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Laryngeal inflammatory pseudotumour: An unusual cause of airway obstruction
International Journal of Pediatric Otorhinolaryngology (2005) 69, 1253—1255
www.elsevier.com/locate/ijporl
CASE REPORT
Laryngeal inflammatory pseudotumour: An unusual cause of airway obstruction S.J. Hanna *, E. Blenke, R. Sharma, L.C. Knight Department of Otolaryngology, Brotherton Wing, Leeds General Infirmary, Great George Street, Leeds LS1 3EX, UK Received 11 December 2004; received in revised form 19 February 2005; accepted 20 February 2005
KEYWORDS Laryngeal neoplasms; Inflammatory pseudotumour; Larynx
Summary We report a rare case of a laryngeal pseudotumour in a child presenting with stridor, which was treated by endoscopic laryngeal resection, without the need for a tracheostomy. A short review of the literature is also presented. Although rare, laryngeal pseudotumour should be considered as part of the differential diagnosis of stridor and upper airway obstruction in children. # 2005 Elsevier Ireland Ltd. All rights reserved.
1. Introduction
2. Case report
An inflammatory pseudotumour is a non-neoplastic lesion with an inflammatory cell infiltrate which mimics a malignant neoplasm. A variety of names have been given to such lesions including plasma cell granulomas, plasmacytomas and histiocytomas [1,2]. These tumours are most commonly found in the lungs but isolated cases have been reported in the adult larynx. Only three cases in the paediatric larynx have been described in the literature. This is the first case in a child where an endoscopic approach has been adopted and a tracheostomy has been avoided.
A 7-year-old male was admitted as an emergency to his local District General Hospital with a 2-week history of progressive stridor, which had been preceded by a 4-month history of increasing hoarse voice, noisy breathing and cough. There was no history of foreign body ingestion or an acute infection. He had no past medical history of note and had been born at full term by normal vaginal delivery. He was using a salbutamol inhaler as required, had no known drug allergies, was up-to-date with immunisations and was developing normally. There was no family history of note and he had not been exposed to cigarette smoke. On examination he was noted to be comfortable, alert and orientated and apyrexial. He was noted to have inspiratory stridor. Chest examination revealed transmitted upper airway sounds only and his oxygen saturation on pulse oximetry was 98% on air. There was no cyanosis. A plain radiograph of the
* Corresponding author. Tel.: +44 113 3928040; fax: +44 113 3923165. E-mail address: [email protected] (S.J. Hanna).
0165-5876/$ — see front matter # 2005 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2005.02.023
1254
S.J. Hanna et al.
chest was normal. However, shortly after being admitted he had one short-lived episode of respiratory distress characterised by worsening stridor, sweating, cyanosis, intercostal recession and a drop in his oxygen saturation to 85% on air. This was selflimiting. He was subsequently transferred to our institution for evaluation. Direct laryngoscopy under general anaesthesia, using a 308 Storz—Hopkin rod endoscope, revealed a large, firm papillomatous mass arising from the right laryngeal ventricle causing marked airway obstruction (Fig. 1). This was debulked using a fibreopticdelivered diode laser, resulting in improved airway patency. A spontaneous ventilation technique without the use of an endotracheal tube was used. Samples were sent for histology and virology. He received intravenous steroids peroperatively followed by a 2-day course of oral steroids. He had an unremarkable postoperative recovery. Microscopic examination of the polypoid mass revealed the stroma to be composed of spindle cells which were moderately pleomorphic, with plump nuclei, inconspicuous nucleoli and a variable amount of amphophilic cytoplasm. The cells were in a loose watery myxoid stroma. There was no muscle differentiation and no cambium layer. The cells were positive for vimentin and actin and negative for desmin. There was no evidence of malignancy. It was concluded that this represented an inflammatory pseudotumour of the larynx. Human papilloma virus DNA was not detected in the tissue sample. Over the subsequent 8 months (1, 4 and 7 months after the initial procedure) he has had three further laryngoscopies and laser therapy to the laryngeal mass, resulting in its eradication. These were neces-
Fig. 1
sary because not all of the lesion was removed at the first procedure and also served to document progress; they were not done because of any clinical deterioration. He has had no further emergency admissions. He has no current airway symptoms and a normal voice.
3. Discussion The aetiology and pathogenesis of inflammatory pseudotumours is unknown, but it is postulated that they represent a non-specific inflammatory response to previous trauma, infection or irradiation [1—3]. There is no clear aetiological cause evident in our case. Inflammatory pseudotumours have been reported at a number of anatomical sites including the lung, gastrointestinal tract, abdominal cavity, spleen, breast, orbit and the central nervous system [1]. Although there have been several reports of such lesions in the adult larynx [3—11], we are aware of only three previous cases in children [12—14]; this is the first paediatric case in which a tracheostomy has not been required. It is also the first in which an endoscopic operative approach was used and the first paediatric case report to include intraoperative photographs of the lesion. The three previous case reports present a similar history of approximately 3 months duration of worsening dyspnoea progressing to stridor, dysphonia and persistent cough have also been a feature in two of the cases. The other features of the previous cases are summarised in Table 1. Of note, there has been no reported recurrence in any of these cases.
