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Lower airway papillomatosis in children
International Journal of Pediatric Otorhinolaryngology (2003) 67, 1117 /1121
www.elsevier.com/locate/ijporl
CASE REPORT
Lower airway papillomatosis in children Lidia Zawadzka-Glosa,*, Anna Jakubowskab, Mieczyslaw Chmielika, Anna Bielickaa, Michal Brzewskib a
Department of Paediatric Otorhinolaryngology, The Medical University of Warsaw, 24 Marszalkowska Str., 00-576 Warsaw, Poland b Department of Paediatric Radiology, The Medical University of Warsaw, Warsaw, Poland Received 11 March 2003; received in revised form 30 May 2003; accepted 1 June 2003
KEYWORDS Juvenile laryngeal papilloma; Tracheobronchial papillomatosis; Lung lesions; Child
Summary Laryngeal papilloma in children is a frequent disease caused by human papilloma virus (HPV) type 6 or type 11. This disease has a tendency to recur and the changes are histologically benign. In some cases papilloma may affect the lower levels of the respiratory tract. In this study, among 90 patients treated for laryngeal papillomatosis, in four children papilloma of trachea, bronchi and lung tissue were detected in endoscopic and radiological examination. This constitutes 4.4% of all patients. Compact nodules and acquired cysts between 5 and 50 mm long were found in chest X-rays and in computerised tomography. These cysts appeared from 4 to 8 years after establishing a diagnosis of laryngeal papilloma, and 1 year after recognising papilloma in the trachea. In all four children the presence of nodules and cysts in the lungs was preceded by recurrent pneumonia, emphysema or atelectasis of the lungs. All children with laryngeal papillomatosis should have a chest X-ray. Detection of acquired cyst-like changes in lung tissue in children with laryngeal papillomatosis is a warning of future papilloma in the trachea and bronchi, with involvement of lung tissue. In differential diagnosis of these changes in the lungs we should take into consideration the presence of papilloma in the bronchi. A prognosis of papillomatosis in the lower airways in children is always serious. – 2003 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Laryngeal papillomatosis constitutes a disease of viral aetiology. The human papilloma virus (HPV) group of viral infections is the cause of papillomatous changes. Due to the molecular hybridisation technique, nearly 70 types of HPVs have been isolated; type 6 and type 11 are responsible for benign changes in the larynx and respiratory tract. Types 16, 18, 31, 45 HPV are rarely met, being
*Corresponding author. Tel./fax: /48-22-628-0584. E-mail address: [email protected] (L. Zawadzka-Glos).
responsible for progressive changes in the papilloma to malign neoplasm [6,10,14]. Clinically, laryngeal papilloma may be differentiated as juvenile-onset laryngeal papilloma and adult-onset laryngeal papilloma, but it is likely that the natural history of papilloma growth is one continuum of age distribution, usually with exacerbations and remissions. Juvenile papilloma are benign changes, and do not undergo malignant transformations. They are characterised by massive growth, frequently multifocal, a tendency to spread causing changes and involvement of healthy mucous membrane. They also show multiple recurrences, but often a regression of the disease at puberty is
0165-5876/03/$ - see front matter – 2003 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/S0165-5876(03)00191-5
1118
observed. Frequent recurrence of this disease and different periods of remission are connected with the existence of the virus in a latent form, which activates periodically. The etiological factors in juvenile laryngeal papilloma are HPV type 6 and the more aggressive type 11. Adult-onset laryngeal papilloma often grows as a single change, growing slowly, and sometimes malignant changes occur [6,11]. Juvenile laryngeal papilloma are most frequently localised in the vestibule of the larynx, vocal cords and in the subglottal area. These changes may affect the trachea, bronchi, and lung tissue. Tracheotomy is a procedure which accelerates the dissemination of changes lower in the respiratory tract [5]. According to some authors, lower respiratory tract papillomatosis concerns 1 /4% of patients with juvenile laryngeal papillomatosis. The prognosis in these cases is always serious [2,6,9].
2. Materials In the Department of Paediatric Otorhinolaryngology of the Medical University in Warsaw, between 1980 and 2002 we treated 90 children with laryngeal papillomatosis. In four children papilloma of the trachea, bronchi and lungs were recognised. The period of observation was 8 /16 years. Three children had tracheotomy due to progressing respiratory insufficiency.
