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Vol. 8. Issue 4.
Pages 329-340 (July - August 2002)
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Vol. 8. Issue 4.
Pages 329-340 (July - August 2002)
ARTIGO ORIGINAL/ORIGINAL ARTICLE
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Traqueobroncopatia osteocondroplástica
Tracheobronchopathia osteochondroplastica
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João Moura e Sá*, José Almeida**, Joana Amado***, Beatriz Fernandes***, José Caminha*, José Miguel Ferraz****
* Assistente Hospitalar Graduado de Pneumologia
** Assistente Hospitalar de Pneumologia
*** Interna Complementar de Pneumologia
**** Chefe de Serviço de Pneumologia
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RESUMO

A traqueobroncopatia osteocondroplástica (TO) é uma doença rara e de etiopatogenia desconhecida. Caracteriza-se pela presença de múltiplos nódulos osteocartilagíneos na submucosa da árvore laringotraqueobrônquica, que se dispõem caracteristicamente na superfície luminal das suas paredes anteriores e laterais. A evolução é habitualmente benigna e silenciosa, pelo que a sua incidência in vivo (1:125 - 1:6000) poderá estar subestimada.

O aspecto broncoscópico típico de nódulos de consistência dura, tipo “grão de arroz” na submucosa e a confirmação histológica nos fragmentos biopsados são os elementos-chave para o diagnóstico.

Não se conhece terapêutica específica, sendo o tratamento, quando necessário, quase sempre exclusivamente sintomático. Numa minoria de casos cursa com complicações graves — infecção, hemoptises e obstrução—que poderão implicar o tratamento broncoscópico / cirúrgico das lesões.

Os autores procederam à análise retrospectiva da incidência, apresentação clínica e broncoscópica dos casos de TO diagnosticados na Unidade de Broncologia do Centro Hospitalar de Vila Nova de Gaia durante 14 anos, de 1987 a 2001.

Neste período foram realizadas 16 888 broncoscopias. Em 13 doentes (11 homens, 2 mulheres; idade média - 59,5±12,4 anos) foi diagnosticada acidentalmente traqueobroncopatia osteocondroplástica. Todos os casos, confirmados histologicamente, apresentaram o aspecto broncoscópico típico de TO e em nenhum foi necessário tratamento. Em quatro doentes identificaram-se simultaneamente neoplasias malignas do pulmão.

Na série descrita a incidência estimada de TO (1:1299 broncoscopias) sobrepôs-se à publicada na literatura e, tal como documentado por alguns autores, identificaram-se simultaneamente vários casos de neoplasias malignas do pulmão.

REV PORT PNEUMOL 2002; VIII(4): 329-339

Palavras-chave:
traqueobroncopatia osteocondroplástica
broncoscopia rígida
broncofibroscopia
traqueia
brônquios
ABSTRACT

Tracheobronchopathia osteochondroplastica (TO) is a rare disease of unknown pathogenesis and etiology. It is characterized by the presence of multiple submucosal osteocartilaginous nodules that involve luminal surfaces of anterior and lateral walls of the laryngotracheobronchial tree. In most cases the dis ease is benign and silent and its incidence in vivo (1:125 - 1:6000) is probably underestimated.

Typical bronchoscopic examination of submucosal hard nodules and histologic confirmation in biopsy specimens are essential in establishing the diagnosis. In the absence of specific treatment, management is aimed at symptoms relieve. A minority of patients develop serious complications — infection, hemoptysis and obstruction — and surgical or endoscopic excision may be necessary.

Incidence, clinical and bronchoscopic presentation of TO cases diagnosed in the Bronchology Unit of Centro Hospitalar de Vila Nova de Gaia during 14 years (since 1987 to 2001) were reviewed.

In this period, 16 888 bronchoscopies were performed. TO was incidentally diagnosed in 13 patients (11 men, 2 women; mean age - 59,5±12,4years). Bronchoscopy showed the characteristic endoscopic findings and all cases were histopathologically proven. None of the patients received treatment. Four patients presented with concomitant malignant lung cancer.