Endoscopic view of the laryngeal pseudotumour arising from the right laryngeal ventricle.
Laryngeal inflammatory pseudotumour: An unusual cause of airway obstruction
1255
Table 1 Summary of the previous paediatric cases Paper
Age M/F Symptoms (years)
Duration of Site symptoms (months)
Size (mm)
Cause
Treatment
10 15 None apparent Open excision, tracheostomy
Recurrence
Zapatero et al. [12]
6
M
Dysphonia, 3 dyspnoea, stridor
Cricotracheal
Munoz et al. [13]
5
M
Dyspnoea, 3 stridor ‘upper respiratory failure’
Subglottis
Surgical excision ? Not stated 5 18 Resection of route, thyroglossal duct remnant tracheostomy and associated ETT aged 1
Keen et al. [14]
11
F
Cough, wheeze, dyspnoea, stridor
3
Subglottis
5
None apparent
Open excision, tracheostomy
Disease free at 8 years
Disease free at 6 years
Abbreviations: M, male; F, female; ETT, endotracheal tube.
In conclusion, we present the case of a 7-year-old male presenting with progressive dyspnoea and stridor, which was due to a laryngeal pseudotumour and was successively treated by surgical excision. The macroscopic appearance of this lesion was similar to laryngeal viral papilloma. Endoscopic surgical excision without tracheostomy is recommended. Although rare, laryngeal pseudotumour should be considered as part of the differential diagnosis of stridor and upper airway obstruction in children.
References [1] B.M. Wenig, K. Devaney, M. Bisceglia, Inflamatory myofibroblastic tumor of the larynx, Cancer 76 (1995) 2217—2229. [2] F. Aijaz, A.U. Salam, S. Muzaffar, Y. Akbani, S.H. Hasan, Inlammatory pseudotumour of the trachea: report of a case in an eight-year-old child, J. Laryngol. Otol. 108 (1994) 613— 616. [3] J.J. Manni, J.J.S. Mulder, H.E. Schaafsma, U.J.G.M. van Haelst, Inflammatory pseudotumor of the subglottis, Eur. Arch. Otorhinolaryngol. 249 (1992) 16—19. [4] R.L. Mabry, Lymphoid pseudotumor of the nasopharynx and larynx, J. Laryngol. Otol. 81 (1967) 441—443. [5] M. Fradis, D. Rosenman, L. Podoshin, Y. Ben-David, A. Misslewitch, Steroid therapy for plasma cell granuloma of the larynx, Ear Nose Throat J. 67 (1988) 558—564.
[6] P. Zbaren, H. Lang, K. Beer, M. Becker, Plasma cell granuloma of the supraglottic larynx, J. Laryngol. Otol. 109 (1995) 895—898. [7] C. Albizzati, K.C.R.B. Ramesar, B.C. Davis, Plasma cell granuloma of the larynx, J. Laryngol. Otol. 102 (1988) 187—189. [8] B. Arellano, F.M. Gonzalez, G. Martinez, C. Salas, R. Ramirez-Camacho, J. Vergara, et al. Inflammatory pseudotumor of the larynx, Acta Otorinolaringol. Esp. 50 (1999) 236—238. [9] T.I. Al-Saleem, A.R. Peale, R. Robbins, C.M. Norris, Lymphocytic pseudotumour of the larynx, Laryngoscope 80 (1970) 133—136. [10] S.T. Anstey, J. Marais, W. Wallace, An unusual case of laryngeal pseudotumour, J. Laryngol. Otol. 109 (1995) 142—143. [11] A.P. Sclafani, C.P. Kimmelman, S.A. McCor5mick, Inflammtory pseudtumor of the larynx: comparison with orbital inflammatory pseudotumor with clinical implications, Otolaryngol. Head Neck Surg. 109 (1993) 548—551. [12] J. Zapatero, J. Lago, L. Madrigal, B. Baschwitz, E. Perez Rodriguez, R. Penalver, et al. Subglottic inflammatory pseudotumor in a 6-year-old child, Pediatr. Pulmonol. 6 (1989) 268—271. [13] A. Munoz, M. Villafruela, Inflammatory pseudotumor of the larynx: MR findings in a child, Pediatr. Radiol. 31 (2001) 459— 460. [14] M. Keen, H.T. Cho, L. Savetsky, Pseudotumor of the larynx–— an unusual cause of airway obstruction, Otolaryngol. Head Neck Sur. 94 (1986) 243—246.
