• 제목/요약/키워드: birth defects

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분자유전학적으로 진단된 가부키 증후군 1례 (A Case of Kabuki Syndrome Confirmed by Genetic Analysis: A Novel Frameshift Mutation in the KMT2D Gene)

  • 박수진;안문배;장우리;조원경;채효진;김명신;서병규
    • 대한유전성대사질환학회지
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    • 제17권3호
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    • pp.103-108
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    • 2017
  • 가부키 증후군(Kabuki syndrome)은 특징적인 얼굴(아치형의 넓은 눈썹, 낮은 코끝, 큰 귓바퀴, 아래 외측 안검 외전), 골격계 이상, 출생 후 성장 지연, 그리고 경도의 정신 지체를 특징으로 나타내는 선천성 이상 질환이다. 또한, 선천성 심장기형, 비뇨생식기 기형, 구개구순열을 포함한 위장관 기형, 안검 하수 등의 증상이 동반되기도 한다. 감염과 면역질환, 경련, 내분비 문제, 청력 손실이 나타날 수 있다. 가부키 증후군의 대부분은 12번 염색체의 장완에 위치한 KMT2D 유전자의 돌연변이에 의해 발병된다. KMT2D 유전자의 돌연변이로 생성된 비정상적인 KMT2D 단백질로 인해 신체의 여러 장기와 조직의 특정 유전자의 활성이 정상적으로 이뤄지지 못하여, 결과적으로 가부키 증후군의 특징적인 외형과 기능장애가 나타난다. 출생시 선천성 심장 기형, 갑상선 기능 저하 및 일과성 저혈당 과거력이 있던 환아가 만 4세경 성장 지연, 아치형 눈썹, 낮은 코끝, 아래 외측 안검 외전, 큰 귓바퀴 등의 얼굴 생김새를 보여 시행한 분자유전학적 검사에서 KMT2D의 exon 15에서 새로운 frameshift mutation이 발견되어 증례를 보고하는 바이다. 가부키 증후군은 성장 지연 뿐 아니라, 발달 장애 및 행동 장애, 사춘기 여아의 경우 조기 가슴 발육을 포함한 내분비적 문제, 사시, 안검하수 등의 안과적 문제, 만성 중이염, 청력 손실, 감염, 경련 등 다양한 증상을 나타낼 수 있기 때문에 조기에 질환을 진단하고, 임상증상에 따라 적절한 중재를 할 수 있도록, 정기적으로 신장, 몸무게, 머리둘레, 발달상태, 청각과 시각상태를 확인하는 것이 중요하다. 따라서 여러 장기의 선천 기형을 보이는 환아의 경우, 임상의는 출생 당시에는 두드러지지 않을 수 있는 특징적인 얼굴 생김새, 성장 지연, 정신 지체 등의 이상에 대해 유의하여야 한다. 분자유전학적 검사는 임상적으로 가부키 증후군이 의심되는 환아를 조기에 진단할 수 유용한 방법이며, 앞으로도 가부키 증후군과 연관된 유전자 변이에 대한 연구가 더욱 필요할 것으로 사료된다.

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분만성 상완 신경총 손상, 선천성근성사경 및 구개열의 병발 (Concurrence of Obstetric Brachial Plexus Injury, Congenital Muscular Torticollis and Cleft Palate)

  • 이한별;박명철;김치선;한재덕;이승재;김세온;임신영
    • Journal of Genetic Medicine
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    • 제8권1호
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    • pp.71-75
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    • 2011
  • 임신성 당뇨 및 조기양막파수를 보인 산모의, 다른 산전 병력 없이 과체중아로 태어난 생후 17일된 남아에서, 분만성 상완 신경총 손상, 선천성근성사경 및 구개열의 병발이 진단 되었다. 동시에 세 질환의 병발이 단순 병발일 수 있으나, 세 질환을 동시에 유발할 수 있는 병인에 의한 것일 수 있으므로, 증례의 분석을 통하여 어머니의 임신성 당뇨에 의하여 이 세 질환의 발생이 가능할 것으로 가정하였다. 모체의고혈당증은 주로 신경능선 조직에서 발생하는 태아구조에 영향을 주는 것으로 알려져 있고, 신경능선 조직에서 분화하는 조직들 중 하나인 구개골의 형성에 영향을 주어 구개열의 발생이 가능할 것으로 보이며, 임신성 당뇨에서 과체중아의 출생 빈도가 증가하므로, 이와 관련된 분만 손상 중 분만성 상완 신경총 손상 및 선천성근성사경의 발생이 가능할 것으로 판단된다. 따라서 임신성 당뇨가 있는 산모의 태아에서 구개열등의 선천성 기형의 위험성이 높으므로 산전 검사 시 유의해야하며, 과체중아에서 분만성 상완 신경총 손상과 선천성근성사경의 위험성이 높으므로 이에 대한 주의와 확인이 필요할 것으로 판단된다. 그러나, 분만성 상완 신경총 손상, 선천성근성사경 및 구개열의 병발이 산모의 임신성 당뇨와 연관된 합병증일 수 있다는 것을 증명하기 위하여 향후 더 많은 증례를 통한 심화된 연구가 필요하다.