L. Zawadzka-Glos et al.
were present 4 /8 years after establishing the original diagnosis. Three children died, and 1 has chronic respiratory insufficiency. The cause of first endoscopic examination of the larynx was progressing hoarseness, aphonia, laryngeal stridor, and progressive respiratory disturbances. In all patients there was recurrent pneumonia, confirmed by chest X-rays. Characteristic cystic changes or small nodules in the lungs appeared in the first patient at age 5, in the second at age 11 and in the third and fourth at 5 and 3 years, respectively. In all children the size of cysts was from 5 to 50 mm. Cysts were recognised 4 /8 years after establishing the diagnosis and about 1 year after finding papilloma in the larynx. Their number and size increased during observation, often with associated inflammatory symptoms, parenchymatous thickening of lung tissue, and levels of fluid in cysts in chest X-rays (Fig. 1). In all children the presence of cysts and nodules in the lungs was preceded by non-characteristic, but specially important changes, being recurrent pneumonia in three children at about 1 / 1.5 years after establishing the diagnosis of laryngeal papillomatosis, emphysema in two children, and atelectasis of the lung in one child (Fig. 2). In ultrasonography of the larynx papilloma were visible as unevenness in the contours of the anatomical structures of the larynx, which indents to the lumen of the larynx. Computerised tomography of the chest confirmed the presence of cysts and nodules in the lung tissue, and changes
3. Methods Papilloma of the larynx, trachea, and bronchi were recognised during laryngotracheobronchoscopy. These changes were removed in classical papillotomy using rigid bronchoscope and the Kleinsasser arrangement (Karl Storz GmbH & Co). All children had ultrasonography of the larynx and a chest X-ray. In two children combined surgical and pharmacological treatment with interferon alpha was applied; one of these children had had chemotherapy in another medical centre. The two remanding children were only treated surgically.
4. Results In all four children (two girls and two boys) a clinical diagnosis of laryngeal papillomatosis was made between 1 and 3 years of age; papilloma in the trachea were recognised 3 /7 years later, and characteristic radiological changes in the lungs
Fig. 1 Cysts in the lung tissue.
Lower airway papillomatosis in children
1119
5. Discussion
Fig. 2 Right middle lobus atelectasis in a child with lower airway papillomatosis.
typical of chronic pulmonary diseases (Figs. 3 and 4). Multiple papillotomies had indicated a progression of papillomatous changes in the trachea and bronchi, and the frequency of surgery was increased (Figs. 5 and 6). Three children died at the 13th, 9th and 7th year of age due to chronic respiratory insufficiency. One patient is now 22 years old, and 2 years ago had tracheotomy because of a massive growth of papilloma and progressive respiratory insufficiency.
Laryngeal papilloma are benign changes of viral aetiology. They are most common in 2 /3-year-old children, and rare in babies. In our Department the youngest child with laryngeal papillomatosis was 9 months old [4]. Depending on the site and extent of papillomatous changes, the following symptoms may dominate: hoarseness (in 45% cases), laryngeal stridor, expiratory /inspiratory dyspnoea, aphonia, recurrent pneumonia, recurrent laryngitis, or chronic cough. These last symptoms */not particularly specific for papilloma */may delay proper treatment because patients are diagnosed as having asthma, allergy, or chronic laryngitis. In about 4 /5% of cases papilloma may affect the trachea; in these cases pulmonary changes are detected in about 1% of cases [2,6]. In our material, children with papillomatosis of the trachea and bronchi constituted 4.4% of the group. It is known that extensive and easily disseminated changes are caused by HPV type 11 [11]. This replicates in the basal cells of the mucous membrane. These cells undergo abnormal proliferation, or their maturation process is disturbed. This results in aggregation of abnormal cells; clinically, it manifests as hyperplasia and papilloma [3]. This disease has a tendency to recur. Periods of remission and the course of a disease are difficult to anticipate. The course is related to the presence of latent forms of the virus in epithelial cells, which may activate at any time and may occupy new structures of the larynx, trachea and bronchi in an uncontrolled way [6]. The prognosis in cases when there are papilloma in the trachea, bronchi and in lung tissue is
Fig. 3 CT scan showing papilloma in the trachea.
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L. Zawadzka-Glos et al.
Fig. 4
Fig. 5 chea.
CT scan showing cystic changes in the lung tissue.