www.elsevier.com/locate/ijporl
CASE REPORT
Laryngeal inflammatory pseudotumour: An unusual cause of airway obstruction S.J. Hanna *, E. Blenke, R. Sharma, L.C. Knight Department of Otolaryngology, Brotherton Wing, Leeds General Infirmary, Great George Street, Leeds LS1 3EX, UK Received 11 December 2004; received in revised form 19 February 2005; accepted 20 February 2005
KEYWORDS Laryngeal neoplasms; Inflammatory pseudotumour; Larynx
Summary We report a rare case of a laryngeal pseudotumour in a child presenting with stridor, which was treated by endoscopic laryngeal resection, without the need for a tracheostomy. A short review of the literature is also presented. Although rare, laryngeal pseudotumour should be considered as part of the differential diagnosis of stridor and upper airway obstruction in children. # 2005 Elsevier Ireland Ltd. All rights reserved.
1. Introduction
2. Case report
An inflammatory pseudotumour is a non-neoplastic lesion with an inflammatory cell infiltrate which mimics a malignant neoplasm. A variety of names have been given to such lesions including plasma cell granulomas, plasmacytomas and histiocytomas [1,2]. These tumours are most commonly found in the lungs but isolated cases have been reported in the adult larynx. Only three cases in the paediatric larynx have been described in the literature. This is the first case in a child where an endoscopic approach has been adopted and a tracheostomy has been avoided.
A 7-year-old male was admitted as an emergency to his local District General Hospital with a 2-week history of progressive stridor, which had been preceded by a 4-month history of increasing hoarse voice, noisy breathing and cough. There was no history of foreign body ingestion or an acute infection. He had no past medical history of note and had been born at full term by normal vaginal delivery. He was using a salbutamol inhaler as required, had no known drug allergies, was up-to-date with immunisations and was developing normally. There was no family history of note and he had not been exposed to cigarette smoke. On examination he was noted to be comfortable, alert and orientated and apyrexial. He was noted to have inspiratory stridor. Chest examination revealed transmitted upper airway sounds only and his oxygen saturation on pulse oximetry was 98% on air. There was no cyanosis. A plain radiograph of the
* Corresponding author. Tel.: +44 113 3928040; fax: +44 113 3923165. E-mail address: [email protected] (S.J. Hanna).
0165-5876/$ — see front matter # 2005 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2005.02.023
1254
S.J. Hanna et al.
chest was normal. However, shortly after being admitted he had one short-lived episode of respiratory distress characterised by worsening stridor, sweating, cyanosis, intercostal recession and a drop in his oxygen saturation to 85% on air. This was selflimiting. He was subsequently transferred to our institution for evaluation. Direct laryngoscopy under general anaesthesia, using a 308 Storz—Hopkin rod endoscope, revealed a large, firm papillomatous mass arising from the right laryngeal ventricle causing marked airway obstruction (Fig. 1). This was debulked using a fibreopticdelivered diode laser, resulting in improved airway patency. A spontaneous ventilation technique without the use of an endotracheal tube was used. Samples were sent for histology and virology. He received intravenous steroids peroperatively followed by a 2-day course of oral steroids. He had an unremarkable postoperative recovery. Microscopic examination of the polypoid mass revealed the stroma to be composed of spindle cells which were moderately pleomorphic, with plump nuclei, inconspicuous nucleoli and a variable amount of amphophilic cytoplasm. The cells were in a loose watery myxoid stroma. There was no muscle differentiation and no cambium layer. The cells were positive for vimentin and actin and negative for desmin. There was no evidence of malignancy. It was concluded that this represented an inflammatory pseudotumour of the larynx. Human papilloma virus DNA was not detected in the tissue sample. Over the subsequent 8 months (1, 4 and 7 months after the initial procedure) he has had three further laryngoscopies and laser therapy to the laryngeal mass, resulting in its eradication. These were neces-
Fig. 1
sary because not all of the lesion was removed at the first procedure and also served to document progress; they were not done because of any clinical deterioration. He has had no further emergency admissions. He has no current airway symptoms and a normal voice.
3. Discussion The aetiology and pathogenesis of inflammatory pseudotumours is unknown, but it is postulated that they represent a non-specific inflammatory response to previous trauma, infection or irradiation [1—3]. There is no clear aetiological cause evident in our case. Inflammatory pseudotumours have been reported at a number of anatomical sites including the lung, gastrointestinal tract, abdominal cavity, spleen, breast, orbit and the central nervous system [1]. Although there have been several reports of such lesions in the adult larynx [3—11], we are aware of only three previous cases in children [12—14]; this is the first paediatric case in which a tracheostomy has not been required. It is also the first in which an endoscopic operative approach was used and the first paediatric case report to include intraoperative photographs of the lesion. The three previous case reports present a similar history of approximately 3 months duration of worsening dyspnoea progressing to stridor, dysphonia and persistent cough have also been a feature in two of the cases. The other features of the previous cases are summarised in Table 1. Of note, there has been no reported recurrence in any of these cases.
