Discussion
As reported in the literature, all cases but one have been right sided. Treatments have included antenatal steroids, fetal pneumonectomy, fetal thoracoamniotic shunt placement, observation and elective termination2-6. Only three of the ten reported cases survived to delivery but none survived past day one of life. One had undergone fetal thoracoamniotic shunt at 23 weeks GA, was delivered at 25 weeks GA3. Another had a pneumonectomy at 21 weeks GA and delivered at 24 weeks GA2. The third underwent fetal pneumonectomy at 26 weeks GA, and was delivered at 32 weeks GA5.
In all cases, the atretic MSB caused hyperexpansion of the ipsilateral lung, and compression of the contralateral lung in utero. Although fetal pneumonectomy theoretically allows the uninvolved lung time to grow and compensate, this did not occur likely secondary to prematurity. Here, the right lung distal to the atretic MSB was hyperexpanded but later collapsed (Figure 1A and 1B), with evidence of retained secretions and a normal bronchial branching pattern, allowing the left lung to function, albeit with significant mediastinal shift and consequent hemodynamic changes. We hypothesize that atresia at the mainstem level initially caused significant hyperexpansion on the right side, with mediastinal shift to the left, and a consequent developmental abnormality of the (short) LPA. At some point, the elevated pressure of lung fluid in the right bronchial tree presumably resulted in the formation of a fistulous connection, allowing decompression of the hyperexpanded right lung. The small fistula would have then collapsed under lower pressure and closed later in the 3rd trimester when the fetal lungs are not producing high volumes of fluid, leaving behind a 5 mm long fibrous attachment between the trachea and MSB. Postnatally, ventilation and expansion of the left lung further collapsed the right lung with worsening mediastinal shift. This exacerbated the stretching of the developmentally short LPA, with consequent narrowing of the stretched vessel and elevation of RVP. Slide tracheobronchoplasty allowed re-expansion of the right lung with mediastinal normalization, lessened the stretch and narrowing of the LPA, lowered the vascular resistance in the newly aerated lungs, with normalization of the RVP.
Slide tracheobronchoplasty for isolated bronchial pathology is a rare operation. The absence of bronchial cartilage causing a ball-valve effect and obstruction of the right MSB is the usual indication. This is a rare case of MBA successfully treated with slide tracheobronchoplasty following spontaneous resolution of fetal hydrops. Our management and surgical approach preserved his entire right lung and maximized the possibility of a long-term favorable outcome without the complications associated with pneumonectomy.Acknowledgement
We acknowledge Christine Schuler MD and Stephanie Merhar MD for their edits of the manuscript.