Endoscopic examination. Papilloma in the tra-
Fig. 6 Endoscopic examination. Papilloma in the left bronchus.
serious, and the mortality in these cases is high. In an examined group of four children we noted three deaths because of chronic respiratory insufficiency, and the condition of the fourth patient continues to deteriorate in spite of surgical and pharmacological treatment. Tracheotomy is a procedure which may facilitate the spread papilloma to the lower respiratory tract. Among treated patients progressive changes to the lower respiratory tract were observed. These data show how difficult the treatment of extensive papillomatous changes is when the lung tissue is also affected. Until now, no causal treatment has been found to be totally effective in the eradication of juvenile laryngeal papillomatosis. In some laryngological centres combined surgical (laserotherapy, classical papillotomy, argon plasma coagulation, photodynamic therapy using dihaematoporphyrin) and pharmacological treatment (Acyclovir, Groprinosin, Alpha-interferon, Cidofovir) is used [1,7,13,15]. Many therapeutic methods were rejected (antimitotic drugs, cytostatic agents, corticosteroids, oestrogens, radiotherapy) as a result of their ineffectiveness or side effects. These include, for example, malignant papillomatous transformations after radiotherapy [12]. In the described material a combined surgical and pharmacological (alpha-interferon) treatment was used in two children without any positive effects, and in one patient chemotherapy in another centre was used. No improvement was noted in these patients, and in a post-chemotherapy patient, leucopenia and a deterioration of general condition were observed. Radiological changes found in cases of
Lower airway papillomatosis in children
lower respiratory tract papillomatosis are connected with hyperplasia papillomatous changes in the pulmonary alveoli and in lung parenchyma. Cysts in the lungs increase concentrically and the walls of pulmonary alveoli become their walls causing destructive changes in pulmonary tissue. Laryngeal papilloma grow below the larynx, usually continuously to the main bronchi, rarely below. This localisation may suggest that papilloma detached during a surgical procedure or by coughing can be carried by aspiration to the lower respiratory tract. Thus, aspirated papilloma may be expectorated or removed, or may grow in pulmonary tissue. It is possible that cells or clumps of cells are being aspirated into the lower respiratory tract all the time. If the parenchyma of the lungs is involved in papillomatous changes, the patient will have symptoms of restrictive pulmonary disease connected with obturation of the upper respiratory tract [2,6,8,16]. In all children recurrent pneumonia was seen, and in endoscopic examinations we observed that papillomatous changes had descended gradually; radiological changes (cysts and nodules) appeared 4 /8 years after making the diagnosis, and 1 year after finding papilloma in the trachea. In physical examination these children demonstrated a soft aphonic voice and progressive respiratory insufficiency. At the beginning this was inspiratory /expiratory dyspnoea appearing only during effort, but later also appearing at rest. All children required complex laryngological and pulmonological care.
6. Conclusions 1) Recurrent pneumonia in children with recognised laryngeal papillomatosis may suggest the spread of the disease to lung tissue. 2) All children with recognised laryngeal papillomatosis should be given chest X-rays. 3) In differential diagnosis of cyst-like changes in lung tissue, it is necessary to take account of lower respiratory tract papillomatosis. 4) Tracheotomy is a procedure, which may facilitate the spreading of papilloma to the lower respiratory tract.
1121
5) The prognosis in cases when papilloma are in the trachea, bronchi and lung tissue is serious.
