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Titolo:
Pleuropulmonary blastoma: A rare pathology with an even rarer presentation
Autore:
Lallier, M; Bouchard, S; Di Lorenzo, M; Youssef, S; Blanchard, H; Lapierre, JG; Vischoff, D; Tucci, M; Brochu, P;
Indirizzi:
Hop St Justine, Div Pediat Gen Surg, Montreal, PQ H3T 1C5, Canada Hop St Justine Montreal PQ Canada H3T 1C5 g, Montreal, PQ H3T 1C5, Canada Hop St Justine, Dept Pediat, Montreal, PQ H3T 1C5, Canada Hop St Justine Montreal PQ Canada H3T 1C5 t, Montreal, PQ H3T 1C5, Canada Hop St Justine, Dept Anesthesia, Montreal, PQ H3T 1C5, Canada Hop St Justine Montreal PQ Canada H3T 1C5 a, Montreal, PQ H3T 1C5, Canada Hop St Justine, Dept Pathol, Montreal, PQ H3T 1C5, Canada Hop St Justine Montreal PQ Canada H3T 1C5 l, Montreal, PQ H3T 1C5, Canada
Titolo Testata:
JOURNAL OF PEDIATRIC SURGERY
fascicolo: 7, volume: 34, anno: 1999,
pagine: 1057 - 1059
SICI:
0022-3468(199907)34:7<1057:PBARPW>2.0.ZU;2-V
Fonte:
ISI
Lingua:
ENG
Soggetto:
PULMONARY BLASTOMA; CHILDHOOD;
Keywords:
pleuropulmonary blastoma; renal cysts; intestinal polyps;
Tipo documento:
Article
Natura:
Periodico
Settore Disciplinare:
Clinical Medicine
Citazioni:
10
Recensione:
Indirizzi per estratti:
Indirizzo: Di Lorenzo, M Hop St Justine, Div Pediat Gen Surg, 3175 Chemin Cote St Catherine, Montreal, PQ H3T 1C5, Canada Hop St Justine 3175 Chemin Cote St Catherine Montreal PQ Canada H3T 1C5
Citazione:
M. Lallier et al., "Pleuropulmonary blastoma: A rare pathology with an even rarer presentation", J PED SURG, 34(7), 1999, pp. 1057-1059

Abstract

Background: Pleuropulmonary blastoma is among the rarest tumors of childhood. Three types have been described: cystic, solid, and mixed. To date, bilateral disease has not been documented. Methods and Results: A 5-week-old girl presented with a history of fever. Chest x-ray showed bilateral diffuse cystic lesions. Bowel obstruction developed that required laparotomy. Multiple small bowel polyps were resected. The patient was readmitted 4 months later with deteriorating respiratory status. She underwent sequential thoracotomies for resection of multiple bullae under high-frequency oscillatory ventilation. Small bowel polypectomies were again required because of obstruction. Lung lesions were compatible with pulmonary blastoma but could not be correlated with intestinal polyposis. Bilateral cystic renal lesions were seen on ultrasound scan. Her disease progressed, despite chemotherapy, with the appearance of metastatic iris lesions. She again underwent laparotomies for multiple recurrent generalized small bower polyps that were causing obstruction. Expanding renal cysts affected kidney function, and she died at 14 months of age. Conclusions: The rare association between pleuropulmonary blastoma a nd Wilms' tumor or nephroblastomatosis is known but rarely reported. Lacking pathological evidence, we can only speculate that this was the case. We have been unable to demonstrate any histological association between the renopulmonary and digestive lesions. Despite many unanswered questions, we are likely dealing with a "syndrome" of sorts with a dire outcome, despite aggressive treatments. J Pediatr Surg 34: 1057-1059. Copyright (C) 1999 by W.B. Saunders Company.

ASDD Area Sistemi Dipartimentali e Documentali, Università di Bologna, Catalogo delle riviste ed altri periodici
Documento generato il 03/12/20 alle ore 15:57:43