The incidence of TO in our series (1: 1299 bronchoscopies) is similar to that reported in the literature and several cases of malignant neoplasms associated with TO were identified as has been observed by other authors.

REV PORT PNEUMOL 2002; VIII(4): 329-339

Key-words:
tracheobronchopathia osteochondroplastica
rigid bronchoscopy
flexible bronchoscopy
trachea
bronchi
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BIBLIOGRAFIA
[1.]
J.B. Dalgaar.
Tracheopathia chondro-osteoplastica: a case elucidating the problems concerning development and ossification of elastic cartilage.
Acta Pathol Microbiol Scand, 24 (1947), pp. 118-134
[2.]
S. Wilks.
Ossific deposits on the larynx, trachea and bronchi.
Trans Pathol Soc Lond, 8 (1857), pp. 88
[3.]
D. Coetmeur, G. Bovyn, P. Leroux, M. Niel-Duriez.
Tracheobronchopatia osteochondroplastica presenting at the time of a difficult intubation.
Respiratory Medicine, 91 (1997), pp. 496-498
[4.]
J.P. Duchateau, L.M. Delvigne, J. Melon.
La tracheopathie osteoplastique.
Acta Oto-Rhino-Laryngologica Belgica, 29 (1975), pp. 661-670
[5.]
D.L. Ragaini, D.P. Piccol.
Tracheo-bronchopathia chodro-osteoplastica.
Riv Anat Pat, 13 (1957), pp. 188-208
[6.]
D.J. Pounder, A.S. Pieterse.
Tracheopathia osteoplastica: Report of four cases.
J Pathol, 14 (1982), pp. 429-433
[7.]
G. Primer.
Tracheobronchopatia osteochondroplastica.
Prax Klin Pneumol, 33 (1979), pp. 1060-1063
[8.]
M.A.M.F. Nierop, S.S. Wagenaar, J.M.M. Bosch, C.J.J. Westermann.
Tracheobronchopathia osteochondroplastica. Report of four cases.
Eur J Respir Dis, 64 (1983), pp. 129-133
[9.]
D.C. Smith, R. Pillai, C.E. Gillbe.
Tracheopathia osteochondroplastica. A cause of unexpected difficulty in tracheal intubation.
Anaesthesia, 42 (1987), pp. 536-538
[10.]
U.B.S. Prakash.
What is Tracheo(broncho)pathia Osteo (chondro)plastica?.
Journal of Bronchology, 8 (2001), pp. 75-76
[11.]
J.A. Verschakelen, L.F. Roex, F. Vanneste, A.L. Beart, L. Biliet.
Tracheobronchopathia osteochondroplastica. Diagnosis on convencional radiology and computed tomography.
Fortschr Rontgenstr, 147 (1987), pp. 575-576
[12.]
J.E. Manning, J.G. Goldin, R.B. Shpiner, D.R. Aberle.
Case report: Tracheobronchopathia osteochondroplastica.
Clinical Radiology, 53 (1998), pp. 302-304
[13.]
C.N. Meyer, M. Dossing, H. Broholm.
Trachebronchopathia osteochondroplastica.
Respiratory Medicine, 9 (1997), pp. 499-502
[14.]
R.A. Harma, S. Suurkari.
Tracheopathia chondroosteoplastica. A clinical study of thirty cases.
Acta Otolaryngol, 84 (1977), pp. 118-123
[15.]
M.D. Clee, J.M. Anderson, R.N. Johnston.
Clinical Aspects of Tracheobronchopathia Osteochondroplastica.
Br J Dis Chest, 77 (1983), pp. 308-314
[16.]
P.E. Baugnee, L.M. Delaunois.
Mycobacterium avium-intracellulare associated with tracheobronchopathia osteochondroplastica.
Eur Respir J, 8 (1995), pp. 180-182
[17.]
U.S. Prakash, A.E. McCulloch, E.S. Edell, D.M.M. Nienhuis.
