以顽固性腹泻为主要症状的神经母细胞瘤2例报告并文献复习
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摘要
目的总结以顽固性腹泻为主要症状的儿童神经母细胞瘤(NB)的临床特点。方法回顾分析2例以顽固性腹泻为主要症状的神经母细胞瘤患儿的临床资料,并进行文献复习。结果 2例患儿均为女性、1岁余,均有顽固性腹泻及低钾血症。例2为经术后病理确诊的神经母细胞瘤,在化疗过程中出现反复腹泻;例1以腹泻发病,且为最明显症状,伴电解质紊乱、腹胀、体质量减轻等,腹部可触及包块,经影像学及病理检查确诊为神经母细胞瘤。2例患儿均为节细胞型神经母细胞瘤,手术联合化疗后症状缓解,现均无病生存。结论以顽固性腹泻为主要症状的神经母细胞瘤比较罕见,目前尚无标准诊断及治疗方案,经过手术及化疗联合治疗效果较好。
Objective To summarize the clinical characteristics of neuroblastoma(NB) with refractory diarrhea as the main symptom in children. Method The clinical data of neuroblastoma with refractory diarrhea as the main symptoms in 2 children were retrospectively analyzed and the literature was reviewed. Results Both cases of neuroblastoma were female and over one year old and suffered from refractory diarrhea and hypokalemia. In case 1 neuroblastoma was confirmed by postoperative pathology and recurrent diarrhea occurred during chemotherapy. In case 2 onset symptom was diarrhea which was also most obvious symptom. She had electrolyte disturbance, abdominal distension and weight loss. Palpable abdominal mass was observed in the child. Neuroblastoma was diagnosed by imaging and pathological examination. Both cases were ganglion cell type NB, and their symptoms were relieved after surgery combined with chemotherapy. Both survived disease free.Conclusion Neuroblastoma with refractory diarrhea as the main symptom is rare. There is no standard diagnosis and treatment.The combination of surgery and chemotherapy is effective.
Objective To summarize the clinical characteristics of neuroblastoma(NB) with refractory diarrhea as the main symptom in children. Method The clinical data of neuroblastoma with refractory diarrhea as the main symptoms in 2 children were retrospectively analyzed and the literature was reviewed. Results Both cases of neuroblastoma were female and over one year old and suffered from refractory diarrhea and hypokalemia. In case 1 neuroblastoma was confirmed by postoperative pathology and recurrent diarrhea occurred during chemotherapy. In case 2 onset symptom was diarrhea which was also most obvious symptom. She had electrolyte disturbance, abdominal distension and weight loss. Palpable abdominal mass was observed in the child. Neuroblastoma was diagnosed by imaging and pathological examination. Both cases were ganglion cell type NB, and their symptoms were relieved after surgery combined with chemotherapy. Both survived disease free.Conclusion Neuroblastoma with refractory diarrhea as the main symptom is rare. There is no standard diagnosis and treatment.The combination of surgery and chemotherapy is effective.
引文
[1]Bourdeaut F,de Carli E,Timsit S,et al.VIP hypersecretion as primary or secondary syndrome in neuroblastoma:a retrospective study by the Societe Francaise des Cancers de l'Enfant(SFCE)[J].Pediatr Blood Cancer,2009,52(5):585-590.
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[10]Wildhaber B,Niggli F,Bergstrasser E,et al.Paraneoplastic syndromes in ganglioneuroblastoma:contrasting symptoms of constipation and diarrhoea[J].Eur J Pediatr,2003,162(7-8):511-513.
[11]Reindl T,Degenhardt P,Luck W,et al.The VIP-secreting tumor as a differential diagnosis of protracted diarrhea in pediatrics[J].Klin Padiatr,2004,216(5):264-269.
[12]Zhang YT,Feng LH,Zhang Z,et al.Different kinds of paraneoplastic syndromes in childhood neuroblastoma[J].Iran J Pediatr,2015,25(1):e266.
[13]Gesundheit B,Smith CR,Gerstle JT,et al.Ataxia and secretory diarrhea:two unusual paraneoplastic syndromes occurring concurrently in the same patient with ganglioneuroblastoma[J].J Pediatr Hematol Oncol,2004,26(9):549-552.
[14]王玉环,黄瑛,陈莲,等.以腹泻为主要临床表现的肿瘤性疾病2例[J].中国循证儿科杂志,2008,3(1):74-76.
[15]Czkwianianc E,Zalewska-Szewczyk B,Kobos J,et al.Uncommon reasons of the digestive tract-related paraneoplastic syndromes in children with neuroblastic tumors:three case reports[J].Contemp Oncol(Pozn),2018,22(1):42-46.
[16]Matthay KK,Maris JM,Schleiermacher G,et al.Neuroblastoma[J].Nat Rev Dis Primers,2016,2:16078.
