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The First of Congenital Cystic Adenomatoid Lung Malformation in Iraq: A Radiologic and Pathologic Study

Background: Congenital cystic adenomatoid lung malformation is a very rare condition characterized by replacement of normal pulmonary tissue with cysts of variable size and distribution. The condition is usually unilateral. Very few cases of bilateral and congenital cystic adenomatoid malformation with good outcome after resections of the lesions have been reported, and most cases were stillborn or died early during life. Patients and methods: A forty-day male infant presented with progressive respiratory distress since the first week of life observed at the Children Teaching Hospital of Baghdad Medical City was studied. The literature was reviewed with aim of describing the early documentation of the condition in the literature. Results: Clinically, the infant had significant respiratory distress interfering with feeding and evidence of shift of the mediastinum to the right. Chest X ray showed increased translucency of left lung field and mediastinal shit to the right. CT-scan showed large air-filled cyst in the left lung and small air-filled cyst in the right lung. The child was treated successfully by left upper lobecomy. The gross examination of two gray pieces of lung tissue showed on cut section microcysts that were apparent grossly. Microscopical examination showed intercommunicating cysts lined by cuboidal epithelium with gland like appearance confirming the diagnosis of congenital cystic adenomatoid malformation of the lung. Conclusion: A very rare case of bilateral congenital cystic adenomatoid malformation of the lung is reported and the early documentation of the condition in the literature is described.


Introduction
Congenital cystic adenomatoid lung malformation is a very rare condition characterized by replacement of normal pulmonary tissue with cysts of variable size and distribution. The condition is usually unilateral. The term "Congenital adenomatoid malformation of the lung" was first suggested by Ch'in and Tang in 1949. They reported a stillborn who had a large cystic mass with epithelial lining affecting the lower lobe of the left lung. Ch'in and Tang reviewed ten previously reported cases from the literature including the case of Stoerk (1897) [1,2]. In 1989, Beluffi et al from et al from Italy emphasized that congenital cystic adenomatoid malformation of the lung is one of the rarest causes of neonatal distress. They counted 405 previously reported cases in the literature, and reported an other sixteen cases, one with bilateral disease [3]. Giubergia et al (2012) from Buenos Aires, Argentina studied 172 cases of congenital cystic adenomatoid malformation, and found that the most common presenting symptoms of the disorder are respiratory distress in children under 6 months of age (40%) and recurrent pneumonia in older children. Lobectomy was the procedure of choice in the majority of patients [4].
Very few cases of bilateral and congenital cystic adenomatoid malformation with good outcome after resections of the lesions have been reported, and most cases were stillborn or died early during life [5,6,7,8].

Patients and methods
A forty-day male infant presented with progressive respiratory distress since the first week of life observed at the Children Teaching Hospital of Baghdad Medical City was studied. The literature was reviewed with aim of describing the early documentation of the condition in the literature.

Results
Clinically, the infant had significant respiratory distress interfering with feeding and evidence of shift of the mediastinum to the right. Chest X ray ( Figure-1) showed increased translucency of left lung field and mediastinal shit to the right. CT-scan showed large air-filled cyst in the left lung and small air-filled cyst in the right lung ( Figure-2A). The child was treated successfully by left upper lobecomy. The gross examination of two gray pieces of lung tissue showed on cut section microcysts that were apparent grossly. Microscopical examination showed intercommunicating cysts lined by cuboidal epithelium with gland like appearance confirming the diagnosis of congenital cystic adenomatoid malformation of the lung. Figure-3 shows the patient few days after surgery.

Discussion
Congenital cystic adenomatoid malformation of the lung is a very rare condition that was first reported in a stillborn female by Grawitz in 1880 [9]. Storek reported a second case of the disorder diagnosed in a male newborn necropsy in 1897 [2]. Couvelaire reported a third case in a sixday old term neonate in 1904 [10]. Thereafter, another 15 cases of congenital cystic adenomatoid malformation with necropsy diagnosis were reported including the reports of Von Graff (1905), Pappenheimer In 1943, Fischer, Tropea, and Bailey reported the 19 th case in a female term infant and it was first case to be diagnosed by surgical resection and not after death [16]. However, Ch'in and Tang (1949) [1], Thomas (1949) [17], and Potter (1952) reported another three cases diagnosed by necropsy [15].
Graham and Singleton (1955) reported a female term infant and treated by surgical resection at six weeks of age and survived [15]. Craig, Kirkpatrick, and Neuhauser (1956) reported four more infants, three of whom were successfully treated by lobectomy in the newborn period [18].

Conclusion
A very rare case of bilateral congenital cystic adenomatoid malformation of the lung is reported and the early documentation of the condition in the literature is described.

Acknowledgement
The author would like to express his gratitude for the parents of the patient who willingly accepted publishing his photo.