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We present the first documented case of agnathia-holoprosencephaly (an uncommon form of craniofacial anomaly) associated with situs inversus. This case may represent the concordance of multiple field complex anomalies, but the possibility of a major midline malformation (situs inversus) caused by a timed insult (environmental or genetic) which affects multiple structures and occurs concurrently with a major field defect during early embryogenesis cannot be excluded.
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Leech et al. (1988) studied this question.
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