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生长激素分泌型垂体大腺瘤(肢端肥大症)伴进行性牙列不齐及持续气道正压通气治疗无效

GH-secreting pituitary macroadenoma (acromegaly) associated with progressive dental malocclusion and refractory CPAP treatment.

作者信息

Miranda-Rius Jaume, Brunet-LLobet Lluís, Lahor-Soler Eduard, de Dios-Miranda David, Giménez-Rubio Josep Anton

机构信息

Department of Odontostomatology, Faculty of Medicine & Health Sciences, Universitat de Barcelona, Feixa Llarga, s/n, L'Hospitalet de Llobregat, 08907, Barcelona, Spain.

Division of Orthodontics and Paediatric Dentistry, Hospital Sant Joan de Déu, Universitat de Barcelona, Barcelona, Spain.

出版信息

Head Face Med. 2017 May 10;13(1):7. doi: 10.1186/s13005-017-0140-6.

DOI:10.1186/s13005-017-0140-6
PMID:28490347
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5424328/
Abstract

BACKGROUND

A link between progressive dental malocclusion, the use of a continuous positive airway pressure mask and GH-secreting pituitary macroadenoma (acromegaly) has not been previously reported. The present clinicopathological analysis stresses that tooth malposition should not be seen exclusively as a local process.

CASE PRESENTATION

A 62-year-old caucasian man with no relevant medical history reported difficulty chewing food and perceived voice alteration during his annual periodontal check-up. He also referred stiffness of the tongue, face, and submandibular area. The patient had been diagnosed with obstructive sleep apnea syndrome two years previously, since when he had worn a continuous positive airway pressure device during sleep. Exploration of the occlusion revealed significant changes: an atypical left lateral and anterior open bite with major buccoversion of teeth 33, 34, 35, 36. Inspection of the soft tissue revealed only macroglossia, although external palpation indicated a subcutaneous stiffness of the submandibular area. General analytical tests, including hormone profiles, and magnetic resonance imaging confirmed the diagnosis of acromegaly induced by a pituitary adenoma. Intrasellar tumor resection via transsphenoidal approach was performed. After surgery, the patient already noted a marked improvement of all symptoms associated with the acromegaly. Desaturation data also evolved favourably and the pulmonologist advised the patient to abandon the continuous positive airway pressure treatment.

CONCLUSION

Progressive dental malocclusion may be associated with a systemic disease and the use of a nasal mask with premaxillary support may distort the diagnosis of acromegaly.

摘要

背景

此前尚未报道过进展性牙颌面畸形、持续气道正压通气面罩的使用与生长激素分泌型垂体大腺瘤(肢端肥大症)之间的联系。本临床病理分析强调,牙齿错位不应仅被视为局部过程。

病例介绍

一名62岁的白种男性,无相关病史,在年度牙周检查时报告咀嚼食物困难并感觉声音改变。他还提到舌头、面部和下颌下区域僵硬。该患者两年前被诊断为阻塞性睡眠呼吸暂停综合征,自那时起他在睡眠期间一直佩戴持续气道正压通气设备。咬合检查发现显著变化:非典型的左侧及前方开颌,33、34、35、36号牙严重颊向错位。软组织检查仅发现巨舌症,尽管外部触诊显示下颌下区域皮下僵硬。包括激素水平检测在内的常规分析检查以及磁共振成像确诊为垂体腺瘤所致肢端肥大症。通过经蝶窦入路进行了鞍内肿瘤切除术。术后,患者已注意到与肢端肥大症相关的所有症状有明显改善。血氧饱和度数据也向好发展,肺科医生建议患者停止持续气道正压通气治疗。

结论

进展性牙颌面畸形可能与全身性疾病有关,使用带有上颌前部支撑的鼻面罩可能会使肢端肥大症的诊断出现偏差。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/057f/5424328/150e758a59de/13005_2017_140_Fig7_HTML.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/057f/5424328/b25cec5f8c82/13005_2017_140_Fig5_HTML.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/057f/5424328/150e758a59de/13005_2017_140_Fig7_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/057f/5424328/73029e59957f/13005_2017_140_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/057f/5424328/46d40274db8b/13005_2017_140_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/057f/5424328/1e48e6316f5a/13005_2017_140_Fig3_HTML.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/057f/5424328/b25cec5f8c82/13005_2017_140_Fig5_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/057f/5424328/d6d46b61898d/13005_2017_140_Fig6_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/057f/5424328/150e758a59de/13005_2017_140_Fig7_HTML.jpg

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