References [1] C. Armbruster, Novel treatment for recurrent respiratory papillomatosis, Expert Opin. Investig. Drugs 11 (8) (2002) 1139 /1148. [2] F.A. Blackledge, V.K. Anand, Tracheobronchial extension of recurrent respiratory papillomatosis, Ann. Otol. Rhinol. Laryngol. 109 (9) (2000) 812 /818. [3] M. Chmielik, M. De ˛bska, I. Jakubczyk, Brodawczaki krtani u dzieci, Nowa Pediatr. 11 (1) (1999) 22 /23. [4] M. Chmielik, M. De ˛bska, A. Bielicka, A. Gabryszewska, Brodawczaki krtani u 9-miesie ˛cznego chlopca-opis przypadku, Nowa Pediatr. 24 (4) (2000) 7 /8. [5] R.R. Cole, C.M. Myer, 3rd, R.T. Cotton, Tracheotomy in children with recurrent respiratory papillomatosis, Head Neck 11 (3) (1989) 226 /230. [6] R.T. Cotton, C.M. Meyer, Recurrent Respiratory Papillomatosis. Practical Pediatric Otolaryngology, Lippincott-Raven Publishers, Philadelphia, 1999, pp. 639 /658. [7] J.P. Harcourt, G. Worley, S.E. Leighton, Cimetidine treatment for recurrent respiratory papillomatosis, Int. J. Pediatr. Otorhinolaryngol. 51 (2) (1999) 109 /113. [8] A. Jakubowska, L. Zawadzka-Glos, M. Brzewski, A. Biejat, A. Marcin ´ski, Zmiany plucne w mlodzien ´czych brodawczakach krtani i tchawicy, Polish J. Radiol. 67 (3) (2002) 48 / 52. [9] S.S. Kramer, W.D. Wchunt, J.T. Stocker, H. Kashima, Pulmonary manifestations of juvenile laryngotracheal papillomatosis, Am. J. Roentgenol. 144 (4) (1985) 687 /694. [10] L.N. Murray, R.I.L. Miller, Recurrent respiratory papillomatosis, J. La State Med. Soc. 150 (10) (1998) 456 /459. [11] R. Rabah, W.D. Lancaster, R. Thomas, L. Gregoire, Human papillomavirus-11-associated recurrent respiratory papillomatosis is more aggressive than human papillomavirus-6associated disease, Pediatr. Dev. Pathol. 4 (1) (2001) 68 / 72. [12] S.E. Siegel, H. Isaacs, S.R. Cohen, P. Stanley, Malignant transformation of tracheobronchial juvenile papillomatosis without prior radiotherapy, Ann. Otol. 88 (1979) 192 /197. [13] R. Snoeck, G. Andrei, E. De Clerq, Cidofovir in the treatment of HPV-associated lesions, Verh K Acad. Geneeskd Belg. 63 (2) (2001) 93 /120. [14] C. Swygart, Human papillomavirus: disease and laboratory diagnosis, Br. J. Biomed. Sci. 54 (4) (1997) 299 /303. [15] I. Van Valckenborgh, W. Wellens, K. De Boeck, R. Snoeck, et al., Systemic cidofovir in papillomatosis, Clin. Infect. Dis. 32 (3) (2001) E62 /E64. [16] A. Vourtsi, A. Gouliamos, et al., CT appearance of solitary and multiple cystic and cavitary lung lesions, Eur. Radiol. 11 (2001) 612 /622.
www.elsevier.com/locate/ijporl
CASE REPORT
Lower airway papillomatosis in children Lidia Zawadzka-Glosa,*, Anna Jakubowskab, Mieczyslaw Chmielika, Anna Bielickaa, Michal Brzewskib a
Department of Paediatric Otorhinolaryngology, The Medical University of Warsaw, 24 Marszalkowska Str., 00-576 Warsaw, Poland b Department of Paediatric Radiology, The Medical University of Warsaw, Warsaw, Poland Received 11 March 2003; received in revised form 30 May 2003; accepted 1 June 2003
KEYWORDS Juvenile laryngeal papilloma; Tracheobronchial papillomatosis; Lung lesions; Child
Summary Laryngeal papilloma in children is a frequent disease caused by human papilloma virus (HPV) type 6 or type 11. This disease has a tendency to recur and the changes are histologically benign. In some cases papilloma may affect the lower levels of the respiratory tract. In this study, among 90 patients treated for laryngeal papillomatosis, in four children papilloma of trachea, bronchi and lung tissue were detected in endoscopic and radiological examination. This constitutes 4.4% of all patients. Compact nodules and acquired cysts between 5 and 50 mm long were found in chest X-rays and in computerised tomography. These cysts appeared from 4 to 8 years after establishing a diagnosis of laryngeal papilloma, and 1 year after recognising papilloma in the trachea. In all four children the presence of nodules and cysts in the lungs was preceded by recurrent pneumonia, emphysema or atelectasis of the lungs. All children with laryngeal papillomatosis should have a chest X-ray. Detection of acquired cyst-like changes in lung tissue in children with laryngeal papillomatosis is a warning of future papilloma in the trachea and bronchi, with involvement of lung tissue. In differential diagnosis of these changes in the lungs we should take into consideration the presence of papilloma in the bronchi. A prognosis of papillomatosis in the lower airways in children is always serious. – 2003 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Laryngeal papillomatosis constitutes a disease of viral aetiology. The human papilloma virus (HPV) group of viral infections is the cause of papillomatous changes. Due to the molecular hybridisation technique, nearly 70 types of HPVs have been isolated; type 6 and type 11 are responsible for benign changes in the larynx and respiratory tract. Types 16, 18, 31, 45 HPV are rarely met, being
*Corresponding author. Tel./fax: /48-22-628-0584. E-mail address: [email protected] (L. Zawadzka-Glos).
