Ileal-lymphoid-nodular hyperplasia, non-specific colitis, and pervasive developmental disorder in children (Retracted article. See vol 375, pg 445, 2010)

Ileal-lymphoid-nodular hyperplasia, non-specific colitis, and pervasive developmental disorder in children (Retracted article. See vol 375, pg 445, 2010)
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DOI:
10.1016/s0140-6736(97)11096-0
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发表时间:
1998-02-28
期刊:
影响因子:
168.9
通讯作者:
Walker-Smith, JA
Walker-Smith, JA
中科院分区:
医学1区
文献类型:
--
作者:
Wakefield, AJ;Murch, SH;Walker-Smith, JA

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研究背景:我们研究了一系列患有慢性小肠结肠炎和退行性发育障碍的儿童。方法12例儿童(平均年龄6岁[范围3-10岁],其中11例男孩)均有正常发育史,随后出现语言等后天技能丧失,并伴有腹泻和腹痛。儿童接受胃肠病学、神经学和发育评估,并回顾发育记录。在镇静状态下进行回肠结肠镜检查、活检、磁共振成像、脑电图和腰椎穿刺。在可能的情况下进行了钡透视。进行生化、血液学和免疫学检查。研究结果:12名儿童中有8名儿童接种麻疹、腮腺炎和风疹疫苗,1名儿童感染麻疹,1名儿童感染中耳炎,父母认为行为症状的发作与此相关。所有12名儿童均有肠道异常,从淋巴样结节增生到蚜虫溃疡。组织学显示11例患儿结肠呈斑片状慢性炎症,7例患儿反应性回肠淋巴样增生,但未见肉芽肿。行为障碍包括自闭症(9例)、分裂性精神病(1例)和可能的病毒或疫苗后脑炎(2例)。无局灶性神经异常,MRI和脑电图检查正常。与年龄匹配的对照组相比,异常的实验室结果是尿甲基丙二酸显著升高(p=0.003), 4名儿童血红蛋白低,4名儿童血清IgA低。我们在一组以前正常的儿童中发现了相关的胃肠道疾病和发育倒退,这通常与可能的环境触发因素有关。
Background We investigated a consecutive series of children with chronic enterocolitis and regressive developmental disorder.Methods 12 children (mean age 6 years [range 3-10], 11 boys) were referred to a paediatric gastroenterology unit with a history of normal development followed by loss of acquired skills, including language, together with diarrhoea and abdominal pain. Children underwent gastroenterological, neurological, and developmental assessment and review of developmental records. Ileocolonoscopy and biopsy sampling, magnetic-resonance imaging (MRI), electroencephalography (EEG), and lumbar puncture were done under sedation. Barium follow-through radiography was done where possible. Biochemical, haematological, and immunological profiles were examined.Findings Onset of behavioural symptoms was associated, by the parents, with measles, mumps, and rubella vaccination in eight of the 12 children, with measles infection in one child, and otitis media in another. All 12 children had intestinal abnormalities, ranging from lymphoid nodular hyperplasia to aphthoid ulceration. Histology showed patchy chronic inflammation in the colon in 11 children and reactive ileal lymphoid hyperplasia in seven, but no granulomas. Behavioural disorders included autism (nine), disintegrative psychosis (one), and possible postviral or vaccinal encephalitis (two). There were no focal neurological abnormalities and MRI and EEG tests were normal. Abnormal laboratory results were significantly raised urinary methylmalonic acid compared with age-matched controls (p=0.003), low haemoglobin in four children, and a low serum IgA in four children.Interpretation We identified associated gastrointestinal disease and developmental regression in a group of previously normal children, which was generally associated in time with possible environmental triggers.