Rizwan Qureshi.
Synchronous Bilateral Pneumothorax in Pregnant Mother and Newborn Baby - Genetic Component to Etiology?.
J Coll Physicians Surg Pak Jan ;10(11):433-4.

An unusual case is reported here that presented severe respiratory failure due to pneumothorax, just after birth, in a healthy female baby whose mother was a known case of recurrent, bilateral, secondary spontaneous pneumothorax. It is postulated that a genetic etiology may be responsible for this rare coincidence. In this unusual scenario, each entity should be managed on it`s own merit.

CASE REPORT: A 27-year-old female, primigravida of 30 weeks presented with recurrent bilateral secondary pneumothoraces (known as cystic lung disease).On arrival she looked quite healthy, her blood pressure was 130/80, pulse rate was 85/min, her respiration was 25/min and temperature was 36.8°C. Clinical assessment revealed decreased air entry, hyperresonant percussion note and vesicular breathing bilaterally. Initial laboratory investigations comprising of CBC, U&E`s and LFT`s were unremarkable. ABG`s revealed an arterial pH of 7.4, a Pa02 of a 12.6kPa, a PaC02 of 5kPa and a bicarbonate concentration of 24mmol/L. A sputum gram stain and Ziehl-Neelsen`s method did not show any organism. Bilateral tube thoracostomy was carried out while any surgical intervention was postponed till her delivery. Apart from her complicated pneumothorax, she had experienced an uncomplicated pregnancy, no documented oligohydramniosor polyhydramnios, and had taken no medication during the course of her pregnancy. Six weeks later, she had elective caesarian section and gave birth to 36 weeks, pre-term female baby who weighed 2.78 kg. Approximately 90 minutes after birth, the neonate developed respiratory distress. Baby was tachypnoeic and demonstrated an obstructive respiratory pattern with nasal flaring and intercostal recession. Respiration was described as grunting, Apgar score was 9 at 1/minute and 9 at 5 minutes. Pulse oximetery showed Sa02 81% on air. She was immediately transferred to special care baby unit and subsequently was placed in an incubator with 23-35% 02 where her SaO2 improved to > 90%. Due to pre-term birth and low body weight pressure support was avoided. Clinical examination suggested reduced air entry to left side and soft systolic murmur at left upper sternal edge while chest x-ray revealed massive left sided pneumothorax with shifting of mediastinum to right side. Initially pleural aspiration with 21g butterfly needle at 2nd intercostal space in mid clavicle line was carried out, however, her chest was still hyperresonant, respiratory rate was 55-60 and Sa02 had increased to 35% to maintain her oxygenation, hence chest drain was inserted at second intercostal space in anterior axillery line. Murmurs were cautiously monitored which were causing no hemodynamical compromise and necessitated no further investigation. Repeat CXR showed resolution of left sided pneumothorax but strangely revealed right sided apical pneumothorax without any respiratory compromise, hence, was observed conservatively. On day 3, repeat CXR suggested expansion of both lungs with no evidence of right apical pneumothorax which had resolved without any intervention. At day 4, cranial and renal ultrasound scans were carried out to rule out any associated congenital abnormalities which were reported normal. Baby improved remarkably in following days, no further respiratory distress was witnessed and her Sa02 remained > 90% on air. Therefore, on 8th day of her life she was sent home with parents in view of her satisfactory health. She was reviewed in out-patient clinic with her parents after 2 months (8 weeks) with good health. Clinical as well as radiological assessment was remarkably good. Motherhad elective bilateral video-assisted thoracoscopic (VATS) pleurectomy, pleural abrasion for recurrent bilateral secondary pneumothoraces. Mother made uneventful postoperative recovery and was sent home within two weeks of surgery with both lungs up.

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