responsible for progressive changes in the papilloma to malign neoplasm [6,10,14]. Clinically, laryngeal papilloma may be differentiated as juvenile-onset laryngeal papilloma and adult-onset laryngeal papilloma, but it is likely that the natural history of papilloma growth is one continuum of age distribution, usually with exacerbations and remissions. Juvenile papilloma are benign changes, and do not undergo malignant transformations. They are characterised by massive growth, frequently multifocal, a tendency to spread causing changes and involvement of healthy mucous membrane. They also show multiple recurrences, but often a regression of the disease at puberty is
0165-5876/03/$ - see front matter – 2003 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/S0165-5876(03)00191-5
1118
observed. Frequent recurrence of this disease and different periods of remission are connected with the existence of the virus in a latent form, which activates periodically. The etiological factors in juvenile laryngeal papilloma are HPV type 6 and the more aggressive type 11. Adult-onset laryngeal papilloma often grows as a single change, growing slowly, and sometimes malignant changes occur [6,11]. Juvenile laryngeal papilloma are most frequently localised in the vestibule of the larynx, vocal cords and in the subglottal area. These changes may affect the trachea, bronchi, and lung tissue. Tracheotomy is a procedure which accelerates the dissemination of changes lower in the respiratory tract [5]. According to some authors, lower respiratory tract papillomatosis concerns 1 /4% of patients with juvenile laryngeal papillomatosis. The prognosis in these cases is always serious [2,6,9].
2. Materials In the Department of Paediatric Otorhinolaryngology of the Medical University in Warsaw, between 1980 and 2002 we treated 90 children with laryngeal papillomatosis. In four children papilloma of the trachea, bronchi and lungs were recognised. The period of observation was 8 /16 years. Three children had tracheotomy due to progressing respiratory insufficiency.
L. Zawadzka-Glos et al.
were present 4 /8 years after establishing the original diagnosis. Three children died, and 1 has chronic respiratory insufficiency. The cause of first endoscopic examination of the larynx was progressing hoarseness, aphonia, laryngeal stridor, and progressive respiratory disturbances. In all patients there was recurrent pneumonia, confirmed by chest X-rays. Characteristic cystic changes or small nodules in the lungs appeared in the first patient at age 5, in the second at age 11 and in the third and fourth at 5 and 3 years, respectively. In all children the size of cysts was from 5 to 50 mm. Cysts were recognised 4 /8 years after establishing the diagnosis and about 1 year after finding papilloma in the larynx. Their number and size increased during observation, often with associated inflammatory symptoms, parenchymatous thickening of lung tissue, and levels of fluid in cysts in chest X-rays (Fig. 1). In all children the presence of cysts and nodules in the lungs was preceded by non-characteristic, but specially important changes, being recurrent pneumonia in three children at about 1 / 1.5 years after establishing the diagnosis of laryngeal papillomatosis, emphysema in two children, and atelectasis of the lung in one child (Fig. 2). In ultrasonography of the larynx papilloma were visible as unevenness in the contours of the anatomical structures of the larynx, which indents to the lumen of the larynx. Computerised tomography of the chest confirmed the presence of cysts and nodules in the lung tissue, and changes
3. Methods Papilloma of the larynx, trachea, and bronchi were recognised during laryngotracheobronchoscopy. These changes were removed in classical papillotomy using rigid bronchoscope and the Kleinsasser arrangement (Karl Storz GmbH & Co). All children had ultrasonography of the larynx and a chest X-ray. In two children combined surgical and pharmacological treatment with interferon alpha was applied; one of these children had had chemotherapy in another medical centre. The two remanding children were only treated surgically.
4. Results In all four children (two girls and two boys) a clinical diagnosis of laryngeal papillomatosis was made between 1 and 3 years of age; papilloma in the trachea were recognised 3 /7 years later, and characteristic radiological changes in the lungs
Fig. 1 Cysts in the lung tissue.
