A rare clinical presentation of pulmonary hypoplasia

Figen Gülen, Sakine Kar, Levent Midyat, Esen Demir, Süphan Özyurt, Arif Ruhi Özyürek, Remziye Tanaç, Naim Ceylan
DOI: 10.4236/ojped.2011.14018                

Abstract

Pulmonary hypoplasia is a rare congenital anomaly, which is frequently associated with other congenital anomalies. Clinical symptoms vary depending on the other system anomalies and severity of pulmonary hypoplasia. Although it is usually diagnosed in infancy and childhood, some cases do not show any symptoms until the adolescent ages. In adolescents and adults with unilateral hypolucent lung, although it is seen rarely, pulmonary hypoplasia should always be kept in mind. In this article, we present a case with pulmonary hypoplasia who remained asymptomatic until puberty.

Keywords

Lung; Unilateral; Hypoplasia; Adolescent

Conflicts of Interest

The authors declare no conflicts of interest.

References

[1]	Kant, S. (2007) Unilateral pulmonary hypoplasia—A case report. Lung India, 24, 69-71. doi:10.4103/0970-2113.44215
[2]	Hislop, A., Hey, E. and Reid, L. (1979) The lungs in congenital bilateral renal agenesis and dysplasia. Archives of Disease in Childhood, 54, 32-38. doi:10.1136/adc.54.1.32
[3]	Areechon, W. and Reid, L. (1963) Hypoplasia of lung with congenital diaphragmatic hernia. British Medical Journal, 1, 230-233. doi:10.1136/bmj.1.5325.230
[4]	Johnson, R.J. and Haworth, S.G. (1982) Pulmonary vascular and alveolar development in tetralogy of fallot: A recommendation for early correction. Thorax, 37, 893- 901. doi:10.1136/thx.37.12.893
[5]	Cooney, T.P. and Thurlbeck, W.M. (1985) Lung growth and development in anencephly and hydranencephaly. The American Review of Respiratory Disease, 132, 596- 601.
[6]	Cooney, T.P. and Thurlbeck, W.M. (1982) Pulmonary hypoplasia in Down syndrome. New England Journal of Medicine, 307, 1170-1173. doi:10.1056/NEJM198211043071902
[7]	Deniz, O., Tozkoparan, E., Ciftci, F., Ozkan, M., et al. (2004) Sol pulmoner aplazi (agenezi) (olgu sunumu). Gülhane Tip Dergisi, 46, 56-58.
[8]	Fraser, R.S., et al. (Eds.) (1994) Pulmonary abnormalities of developmental origin. Synopsis of diseases of the chest, Second Edition, W. B. Saunders Company, Phildelphia, 256-286.
[9]	Naclerio, E.A. (1959) Congenital diseases, agenesis of lung. Bronchopulmonary Diseases, 35, 309-313.
[10]	Roque, A.S., Burton, E.M., Boedy, R.F., Falls, G., et al. (1997) Unilateral pulmonary agenesis without mediastinal displacement. Southern Medical Journal, 90, 335- 337.
[11]	Odd, D.E., Batin, M.R., Halam, L. and Knight, D.B. (2003) Primary pulmonary hypoplasia: A case report and review of the literature. Journal of Paediatrics and Child Health, 39, 467-469. doi:10.1046/j.1440-1754.2003.00191.x
[12]	Monaldi, V. (1960) Malformative bronchopulmonary diseases caused by anatomical defects. Minerva Medica, 51, 3474-3478.
[13]	Boyden, E.A. (1955) Developmental anomalies of the lungs. American Journal of Surgery, 89, 79-89. doi:10.1016/0002-9610(55)90510-9
[14]	Haddad, G.G. and Fontan, J.J.P. (2000) Development of the respiratory system. Nelson Textbook of Pediatrics, 16th Edition, 1235-1237.
[15]	Swischuk, L.E., Richardson, C.J., Nichols, M.M. and Ingman, M.J. (1979) Primary pulmonary hypoplasia in the neonate. Journal of Pediatric, 95, 573-577. doi:10.1016/S0022-3476(79)80773-8
[16]	Sunam, G. and Ceran, S. (2009) Pulmonary artery agenesis and kung hypoplasia. European Journal of General Medicine, 6, 265-267.
[17]	Mendelsohn, G. and Hutchins, G.M. (1971) Primary pulmonary hypoplasia. Report of a case with polyhydramnios. American Journal of Diseases of Children, 131, 1220-1223.
[18]	Frey, B., Fleishauer, A. and Gersbach, M. (1994) Familial isolated pulmonary hypoplasia: A case report, suggesting autosomal recessive inheritance. European Journal of Pediatrics, 153, 460-463. doi:10.1007/BF01983413
[19]	Hamel, B.C.J. (1995) Familial primary pulmonary hypoplasia. European Journal of Pediatrics, 154, 336. doi:10.1007/BF01957378
[20]	Askenazi, S.S. and Perlman, M. (1979) Pulmonary hypoplasia: Lung weight and radial alveolar count as criteria of diagnosis. Archives of Disease in Childhood, 54, 614-618. doi:10.1136/adc.54.8.614
[21]	Langer, R. and Kaufmann, H.J. (1986) Primary (isolated) bilateral pulmonary hypoplasia: A comparative study of radiologic findings and autopsy results. Pediatric Radiology, 16, 175-179. doi:10.1007/BF02456280
[22]	Laudy, J.A., Tibboel, D., Robben, S.G., de Krijger, R.R., et al. (2002) Prenatal prediction of pulmonary hypoplasia. Clinical, biometric, and doppler velocity correlates. Pediatrics, 109, 250-258. doi:10.1542/peds.109.2.250
[23]	Chang, N., Hertzler, J.H., Gregg, R.H., Lofti, M.W., et al. (1968) Congenital stenosis of the right mainstem bronchus. A case report. Pediatrics, 41, 739-742.
[24]	Goldstein, J.D. and Reid, L.M. (1980) Pulmonary hypoplasia resulting from phrenic nerve agenesis and diaphragmatic amyoplasia. Journal of Pediatric, 97, 282- 287. doi:10.1016/S0022-3476(80)80496-3
[25]	Chin, T. (2009) Pulmonary hypoplasia. Cited March 2009. http://www.emedicine.com/ped/TOPIC2627.HTM