible and laryngeal oedema was still present on fibroscopy. Thyrotomy and left arytenoidectomy were performed by open surgery. Anatomopathological analysis confirmed ossification of the left arytenoid cartilage.

Tracheostomy was required for a total of two months and removed successfully. Clinical examination finally revealed large hands and the size of the patient’s shoes had increased two sizes in 20 years. IGF1 rates rose up to 888.6 ng/ml (normal

An uncommon case of dyspnea with unilateral laryngeal paralysis in acromegaly.

A 61-year-old man with obstructive sleep apnea syndrome and normal BMI complained of dyspnea. Nasofibroscopy revealed a global and major oedema of the...
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