Lower airway papillomatosis in children
1119
5. Discussion
Fig. 2 Right middle lobus atelectasis in a child with lower airway papillomatosis.
typical of chronic pulmonary diseases (Figs. 3 and 4). Multiple papillotomies had indicated a progression of papillomatous changes in the trachea and bronchi, and the frequency of surgery was increased (Figs. 5 and 6). Three children died at the 13th, 9th and 7th year of age due to chronic respiratory insufficiency. One patient is now 22 years old, and 2 years ago had tracheotomy because of a massive growth of papilloma and progressive respiratory insufficiency.
Laryngeal papilloma are benign changes of viral aetiology. They are most common in 2 /3-year-old children, and rare in babies. In our Department the youngest child with laryngeal papillomatosis was 9 months old [4]. Depending on the site and extent of papillomatous changes, the following symptoms may dominate: hoarseness (in 45% cases), laryngeal stridor, expiratory /inspiratory dyspnoea, aphonia, recurrent pneumonia, recurrent laryngitis, or chronic cough. These last symptoms */not particularly specific for papilloma */may delay proper treatment because patients are diagnosed as having asthma, allergy, or chronic laryngitis. In about 4 /5% of cases papilloma may affect the trachea; in these cases pulmonary changes are detected in about 1% of cases [2,6]. In our material, children with papillomatosis of the trachea and bronchi constituted 4.4% of the group. It is known that extensive and easily disseminated changes are caused by HPV type 11 [11]. This replicates in the basal cells of the mucous membrane. These cells undergo abnormal proliferation, or their maturation process is disturbed. This results in aggregation of abnormal cells; clinically, it manifests as hyperplasia and papilloma [3]. This disease has a tendency to recur. Periods of remission and the course of a disease are difficult to anticipate. The course is related to the presence of latent forms of the virus in epithelial cells, which may activate at any time and may occupy new structures of the larynx, trachea and bronchi in an uncontrolled way [6]. The prognosis in cases when there are papilloma in the trachea, bronchi and in lung tissue is
Fig. 3 CT scan showing papilloma in the trachea.
1120
L. Zawadzka-Glos et al.
Fig. 4
Fig. 5 chea.
CT scan showing cystic changes in the lung tissue.
Endoscopic examination. Papilloma in the tra-
Fig. 6 Endoscopic examination. Papilloma in the left bronchus.
serious, and the mortality in these cases is high. In an examined group of four children we noted three deaths because of chronic respiratory insufficiency, and the condition of the fourth patient continues to deteriorate in spite of surgical and pharmacological treatment. Tracheotomy is a procedure which may facilitate the spread papilloma to the lower respiratory tract. Among treated patients progressive changes to the lower respiratory tract were observed. These data show how difficult the treatment of extensive papillomatous changes is when the lung tissue is also affected. Until now, no causal treatment has been found to be totally effective in the eradication of juvenile laryngeal papillomatosis. In some laryngological centres combined surgical (laserotherapy, classical papillotomy, argon plasma coagulation, photodynamic therapy using dihaematoporphyrin) and pharmacological treatment (Acyclovir, Groprinosin, Alpha-interferon, Cidofovir) is used [1,7,13,15]. Many therapeutic methods were rejected (antimitotic drugs, cytostatic agents, corticosteroids, oestrogens, radiotherapy) as a result of their ineffectiveness or side effects. These include, for example, malignant papillomatous transformations after radiotherapy [12]. In the described material a combined surgical and pharmacological (alpha-interferon) treatment was used in two children without any positive effects, and in one patient chemotherapy in another centre was used. No improvement was noted in these patients, and in a post-chemotherapy patient, leucopenia and a deterioration of general condition were observed. Radiological changes found in cases of
Lower airway papillomatosis in children
lower respiratory tract papillomatosis are connected with hyperplasia papillomatous changes in the pulmonary alveoli and in lung parenchyma. Cysts in the lungs increase concentrically and the walls of pulmonary alveoli become their walls causing destructive changes in pulmonary tissue. Laryngeal papilloma grow below the larynx, usually continuously to the main bronchi, rarely below. This localisation may suggest that papilloma detached during a surgical procedure or by coughing can be carried by aspiration to the lower respiratory tract. Thus, aspirated papilloma may be expectorated or removed, or may grow in pulmonary tissue. It is possible that cells or clumps of cells are being aspirated into the lower respiratory tract all the time. If the parenchyma of the lungs is involved in papillomatous changes, the patient will have symptoms of restrictive pulmonary disease connected with obturation of the upper respiratory tract [2,6,8,16]. In all children recurrent pneumonia was seen, and in endoscopic examinations we observed that papillomatous changes had descended gradually; radiological changes (cysts and nodules) appeared 4 /8 years after making the diagnosis, and 1 year after finding papilloma in the trachea. In physical examination these children demonstrated a soft aphonic voice and progressive respiratory insufficiency. At the beginning this was inspiratory /expiratory dyspnoea appearing only during effort, but later also appearing at rest. All children required complex laryngological and pulmonological care.