장애아동과 비장애아동의 의료이용 및 질병특성 비교 (Comparison of Health Care Utilization and Morbidity of Children With and Without Disabilities in Korea)

  • 김유진;김경미;유동철
    • 한국콘텐츠학회논문지
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    • 제17권7호
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    • pp.696-706
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    • 2017
  • 본 연구는 장애아동과 비장애아동의 의료이용 및 질병이환의 특성을 분석하여 건강격차를 비교하고자 시행하였다. 연구자료는 2010년도 국민건강보험공단의 표본코호트자료를 이용하여 0-19세 장애아동과 비장애아동을 대상으로 의료이용량과 진료비, 질병보유수, 다빈도질환과 아동장애와 관련질환의 환자비율 등을 비교하였다. 분석결과 장애아동은 비장애아동보다 의료이용빈도 및 입원율, 질병보유수가 많아 건강상태가 좋지 않고 진료비를 더 많이 지출하였다. 두 집단 간에 많이 겪는 다빈도질환도 차이가 나서 장애아동은 신경계통질환, 순환기계질환 및 정신행동장애의 순으로 많은 반면, 비장애아동은 호흡기계질환, 손상 및 중독질환, 감염성질환 순이였다. 장애아동은 신경계통의 선천기형질환이나 신체장애와 밀접한 건강관련질환의 의료이용이나 진료비지출이 많으나, 비장애아동은 감염성질환에서 높게 나타났다. 결론적으로 비장애아동보다 잦은 진료와 입원, 과다한 의료비 지출과 복합적으로 보유한 질환 등으로 특징지워지는 장애아동의 의료접근성을 향상시키는 다양한 보건의료정책이 필요하다. 그리고 손상으로 인한 장애가 추가적으로 발생하거나 심화되지 않도록 장애아동 및 비장애아동의 부모에게 예방교육이 필요하다.

13번 환염색체의 모자이크 증후군 (A case of mosaic ring chromosome 13 syndrome)

  • 김수영;오수민;김미정;송은송;김영옥;최영륜;우영종;황태주
    • Clinical and Experimental Pediatrics
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    • 제52권2호
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    • pp.242-246
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    • 2009
  • 13번 환염색체 증후군은 정신발달의 지체, 성장 장애, 안면부 기형, 중추신경계 기형, 심장기형, 손발의 기형, 골격계 기형 및 항문기형의 특징을 가진다. 많은 수의 13번 환염색체 증후군에 대한 보고가 있었지만 국내에서는 오직 6예의 임상증례 고가 있다. 이들 보고는 모두 순수한 13번 환염색체 증후군을 보고한 것으로 본 저자들은 13번 환염색체 모자이크 현상의 증례를 경험하였다. 산전진찰 상 자궁내 발육지연이 의심되었던 남아에서 안면부기형이 관찰되었으나 이 외에 이학적 검사상 심장 기형, 골격계 및 외부 생식기 기형은 특별히 관찰되지 않았다. 시행한 세포 유전학 검사상 13번 염색체의 ring/monosomy/dicentric 모자이크 현상이 나타났다. 이후에도 근력저하, 성장과 발달지연을 보이고 있다. 저자들은 안면부 기형, 소두증과 대칭성 자궁내 발육지연을 보인 남아에서 13번 환염색체의 모자이크 증후군을 경험하여 기존에 보고된 다른 증례들과 임상 양상을 비교하여 보고하는 바이다.

