Congenital Adenomatoid Malformation of Lung with Pneumothorax

Adenomatoid hamartoma of the lung in a neonate

“…Of seven of the more recently reported cases (Ch'in and Tang, 1949;Jones, 1949;Thomas, 1949;Graham and Singleton, 1955;Bain, 1959;Goodyear and Shillitoe, 1959;Spector et al, 1960; and the present case), six presented with maternal hydramnios and in three of these infants anasarca was present. Six of the seven were premature, mostly by about 12 weeks.…”

“…The respiratory difficulty must in many cases be attributed to compression of the remaining normal lung, and not to the expanding lung syndrome described by Spector et al (1960), as in four of 11 cases where this symptom was noted there was no demonstrable bronchial communication with the malformation. Furthermore, the hamartoma must in many cases have attained a considerable size in utero to account for the presence of foetal anasarca and hypoplasia of the contralateral lung.…”

“…Ch 'in and Tang (1949), Thomas (1949), and Spector et al (1960) regarded the malformation as being related to congenital cystic disease of the lung, to which it does bear some structural resemblance. Simple lung cysts of apparently congenital origin are relatively common, but the adenomatoid hamartoma must be almost as frequent a cause of respiratory difficulty in the neonate.…”

“…The first description in English of an adenomatoid hamartoma was by Harris and Schattenberg (1942) who gave an account of two cases of single multicystic pulmonary Received for publication 11 September 1968. tumours in neonates. There have since been at least 18 further cases reported in English, including those of Ch'in and Tang (1949), Jones (1949), Thomas (1949), Graham andSingleton (1955), Craig, Kirkpatrick, andNeuhauser (1956), Gottschalk and Abramson (1957), Bain (1959), Goodyear and Shillitoe (1959), Spector, Claireaux, and Williams (1960), Kwittken and Reiner (1962), Nanson (1962), and Belanger, La Fl1che, and Picard (1964). Ch'in and Tang (1949), in their extensive review of the literature, noted that the first report of a case of a congenital adenomatoid malformation of the lung was by Stoerk (1897), and mentioned 10 other cases which were reported in the German literature between 1905and 1928.…”

Congenital Cystic Malformation of the Lung and Its Relation to Hydramnios

“…The authors accepted as true examples of this lesion ten earlier cases-all stillbirths or neonatal deaths-which were observed in various parts of central Europe and reported in German periodicals. Yet we found no further case reports in the German literature after 1939 although cases have been reported from Great Britain (Thomas, 1949;Goodyear and Shillitoe, 1959;Spector et al, 1960;Moffat, 1960;Moncrieff et al, 1969), the United States (Jones, 1949;Gottschalk and Abramson, 1957 ;Merenstein, 1969), Canada (Bain, 1959;Birdsell et al, 1966), Australia (Beischer et al, 1971), New Zealand (Dempster, 1969), France (Gerbeaux et al, 1968 and Holland (van Unnik et al, 1971 ;van Dijk and Wagenvoort, 1973). We believe that CCAML probably has a world-wide distribution and that the number reported is a poor guide to the true incidence.…”