6. Conclusions 1) Recurrent pneumonia in children with recognised laryngeal papillomatosis may suggest the spread of the disease to lung tissue. 2) All children with recognised laryngeal papillomatosis should be given chest X-rays. 3) In differential diagnosis of cyst-like changes in lung tissue, it is necessary to take account of lower respiratory tract papillomatosis. 4) Tracheotomy is a procedure, which may facilitate the spreading of papilloma to the lower respiratory tract.
1121
5) The prognosis in cases when papilloma are in the trachea, bronchi and lung tissue is serious.
References [1] C. Armbruster, Novel treatment for recurrent respiratory papillomatosis, Expert Opin. Investig. Drugs 11 (8) (2002) 1139 /1148. [2] F.A. Blackledge, V.K. Anand, Tracheobronchial extension of recurrent respiratory papillomatosis, Ann. Otol. Rhinol. Laryngol. 109 (9) (2000) 812 /818. [3] M. Chmielik, M. De ˛bska, I. Jakubczyk, Brodawczaki krtani u dzieci, Nowa Pediatr. 11 (1) (1999) 22 /23. [4] M. Chmielik, M. De ˛bska, A. Bielicka, A. Gabryszewska, Brodawczaki krtani u 9-miesie ˛cznego chlopca-opis przypadku, Nowa Pediatr. 24 (4) (2000) 7 /8. [5] R.R. Cole, C.M. Myer, 3rd, R.T. Cotton, Tracheotomy in children with recurrent respiratory papillomatosis, Head Neck 11 (3) (1989) 226 /230. [6] R.T. Cotton, C.M. Meyer, Recurrent Respiratory Papillomatosis. Practical Pediatric Otolaryngology, Lippincott-Raven Publishers, Philadelphia, 1999, pp. 639 /658. [7] J.P. Harcourt, G. Worley, S.E. Leighton, Cimetidine treatment for recurrent respiratory papillomatosis, Int. J. Pediatr. Otorhinolaryngol. 51 (2) (1999) 109 /113. [8] A. Jakubowska, L. Zawadzka-Glos, M. Brzewski, A. Biejat, A. Marcin ´ski, Zmiany plucne w mlodzien ´czych brodawczakach krtani i tchawicy, Polish J. Radiol. 67 (3) (2002) 48 / 52. [9] S.S. Kramer, W.D. Wchunt, J.T. Stocker, H. Kashima, Pulmonary manifestations of juvenile laryngotracheal papillomatosis, Am. J. Roentgenol. 144 (4) (1985) 687 /694. [10] L.N. Murray, R.I.L. Miller, Recurrent respiratory papillomatosis, J. La State Med. Soc. 150 (10) (1998) 456 /459. [11] R. Rabah, W.D. Lancaster, R. Thomas, L. Gregoire, Human papillomavirus-11-associated recurrent respiratory papillomatosis is more aggressive than human papillomavirus-6associated disease, Pediatr. Dev. Pathol. 4 (1) (2001) 68 / 72. [12] S.E. Siegel, H. Isaacs, S.R. Cohen, P. Stanley, Malignant transformation of tracheobronchial juvenile papillomatosis without prior radiotherapy, Ann. Otol. 88 (1979) 192 /197. [13] R. Snoeck, G. Andrei, E. De Clerq, Cidofovir in the treatment of HPV-associated lesions, Verh K Acad. Geneeskd Belg. 63 (2) (2001) 93 /120. [14] C. Swygart, Human papillomavirus: disease and laboratory diagnosis, Br. J. Biomed. Sci. 54 (4) (1997) 299 /303. [15] I. Van Valckenborgh, W. Wellens, K. De Boeck, R. Snoeck, et al., Systemic cidofovir in papillomatosis, Clin. Infect. Dis. 32 (3) (2001) E62 /E64. [16] A. Vourtsi, A. Gouliamos, et al., CT appearance of solitary and multiple cystic and cavitary lung lesions, Eur. Radiol. 11 (2001) 612 /622.