口腔顎裂的長期影響因類型而有不同


  【24drs.com】根據一項在挪威進行之近150萬人的人口基礎世代研究,在沒有任何其他先天性異常下,口腔顎裂新生兒的預後大部份是根據顎裂類型而定。
  
  根據線上發表於9月26日JAMA小兒科期刊的研究報告,在將近30歲時,出生時有唇裂者、不論有無顎裂,死亡風險並未大幅增加,就算有,死亡率也只比無此症的對照組小幅增加。
  
  不過,天生單純顎裂者就相當坎坷,醫療原因死亡風險是無此症之同齡者的3倍,他們在智力殘疾、腦癱和癲癇等狀況的風險也大幅升高。
  
  挪威Bergen大學全球公共衛生暨初級衛生保健系Erik Berg醫師等人寫道,研究結果對於單獨唇裂兒童的家長而言是好消息。相對的,從小對於[單純顎裂嬰幼兒]徹底檢查其他潛在疾病是強烈建議的,以確保必要的介入和儘早治療。
  
  作者們接著表示,他們進一步建議,這個資訊應提供給遺傳諮詢者、顎裂病童的家長、以及參與這些孩童之治療與追蹤的健康照護工作者。
  
  華盛頓大學小兒科、西雅圖兒童研究院臨床和轉化研究中心、西雅圖兒童醫院顱面中心的Carrie L. Heike醫師與Kelly N. Evans醫師在編輯評論中寫道,目前的人口基礎研究為強調連結大數據的潛力、確認相關終點提供了一個重要貢獻,且激勵持續進行研究,以進行最終目標是為了改善顎裂相關健康照護的質量研究。
  
  他們指出,探討治療和整體結果之關聯、以及從病患的角度整合資訊的進一步研究,將增加這類大規模、人口基礎之定量方法,並對成人結果有更全面之理解。
  
  這次的研究中,研究者比較了1967-1992年出生時、單純口腔顎裂的2,337名患者與1,413,819名沒有口腔顎裂者的存活與健康結果, 追蹤到2010年到他們18-43歲時。在顎裂組中, 695人只有唇裂、918人有唇裂和顎裂、724只有顎裂。
  
  Berg醫師等人報告指出,結果顯示,相對於對照組,只有唇裂者這組在青年時的發病率或死亡率病未顯著增加。出生時有唇裂和顎裂者,智力殘疾(相對風險[RR], 2.2;95%信賴區間[CI], 1.2 - 4.1)和腦癱(RR, 2.6;95% CI, 1.1 - 6.2)風險中度增加,死亡風險未增加。
  
  不過,只有顎裂者更可能死於醫療原因(風險比3.4;95% CI, 2.1 - 5.7),他們的智力殘疾(RR, 11.5;95% CI, 8.5 - 15.6)、焦慮異常(RR, 2.9;95% CI, 1.3 - 6.5)、自閉症(RR, 6.6;95% CI, 2.8 - 15.7)、嚴重學習障礙(RR, 10.6;95% CI, 5.5 - 20.2)、腦癱(RR, 4.8;95% CI, 2.3 - 10.0)、癲癇(RR, 4.9;95% CI, 2.2 - 10.8)以及肌肉或骨骼疾病(RR, 2.7;95% CI, 1.4 - 5.4)之風險增加。
  
  資料來源:http://www.24drs.com/
  
  Native link:Long-term Effect of Oral Cleft Depends on Type

Long-term Effect of Oral Cleft Depends on Type

By Susan London
Medscape Medical News

The prognosis for infants born with oral clefts in the absence of any other congenital anomalies largely depends on the type of cleft, according to a population-based cohort study conducted among nearly 1.5 million individuals in Norway.

At approximately 30 years of age, individuals born with a cleft lip, with or without cleft palate, did not have an elevated risk for death and had fairly small increases, if any, in morbidity compared with unaffected counterparts, according to results reported in an article published online September 26 in JAMA Pediatrics.

However, those born with cleft palate alone had a much rockier course, with about three times the risk for death from medical causes relative to that seen in unaffected peers. They also had sharply higher risks for conditions such as intellectual disability, cerebral palsy, and epilepsy.

"The present results are good news for parents of children with isolated cleft lip," write Erik Berg, MD, from the Department of Global Public Health and Primary Care, University of Bergen, Norway, and colleagues. In contrast, "[t]horough screening for other underlying conditions in [infants with cleft palate alone] is highly recommended from a young age to ensure necessary interventions and treatment as early as possible."

The authors continue, "This information should be provided to genetic counselors, parents of children with clefts, and health care workers involved in the treatment or follow-up of these children," they further recommend.

"The current population-based study provides an important contribution, highlights the potential for linking large data sets, identifies relevant end points, and motivates the continued work to conduct quality research with an ultimate goal to improve cleft-related health care," write Carrie L. Heike, MD, and Kelly N. Evans, MD, both from the Craniofacial Center at Seattle Children's Hospital, the Center of Clinical and Translational Research at Seattle Children's Research Institute, and the Department of Pediatrics at the University of Washington, in an accompanying editorial.

"Further investigations to explore the association between treatments and holistic outcomes and to incorporate information from patient perspectives will augment such large scale, population-based quantitative approaches and allow for a more holistic understanding of adult outcomes," they maintain.

For the current study, the investigators compared survival and health outcomes between 2337 individuals with an isolated oral cleft and 1,413,819 patients without an oral cleft born between 1967 and 1992 and followed through 2010, when they were between 18 and 43 years old. In the cleft group, 695 individuals had cleft lip only, 918 had both cleft lip and palate, and 724 had cleft palate only.

Results showed that relative to the unaffected individuals, the group with cleft lip only did not have any significant increase in morbidity or mortality by young adulthood, Dr Berg and colleagues report. Individuals born with both cleft lip and cleft palate had moderately elevated risks for intellectual disability (relative risk [RR], 2.2; 95% confidence interval [CI], 1.2 - 4.1) and cerebral palsy (RR, 2.6; 95% CI, 1.1 - 6.2), and no increase in the risk for death.

However, individuals having cleft palate only were much more likely to have died from medical causes (hazard ratio, 3.4; 95% CI, 2.1 - 5.7). They also had elevated risks for intellectual disability (RR, 11.5; 95% CI, 8.5 - 15.6), anxiety disorders (RR, 2.9; 95% CI, 1.3 - 6.5), autism spectrum disorders (RR, 6.6; 95% CI, 2.8 - 15.7), severe learning disabilities (RR, 10.6; 95% CI, 5.5 - 20.2), cerebral palsy (RR, 4.8; 95% CI, 2.3 - 10.0), epilepsy (RR, 4.9; 95% CI, 2.2 - 10.8), and muscular or skeletal disorders (RR, 2.7; 95% CI, 1.4 - 5.4).

The investigators and editorialists have disclosed no relevant financial relationships.

JAMA Pediatrics. Published online September 26, 2016.

    
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