Tracheopathia osteoplastica: familial occurrence.
Mayo Clin Proc, 64 (1989), pp. 1091-1096
[18.]
E. Vaheri.
Tracheopathia Osteoplastica.
Acta Otolaryngol, 64 (1967), pp. 251-255
[19.]
O. Jepsen, H. Sorensen.
Tracheopathia osteoplastica and ozena.
Acta Otolaryngol, 51 (1960), pp. 79-83
[20.]
L. Smid, B. Lavrencak, M. Zargi.
Laryngo-tracheo-bronchopathia chondro-osteoplastica.
The Journal of Laryngology and Otology, 106 (1992), pp. 845-848
[21.]
K. Hiebaum.
Uber Knochenbildungen in Lunge und Trachea.
Frank Z Pathol, 47 (1934), pp. 249-255
[22.]
D.J.B. Ashley.
Bone metaplasia in trachea and bronchi.
J Pathol, 102 (1970), pp. 186-188
[23.]
K.J. Hempel, A. Glaser.
Zur Pathogenese der Tracheopathia chondro-osteoplastica.
Virchows Arch Pathol Anat Physiol, 331 (1958), pp. 36-50
[24.]
H. Spencer.
Pathology of the lung, Pergamon Press, (1962), pp. 591
[25.]
H. Ribbert.
Ueber die entstehung die Geschwulste.
Dtsch Med Wochenschr, 2 (1895), pp. 24-26
[26.]
A.Y. Chen, D.T. Donovan.
Impaired ciliary clearance from tracheopathia osteoplastica of the upper respiratory tract.
Otolaryngology - Head and Neck Surgery, 117 (1997), pp. S102-S104
[27.]
S.S. Park, D.H. Shin, D.H. Lee, et al.
Tracheopatia osteoplastica simulating asthmatic symptoms: diagnosis by bronchoscopy and computorized tomography.
Respiration, 62 (1995), pp. 43-45
[28.]
S. Vilkman, T. Keistinen.
Tracheobronchopathia osteochondroplastica: report of a young man with severe disease and retrospective review of 18 cases.
Respiration, 62 (1995), pp. 151-154
[29.]
S. Mariotta, G. Pallone, G. Pedicelli, A. Bisetti.
Journal of Computer Assisted Tomography, 21 (1997), pp. 418-420
[30.]
B. Wazières, T. Fest, J.C. Dalphin, E. Ranfaing, G. Capellier, J.L. Dupond.
Trachéopathie ostéochondroplastique associée à une dermatomyosite:à propos d’un cas.
Rev Med Intern, 15 (1994), pp. 121-123
[31.]
C. Roggenbuck, T. Haut, N. De Wall, et al.
Simultaneous occurrence of tracheopathia osteochondroplastica and mucoepidermoide carcinoma.
Chirurg, 66 (1995), pp. 232-234
[32.]
J.B. Dalgaard.
Lungecancer ved tracheopathia chondroosteoplastica.
Nord Med, 53 (1955), pp. 572-575
[33.]
W. Kissler, W. Wierich, D. Meessen.
Tracheobronchopathia osteochondroplastica und bronkialkarzinom.
Prax Klin Pneumol, 31 (1977), pp. 660-663
[34.]
J.C. Bioque, N. Feu, J.M. Rubio, M.A. Martín, F.L. García, J.M. Bravo, F. Bravo, A. Alvarez, C. Villar, L. Muñoz.
Tracheobronchopathia Osteochondroplastica. Clinical Study and Follow-Up in Nine Cases.
Journal of Bronchology, 8 (2001), pp. 78-83
[35.]
S. Morita, N. Yokoyama, S. Yamashita, et al.
Tracheopathia osteochondroplastica complicated with thyroid cancer: case report and review of the literature in Japan.
Jpn J Med, 29 (1990), pp. 637-641
[36.]
M. Kishikawa, Y. Nagashima.
Tracheobronchopathia osteoplastica complicating thymoma.
Jpn J Thorac Dis, 16 (1978), pp. 347-351
[37.]