Roles of GASP-1 and GDF-11 in Dental and Craniofacial Development

  • Lee, Yun-Sil;Lee, Se-Jin
    • Journal of Oral Medicine and Pain
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    • 제40권3호
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    • pp.110-114
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    • 2015
  • Purpose: Growth and differentiation factor (GDF)-11 is a transforming growth factor-${\beta}$ family member that plays important regulatory roles in development of multiple tissues which include axial skeletal patterning, palatal closure, and tooth formation. Proteins that have been identified as GDF-11 inhibitors include GDF-associated serum protein (GASP)-1 and GASP-2. Recently, we found that mice genetically engineered to lack both Gasp1 and Gdf11 have an increased frequency of cleft palate. The goal of this study was to investigate the roles of GDF-11 and its inhibitors, GASP-1 and GASP-2, during dental and craniofacial development and growth. Methods: Mouse genetic studies were used in this study. Homozygous knockout mice for Gasp1 ($Gasp1^{-/-}$) and Gasp2 ($Gasp2^{-/-}$) were viable and fertile, but Gdf11 homozygous knockout ($Gdf11^{-/-}$) mice died within 24 hours after birth. The effect of either Gasp1 or Gasp2 deletion in $Gdf11^{-/-}$ mice during embryogenesis was evaluated in $Gasp1^{-/-}$;$Gdf11^{-/-}$ and $Gasp2^{-/-}$;$Gdf11^{-/-}$ mouse embryos at 18.5 days post-coitum (E18.5). For the analysis of adult tissues, we used $Gasp1^{-/-}$;$Gdf11^{+/-}$ and $Gasp2^{-/-}$;$Gdf11^{+/-}$ mice to evaluate the potential haploinsufficiency of Gdf11 in $Gasp1^{-/-}$ and $Gasp2^{-/-}$ mice. Results: Although Gasp2 expression decreased after E10.5, Gasp1 expression was readily detected in various ectodermal tissues at E17.5, including hair follicles, epithelium in nasal cavity, retina, and developing tooth buds. Interestingly, $Gasp1^{-/-}$;$Gdf11^{-/-}$ mice had abnormal formation of lower incisors: tooth buds for lower incisors were under-developed or missing. Although $Gdf11^{+/-}$ mice were viable and had mild transformations of the axial skeleton, no specific defects in the craniofacial development have been observed in $Gdf11^{+/-}$ mice. However, loss of Gasp1 in $Gdf11^{+/-}$ mice occasionally resulted in small and abnormally shaped auricles. Conclusions: These findings suggest that both GASP-1 and GDF-11 play important roles in dental and craniofacial development both during embryogenesis and in adult tissues.

미숙아와 정상아의 영유아기 성장발달상태 비교연구 (A Comparative Study on the Growth & Developmental Status of Premature and Full Term Infants During the First 3Years)

  • 박영애
    • 대한간호학회지
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    • 제15권3호
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    • pp.62-73
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    • 1985
  • The problems of growth & development due to maladjustment are gradually increasing while need for the treatment of children's diseases is decreasing. The level of developmental deficiency or delay correlates with neonatal birth weight and also with gestational age, i.e. degrees of prematurity. There-fore, developmental defects and potential risk factors' are more Common in premature infants than in full term infants. The purpose of this study is to define the difference in the growth at developmental status between premature and full term infants, and to define the relation between the developmental status and the physical growth during the first 3 years' Data were collected from January 10, 1985 to April 6, 1985 at 3 hospitals including St. Mary's Hospital, and through home visiting. The subjects of this study consisted of 79 Premature infants (G.A. <37wks. & B.W. <2.5kg) and 94 full term infants (G.A.≥37 wks. & B.W.≥2.5kg). The study method used was a questionnaire, anthropometric assessment and DDST for normative data of growth & development. The collected data were analyzed using descriptive statistics, chi-square test and t-test. The results of the study were as follows: Hypothesis: 1 : That the prematures will differ from the full term infants in the physical growth status during the first 3 years was partially supported (p<0.02) : The prematures reached up the full term infants in the physical growth status in the first 6 months. And, the first hypothesis was supported (P<0.01) : There are more cases which is below‘the Korean children's physical. growth standards’in prematures than in full term infants. Hypothesis 2 : That the prematures will differ from the full term infants in the developmental status during the first 3 years was supported (P< 0.001);‘Normal’developmental status due to DDST was less in prematures than in full term infants. And, the second hypothesis was Partially supported (P<0.02) : The developmental status of the pre-matures was different from that of the full term infants within the first 3 months by analysis of passed items in DDST, Hypothesis 3 : That the prematures' developmental status will relate to their physical growth during the first 3 years was supported (P<0.001) : If the prematures' developmental status is in delayed status, then, their physical growth status is also in delayed status. This study shows that the prematures differed significantly from the full term infants in the growth at developmental status during their infancy. This means that the nurse can foster the growth & development of the prematures by supportive care during their infancy. Further longitudinal study is needed to verify these findings for the environmental factors.

