Antenatal sonographic findings of right pulmonary agenesis with ipsilateral microtia: a possible new laterality association

Maymon, Ron; Hegesh, Julius; Herman, Arie; Weinraub, Z.; Achiron, Reuven

doi:10.1002/1097-0223(200102)21:2<125::aid-pd999>3.0.co;2-c

Cited by 26 publications

(14 citation statements)

References 13 publications

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“…5 There has been a few reports of pulmonary agenesis and goldenhar syndrome in the literature. 6,7,8,9 When pulmonary agenesis is associated with Goldenhar syndrome, it is referred as an expanded Goldenhar complex. Most of the case reports have been of infants.…”

Section: Discussionmentioning

confidence: 70%

Goldenhar syndrome with left bronchopulmonary agenesis: A rare association

Ghimire

Thapa

Shrestha

et al. 2011

Journal of Institute of Medicine

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An eleven years female presented with recurrent chest infection since childhood with markedly diminished air entry on her right chest and mediastinal shift. She also had vague facial deformity with multiple auricular appendages. Computed tomography showed collapse of the left lung and on thoracotomy, complete agenesis of the left lung was noted. Goldenhar anomaly is rare and presents with variable degree of external anomaly. Its association with bronchopulmonary agenesis is extremely rare with few cases reported in the world literature. Features of other congenital should be sought for in a child with facial asymmetry, external auditory appendages, malformation of the, nose, soft palate, lip and mandible etc so that appropriate treatment can be administered.

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Section: Discussionmentioning

confidence: 70%

Goldenhar syndrome with left bronchopulmonary agenesis: A rare association

Ghimire

Thapa

Shrestha

et al. 2011

Journal of Institute of Medicine

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“…Among the prenatally diagnosed cases, the right‐to‐left relationship is around 5:1, which may reflect the idea that it is probably easier to diagnose right agenesis of the lung than left agenesis, especially because of the greater mediastinal shift and the higher incidence of other associated abnormalities. These are also some of the reasons that may explain the poorer prognosis of the right‐sided lesions, resulting in increased perinatal and infant death and at an earlier age 1 – 3 , 6 , 7 . In this review, as reported in the literature, right lung agenesis was less frequently isolated than the left‐sided type, but the difference did not reach significance ( P = .18), probably because of the small sample.…”

Section: Resultsmentioning

confidence: 99%

“…However, up to 50% of affected fetuses will have associated congenital abnormalities potentially adversely affecting the outcome 1 – 5 . When unilateral, right lung agenesis appears to carry a worse prognosis, resulting in increased perinatal and infant death and at an earlier age 1 – 3 , 6 , 7 . All of these aspects are of critical importance for parents when making difficult decisions in pregnancy.…”

mentioning

confidence: 99%

Prenatal Diagnosis of Isolated Right Pulmonary Agenesis Using Sonography Alone

Meller

Morris

Desai

et al. 2012

Journal of Ultrasound in Medicine

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Pulmonary agenesis is a rare congenital anomaly, estimated to complicate around 1 per 15,000 pregnancies, in which there is complete absence or severe hypoplasia of one or both lungs, frequently associated with other abnormalities. A prospective prenatal diagnosis is a challenge, and a substantial proportion of cases are diagnosed by fetal magnetic resonance imaging, postnatal computed tomography, or postmortem. Thus, there are only a few reported cases of prenatal diagnosis in the literature. We report the prenatal diagnosis of isolated right lung agenesis diagnosed with sonography alone at a relatively early gestational age. We also present a systematic review of the literature for this condition to accompany this case study.

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“…Three had right lung agenesis and one had left lung agenesis. Three of these four elected to undergo termination of pregnancy (TOP); in one case with right lung agenesis a laterality syndrome was suspected when microtia was also detected18. In the case with isolated left lung agenesis polydactyly was discovered post‐TOP.…”

Section: Resultsmentioning

confidence: 99%