Endoscopic view of the laryngeal pseudotumour arising from the right laryngeal ventricle.
Laryngeal inflammatory pseudotumour: An unusual cause of airway obstruction
1255
Table 1 Summary of the previous paediatric cases Paper
Age M/F Symptoms (years)
Duration of Site symptoms (months)
Size (mm)
Cause
Treatment
10 15 None apparent Open excision, tracheostomy
Recurrence
Zapatero et al. [12]
6
M
Dysphonia, 3 dyspnoea, stridor
Cricotracheal
Munoz et al. [13]
5
M
Dyspnoea, 3 stridor ‘upper respiratory failure’
Subglottis
Surgical excision ? Not stated 5 18 Resection of route, thyroglossal duct remnant tracheostomy and associated ETT aged 1
Keen et al. [14]
11
F
Cough, wheeze, dyspnoea, stridor
3
Subglottis
5
None apparent
Open excision, tracheostomy
Disease free at 8 years
Disease free at 6 years
Abbreviations: M, male; F, female; ETT, endotracheal tube.
In conclusion, we present the case of a 7-year-old male presenting with progressive dyspnoea and stridor, which was due to a laryngeal pseudotumour and was successively treated by surgical excision. The macroscopic appearance of this lesion was similar to laryngeal viral papilloma. Endoscopic surgical excision without tracheostomy is recommended. Although rare, laryngeal pseudotumour should be considered as part of the differential diagnosis of stridor and upper airway obstruction in children.
References [1] B.M. Wenig, K. Devaney, M. Bisceglia, Inflamatory myofibroblastic tumor of the larynx, Cancer 76 (1995) 2217—2229. [2] F. Aijaz, A.U. Salam, S. Muzaffar, Y. Akbani, S.H. Hasan, Inlammatory pseudotumour of the trachea: report of a case in an eight-year-old child, J. Laryngol. Otol. 108 (1994) 613— 616. [3] J.J. Manni, J.J.S. Mulder, H.E. Schaafsma, U.J.G.M. van Haelst, Inflammatory pseudotumor of the subglottis, Eur. Arch. Otorhinolaryngol. 249 (1992) 16—19. [4] R.L. Mabry, Lymphoid pseudotumor of the nasopharynx and larynx, J. Laryngol. Otol. 81 (1967) 441—443. [5] M. Fradis, D. Rosenman, L. Podoshin, Y. Ben-David, A. Misslewitch, Steroid therapy for plasma cell granuloma of the larynx, Ear Nose Throat J. 67 (1988) 558—564.
[6] P. Zbaren, H. Lang, K. Beer, M. Becker, Plasma cell granuloma of the supraglottic larynx, J. Laryngol. Otol. 109 (1995) 895—898. [7] C. Albizzati, K.C.R.B. Ramesar, B.C. Davis, Plasma cell granuloma of the larynx, J. Laryngol. Otol. 102 (1988) 187—189. [8] B. Arellano, F.M. Gonzalez, G. Martinez, C. Salas, R. Ramirez-Camacho, J. Vergara, et al. Inflammatory pseudotumor of the larynx, Acta Otorinolaringol. Esp. 50 (1999) 236—238. [9] T.I. Al-Saleem, A.R. Peale, R. Robbins, C.M. Norris, Lymphocytic pseudotumour of the larynx, Laryngoscope 80 (1970) 133—136. [10] S.T. Anstey, J. Marais, W. Wallace, An unusual case of laryngeal pseudotumour, J. Laryngol. Otol. 109 (1995) 142—143. [11] A.P. Sclafani, C.P. Kimmelman, S.A. McCor5mick, Inflammtory pseudtumor of the larynx: comparison with orbital inflammatory pseudotumor with clinical implications, Otolaryngol. Head Neck Surg. 109 (1993) 548—551. [12] J. Zapatero, J. Lago, L. Madrigal, B. Baschwitz, E. Perez Rodriguez, R. Penalver, et al. Subglottic inflammatory pseudotumor in a 6-year-old child, Pediatr. Pulmonol. 6 (1989) 268—271. [13] A. Munoz, M. Villafruela, Inflammatory pseudotumor of the larynx: MR findings in a child, Pediatr. Radiol. 31 (2001) 459— 460. [14] M. Keen, H.T. Cho, L. Savetsky, Pseudotumor of the larynx–— an unusual cause of airway obstruction, Otolaryngol. Head Neck Sur. 94 (1986) 243—246.