D.M. Nienhuis, U.B.S. Prakash, E.S. Edell.
Tracheobronchopathia Osteochondroplastica.
Ann Otol Rhinol Laryngol, 99 (1990), pp. 689-694
[38.]
M.K. Hodges, E. Israel.
Tracheobronchopathia Osteochondroplastica Presenting as Right Middle Lobe Collapse.
Chest, 94 (1988), pp. 842-844
[39.]
R. Lundgren, N.L. Stjernberg.
Tracheobronchopatia osteochondroplastica: a clinical bronchoscopic and spirometric study.
Chest, 80 (1981), pp. 706-709
[40.]
D. Bergeron, Y. Cormier, M. Desmeules.
Tracheobronchopathia osteo-chondroplastica.
Am Rev Respir Dis, 114 (1976), pp. 803-806
[41.]
R.B. Warner, P.K. Barson.
Tracheobronchopathia osteochondroplastica diagnosed as a result of difficult intubation.
Anesthesiology, 51 (1979), pp. 269-270
[42.]
R.H. Young, R.E. Sandstrom, G.J. Mark.
Tracheopathia osteoplastica: clinical, radiologic and pathologic correlation.
J Thorac Cardiovasc Surg, 79 (1980), pp. 537-541
[43.]
E.M. Webb, B.M. Elicker, W.R. Webb.
Using CT to diagnose nonneoplastic tracheal abnormalities.
AJR, 174 (2000), pp. 1315-1321
[44.]
K. Bottles, D.A. Nyberg, M. Clark, W.A. Hinchcliffe.
CT diagnosis of tracheobronchopatia osteochondroplastica.
J Comput Assist Tomogr, 7 (1983), pp. 324-327
[45.]
M. Hirsh, J. Goldstein, F. Tovi, S.G. Gerzof.
Diagnosis of tracheopathia osteoplastica by computed tomography.
Ann Otol Rhinol Laryngol, 94 (1985), pp. 217-219
[46.]
H. Onitsuka, N. Hirose, K. Watanabe, et al.
Computed tomography of tracheopathia osteoplastica.
American Journal of Roentgenology, 140 (1983), pp. 268-270
[47.]
H. Tukiainen, Torkko, E.O. Terho.
Lung function in patients with tracheopathia osteochondroplastica.
Eur Respir J, 1 (1988), pp. 632-635
[48.]
A. Alvi, M.J. Shikowitz, F. Panjvani.
Tracheobronchopathia osteochodroplastica. Pathologic quiz.
Arch Oto Head Neck Surg, 121 (1995), pp. 585-586
[49.]
G. Mangiapan, M. Prosper, M.H. Marais, L. Rosencher, B. Milleron.
Une étiologie inhabituelle d’hémoptysie.
Rev. Pneumol. Clin., 49 (1993), pp. 43-44
[50.]
N.N. Cálix, E.P. Rodríguez, C.V. Ciordia, et al.
Osteochodroplastic tracheopathy. Diagnostic confirmation: experience with 8 cases.
Rev Clin Esp, 184 (1989), pp. 12-15
[51.]
J. Castella, M.C. Puzo.
Traqueopatía osteocondroplástica.
Broncología, pp. 162-163
[52.]
L.L. Gleich, E.E. Rebeiz, M.M. Pankratov, et al.
The holmium: YAG laser-assisted otolaryngologic procedures.
Arch Otolaryngol Head Neck Surg, 121 (1995), pp. 1162-1166
[53.]
M.U. Akyol, A.A. Martin, N. Dhurandhar, et al.
Trachebronchopathia osteochondroplastica: a case report and a review of the literature.
Ear, Nose, Throat Journal, 72 (1993), pp. 347-350
[54.]
A.R. Molloy, J.N. Mcmahon.
Rapid progression of tracheal stenosis associated with tracheopathia osteochondroplastica.
Intensive Care Med, 15 (1988), pp. 60-62
[55.]
D.T. Carr, A.M. Olsen.
Tracheopathia osteochondroplastica.
JAMA, 155 (1954), pp. 1563-1565
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