PubMed · 15230265
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Robert Dinwiddie. 2004. Short case presentation. Pierre-Robin sequence.. https://pubmed.ncbi.nlm.nih.gov/15230265/
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A case of congenital lobar emphysema associated with pulmonary sequestration, presenting with respiratory distress in infancy, is reported. The lobar hyperinflation was managed by emergency lobectomy. The ipsilateral lower lobe affected with vascular sequestration was salvaged by therapeutic embolization, thus avoiding the long-term sequelae of pneumonectomy. The case is reported for its rarity.
We present an unusual case of Currarino syndrome with a mucosa-lined deep perineal fissure extending to the sacrum, penoscrotal transposition, perineal hypospadias, and a penile ventral skin defect. The child had a sigmoid diverting colostomy because of high anal atresia. Magnetic resonance imaging illustrated absence of the levator ani and muscle complex in the pelvis. At 15 months, perianal examination pointed out a fistula orifice and a sac related to the fistula at the left side of the perineal fissure. The fistula, a fluid-filled sac extending to the sacrum, and mucosa overlying the perineal fissure were removed en bloc. The neck of the sac was ligated and divided at the level of the distal sacrum. In the same session, a Glenn-Anderson procedure was performed for penoscrotal transposition, and the penile chordee was released. X-ray showed a bony deformity of the sacrococcygeal region in the shape of a scimitar. Histopathological examination demonstrated that the sac contained glial neuronal islands and nerve fibers. The boy has no neurologic deficits and seems to be well. To our knowledge, these associated malformations are extremely rare.
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