[17]熊复,叶飘,张勇,等.以慢性腹泻为主要表现的儿童节细胞神经母细胞瘤1例[J].儿科药学杂志,2015,21(03):1.
[18]Stallings RL,Foley NH,Bray IM,et al.MicroRNA and DNA methylation alterations mediating retinoic acid induced neuroblastoma cell differentiation[J].Semin Cancer Biol,2011,21(4):283-290.
[19]Chevrier L,Meunier AC,Cochaud S,et al.Vasoactive intestinal peptide decreases MYCN expression and synergizes with retinoic acid in a human MYCN-amplified neuroblastoma cell line[J].Int J Oncol,2008,33(5):1081-1089.
[20]Colon NC,Chung DH.Neuroblastoma[J].Adv Pediatr,2011,58(1):297-311.
[2]Tomolonis JA,Agarwal S,Shohet JM.Neuroblastoma pathogenesis:deregulation of embryonic neural crest development[J].Cell Tissue Res,2018,372(2):245-262.
[3]Park J R,Eggert A,Caron H.Neuroblastoma:biology,prognosis,and treatment[J].Hematol Oncol Clin North Am,2010,24(1):65-86.
[4]Han W,Wang HM.Refractory diarrhea:a paraneoplastic syndrome of neuroblastoma[J].World J Gastroenterol,2015,21(25):7929-7932.
[5]Kanik A,Baran M,Cayan O,et al.Vasoactive intestinal peptide releasing tumor which caused to chronic watery diarrhea and hypokalemia[J].Turk Pediatri Ars,2014,49(2):160-162.
[6]Moody TW,Nuche-Berenguer B,Jensen RT.Vasoactive intestinal peptide/pituitary adenylate cyclase activating polypeptide,and their receptors and cancer[J].Curr Opin Endocrinol Diabetes Obes,2016,23(1):38-47.
[7]Husain K,Thomas E,Demerdash Z,et al.Mediastinal ganglioneuroblastoma-secreting vasoactive intestinal peptide causing secretory diarrhoea[J].Arab J Gastroenterol,2011,12(2):106-108.
[8]Zhang WQ,Liu JF,Zhao J,et al.Tumor with watery diarrhoea,hypokalaemia in a 3-year-old girl[J].Eur J Pediatr,2009,168(7):859-862.
[9]Sintusek P,Thammarakcharoen T,Shuangshoti S,et al.Unremitting watery diarrhoea in early childhood period[J].BMJ Case Rep,2017,pii:bcr-2016-217532.doi:10.1136/bcr-2016-217532.
[10]Wildhaber B,Niggli F,Bergstrasser E,et al.Paraneoplastic syndromes in ganglioneuroblastoma:contrasting symptoms of constipation and diarrhoea[J].Eur J Pediatr,2003,162(7-8):511-513.
[11]Reindl T,Degenhardt P,Luck W,et al.The VIP-secreting tumor as a differential diagnosis of protracted diarrhea in pediatrics[J].Klin Padiatr,2004,216(5):264-269.
[12]Zhang YT,Feng LH,Zhang Z,et al.Different kinds of paraneoplastic syndromes in childhood neuroblastoma[J].Iran J Pediatr,2015,25(1):e266.
[13]Gesundheit B,Smith CR,Gerstle JT,et al.Ataxia and secretory diarrhea:two unusual paraneoplastic syndromes occurring concurrently in the same patient with ganglioneuroblastoma[J].J Pediatr Hematol Oncol,2004,26(9):549-552.
[14]王玉环,黄瑛,陈莲,等.以腹泻为主要临床表现的肿瘤性疾病2例[J].中国循证儿科杂志,2008,3(1):74-76.
[15]Czkwianianc E,Zalewska-Szewczyk B,Kobos J,et al.Uncommon reasons of the digestive tract-related paraneoplastic syndromes in children with neuroblastic tumors:three case reports[J].Contemp Oncol(Pozn),2018,22(1):42-46.
[16]Matthay KK,Maris JM,Schleiermacher G,et al.Neuroblastoma[J].Nat Rev Dis Primers,2016,2:16078.
[17]熊复,叶飘,张勇,等.以慢性腹泻为主要表现的儿童节细胞神经母细胞瘤1例[J].儿科药学杂志,2015,21(03):1.
[18]Stallings RL,Foley NH,Bray IM,et al.MicroRNA and DNA methylation alterations mediating retinoic acid induced neuroblastoma cell differentiation[J].Semin Cancer Biol,2011,21(4):283-290.
[19]Chevrier L,Meunier AC,Cochaud S,et al.Vasoactive intestinal peptide decreases MYCN expression and synergizes with retinoic acid in a human MYCN-amplified neuroblastoma cell line[J].Int J Oncol,2008,33(5):1081-1089.
[20]Colon NC,Chung DH.Neuroblastoma[J].Adv Pediatr,2011,58(1):297-311.
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