CASE PRESENTATION Unusual dyspnoea

M. Ibro A 22-year-old female student presented at the inhaled and systemic corticosteroids. The treat- I. Peposhi Diagnostic Center “IKEDA” (Tirana, Albania) ment was not effective. The patient did not smoke. J. Bulaj with a 1-year history of progressive dyspnoea. She There was no palpable mass in the , and had significant stridor, but no hoarseness. Her radiography of the chest was normal. A flexible medical history did not reveal any significant dis- fibreoptic examination was performed and this ease, except the fact that she had been treated for suggested expansion of the left aryepiglottic fold the whole of the previous year for bronchial asth- in a saccular shape. ma. The treatment had included 2-agonists and Diagnostic Center “IKEDA”, Tirana, Albania. ab

Correspondence: M. Ibro Rruga “Kongresi” I Manastirit 19/3 00355 Tirana Albania Fax: 355 4365817 E-mail: [email protected]

cd Task 1 View the images presented and suggest a possible diagnosis.

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Breathe | June 2005 | Volume 1 | No 4 349 CASE PRESENTATION Unusual dyspnoea

of the laryngeal saccule, or from retention of Answer 1 mucus in the collecting ducts of submucosal The diagnosis of saccular cyst of glands located around the ventricle. The lesions was considered. are classified as anterior or lateral according to their location. The airway compromise caused by these Subsequently, a 2-mm computed tomogra- lesions may be severe, and in one series reported phy (CT) scan was obtained. The CT scan [1], ~50% of the cases were diagnosed at biopsy. confirmed the presence of a large cyst arising from The importance of early recognition should be the laryngeal ventricle with minimal extension emphasised, and diagnosis has been facilitated through the thyrohyoid membrane, but with a sig- by advances in flexible fibreoptic technology, nificant supraglottic obstruction of the airways. which allows the examination of even very small The were normal. The treatment infants at the bedside. Several modalities of treat- selected was endoscopic laser marsupialisation. ment have been advocated. These range from Tracheotomy was not performed. Follow-up fibre- repeated needle aspiration, mainly as an adjunct optic laryngoscopy 2 weeks after surgery showed to preserve the airways, to endoscopic laser mar- some residual redundant mucosa prolapsing supialisation, and to the transthyrohyoid from the ventricle. Some 6 months after surgery membrane approach that was used in this case. the patient was well. The literature suggests that patients with larger The final diagnosis for this patient was indeed cysts are best treated via an external approach saccular cyst of the larynx. because endoscopic treatment of larger lesions has resulted in a recurrence rate of 22% [2]. In conclusion, the saccular cyst is an uncom- Discussion mon congenital anomaly of the larynx that Saccular cyst of the larynx is an uncommon should be suspected in neonatal airway obstruc- benign cyst. The origin of laryngeal cysts is tion of immediate onset. Diagnosis is aided by the thought to be congenital or acquired. They can be newest generation of fibreoptic endoscopes and the result of prolonged intubation, laryngeal sur- rapid-acquisition CT scans. Smaller lesions may be gery or laryngoscleroma. A saccular cyst is treated endoscopically, whereas larger cysts are thought to result either from atresia of the orifice likely to require an external approach.

Editorial comment References 1. Ward RF, Jones J, Arnold JA. This Case presentation reflects the often-cited problem: “all that wheezes is not ”. This Surgical management of young student had suffered from increasing dyspnoea for 1 year and she had received inhaled congenital saccular cysts of the steroids, as well as systemic steroids and inhaled 2-agonists, without effect. This Case presenta- larynx. Ann Otol Rhinol Laryngol tion underlines the importance of following patients set upon treatment for a chronic disorder. In 1995; 104: 707–710. one way, the treatment can be looked upon as part of the continuing diagnostic process. If, as in 2. Thabet MH, Kotob K. Lateral this case, the treatment is not effective, an explanation should be sought. saccular cyst of the larynx. Flexible bronchoscopy gave the diagnosis of a saccular laryngeal cyst, as seen from the illustrative Aetiology, diagnosis and management. Laryngol Otol and beautiful photographs obtained through the bronchoscope, and the cyst was eventually treated 2001; 115: 293–297. surgically with success. 3. Wansa SA, Jones NS, Watkinson Perhaps, via careful examination by the referring physician, an alternative diagnosis to asthma J. Unusual laryngeal cyst. J could have been suspected earlier. Stridor by laryngeal causes is usually inspiratory, whereas the Laryngol Otol 1990; 104: dyspnoea caused by asthma is usually expiratory. Furthermore, the maximum expiratory and inspira- 145–146. tory flow–volume curve often demonstrates a distinct pattern with extrathoracal airways obstruc- 4. Shandilay M, Colreavy MP, tion. Both the expiratory and the inspiratory parts of the flow–volume curve are often flattened. Hughes J, et al. Endolaryngeal Whether the flow–volume curve in this case would have been a help in the diagnosis is not known, cyst presenting with acute repiratory distress. Clin but it is a reminder to us to employ flow–volume curves as part of the examination of patients with Otolaryngol Allied Sci 2004; 29: asthma, as well as in other respiratory disorders. 492–496.

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