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一名朗格汉斯细胞组织细胞增生症患者延迟诊断后的独特生长模式。

Distinctive growth pattern in a patient with a delayed diagnosis of Langerhans' cell histiocytosis.

机构信息

Department of Endocrinology, 424 Military Hospital, 4 Soulini st, 566 25, Sykies, Thessaloniki, Greece.

出版信息

Pituitary. 2012 Dec;15 Suppl 1:S28-32. doi: 10.1007/s11102-011-0291-6.

DOI:10.1007/s11102-011-0291-6
PMID:21279449
Abstract

We present a 22-year old male patient previously treated with radiotherapy and surgery at the age of 7 for an undefined suprachiasmatic mass. Following treatment he gradually became morbidly obese and besides subsequent panhypopituitarism he achieved his target height probably due to obesity-induced severe hyperinsulinemia. At the age of 21 Langerhans' cell histiocytosis was diagnosed at the right mandible and was surgically treated. One year later he developed a further painful osteolytic hip lesion and a single zoledronate infusion eliminated all symptoms. We highlight the importance of obtaining a histological diagnosis before initiating treatment, and the distinctive course of the disease in a patient who continued to growth besides GH deficiency.

摘要

我们报告 1 例 22 岁男性患者,7 岁时因下丘脑不明性质的肿瘤接受过放疗和手术治疗。此后,患者逐渐出现病态肥胖,且除了垂体功能减退症外,还由于肥胖引起的严重高胰岛素血症而达到了他的目标身高。21 岁时,于右侧下颌骨诊断为朗格汉斯细胞组织细胞增生症,并进行了手术治疗。1 年后,患者出现另一个疼痛性溶骨性髋关节病变,单次唑来膦酸输注消除了所有症状。我们强调在开始治疗前获得组织学诊断的重要性,以及在 GH 缺乏症患者中继续生长的情况下疾病的独特病程。

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本文引用的文献

1
Zoledronic acid in treatment of bone lesions by Langerhans cell histiocytosis.唑来膦酸治疗朗格汉斯细胞组织细胞增多症的骨病变
J Bone Miner Metab. 2009;27(1):110-3. doi: 10.1007/s00774-008-0001-2. Epub 2008 Nov 19.
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Bisphosphonate treatment of benign multifocal and unifocal osteolytic tumours of bone.双膦酸盐治疗骨的良性多灶性和单灶性溶骨性肿瘤。
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Endocrine manifestations in Langerhans cell histiocytosis.朗格汉斯细胞组织细胞增多症的内分泌表现。
Trends Endocrinol Metab. 2007 Aug;18(6):252-7. doi: 10.1016/j.tem.2007.06.003. Epub 2007 Jun 27.
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Growth without growth hormone (GH): a case report.无生长激素情况下的生长:一例报告
Hormones (Athens). 2004 Oct-Dec;3(4):259-65. doi: 10.14310/horm.2002.11135.
5
Evolving radiological features of hypothalamo-pituitary lesions in adult patients with Langerhans cell histiocytosis (LCH).成年朗格汉斯细胞组织细胞增多症(LCH)患者下丘脑 - 垂体病变的影像学特征演变
Neuroradiology. 2006 Jan;48(1):37-44. doi: 10.1007/s00234-005-0011-x. Epub 2005 Nov 15.
6
Incidence of growth hormone deficiency in pediatric-onset Langerhans cell histiocytosis: efficacy and safety of growth hormone treatment.儿童期朗格汉斯细胞组织细胞增多症中生长激素缺乏症的发病率:生长激素治疗的疗效和安全性。
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Bisphosphonate induces remission of refractory osteolysis in langerhans cell histiocytosis.
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More on pamidronate in Langerhans'-cell histiocytosis.
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More on pamidronate in Langerhans'-cell histiocytosis.
N Engl J Med. 2001 Nov 15;345(20):1502-3.
10
Pamidronate for bone pain from osteolytic lesions in Langerhans'-cell histiocytosis.帕米膦酸盐治疗朗格汉斯细胞组织细胞增生症溶骨性病变所致骨痛。
N Engl J Med. 2001 Jul 19;345(3):225. doi: 10.1056/NEJM200107193450318.