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경한 임상 경과를 보인 신생아 시기의 프로피온산혈증 1례 (A Case of Neonatal Onset Propionic Acidemia with Mild Clinical Presentations)

  • 김경란;김진섭;허림;박형두;조성윤;진동규
    • 대한유전성대사질환학회지
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    • 제16권1호
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    • pp.47-51
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    • 2016
  • 프로피온산혈증은 상염색체 열성유전에 의해 발생하는 질환으로, 혈중에 암모니아, 독성 물질 등이 축적되어 경한 증상에서부터 사망에까지 이르는 질환이다. 유병률은 50,000-100,000명당 1명이다. PCCA 또는 PCCB 유전자의 돌연변이로 발생하며, 이것을 규명하는 것이 가장 확실한 진단 방법이다. 두 유형에 따른 임상경과 차이는 뚜렷하게 밝혀져 있지 않다. 발병 시기에 따라 신생아기형과 후기형으로 나눌 수 있다. 증상 발현의 원인은 이화작용을 강화시키는 감염, 스트레스, 변비, 단백질의 과도한 섭취 등이며, 운동실조, 이상행동, 식욕부진, 주기적 구토, 성장장애, 신경발달이상 등의 광범위한 임상 경과를 보인다. 정확한 진단과 조속한 초기 치료가 환자의 생존율 및 신경 발달 장애 여부에 중요한 요소이다. 본 증례는 고암모니아혈증 및 대사산증이 심하지 않았으나 조기에 대사성 질환을 염두에 두고 PCCA와 PCCB 유전자의 돌연변이를 분석하여 프로피온산혈증을 진단하고 적극적인 금식 및 수액 치료와 진단을 통해 신체 발달 및 운동 및 치명적인 신경학적 장애 없이 성장한 고무적인 사례로 이를 보고하는 바이다.

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Effects of Neonatal Exposure of Di (n-butyl) Phthalate and Flutamide on Male Reproduction in Rats

  • Kim, Tae-Sung;Kim, Hyung-Sik;Shin, Jae-Ho;Lee, Su-Jung;Moon, Hyun-Ju;Kang, Il-Hyun;Kim, In-Young;Seok, Ji-Hyun;Oh, Ji-Young;Han, Soon-Young
    • 한국수정란이식학회:학술대회논문집
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    • 한국수정란이식학회 2002년도 국제심포지엄
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    • pp.109-109
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    • 2002
  • In recent reports, the multiple reproductive defects such as cryptorchidism, hypospadias, epididymal cysts, low sperm counts, and testicular cancers are increased in humans, and these changes were doubted by the chemicals with estrogenic or antiandrogenic activities in our environment. To compare the effects of neonatal exposure of di (n-butyl) phthalate and flutamide on the development of reproductive organs and to identify the specific mechanisms of these abnormalities related to the male reproducton, Sprague-Dawley neonate male rats were injected subcutaneously during 5-14 days after birth with corn oil (control), flutamide (0.05, 0.1, and 0.5 mg/animal) and DBP (5, 10, and 20 mg/animal). Animals were killed at 31 (immature) and 42 (pubertal) days of age respectively and blood was collected from abdominal aorta for serum testosterone analysis. Testes, epididymides, seminal vesicles, ventral prostate, levator ani plus bulbocavernosus muscle (LABC), cowpers glands and glans penis were weighed. Expression of steroid hormone receptors (AR and ER) was examined in the testes and ventral prostate. At 31 days of age, ventral prostate, seminal vesicles, LABC, and cowpers glands significantly decreased in the flutamide (0.5 mg/animal) and DBP (20 mg/animal), but serum testosterone levels were not changed. Flutamide slightly delayed the testes descent at the high dose (0.5 mg/animal), but DBP did not show any significant effect on the testes descent at all doses. DBP and flutamide decreased the expression of AR protein in the testes but did not affect the expression of ERa and ER protein in the testes. At 42 days of age, ventral prostate, seminal vesicles, and cowpers glands weights were still significantly decreased at the high dose of flutamide (0.5 mg/animal) and DBP (20 mg/animal), but the weights of testes and epididymides were not different. Serum testosterone decreased significantly in DBP treated animals and slightly, not significantly, in flutamide group. While DBP still significantly decreased the expression of AR protein in testis, flutamide recovered from downregulation of AR protein and did not affect the expression of ERa and ER protein in the testes. Based on these results, flutamide and DBP have shown several similar patterns in reproductive abnormalitis, but some marked differences which may be caused by different acting mechanism.

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스폰지 목욕에 대한 미숙아의 생리적상태 및 행동반응 (Physiologic state and behavioral response to sponge bathing in premature infants)

  • 이혜경;홍경자;남은숙;이영희;정은자
    • Child Health Nursing Research
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    • 제6권1호
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    • pp.32-50
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    • 2000
  • A descriptive exploratory design was used in this study to evaluate the effects of sponge bathing on physiological(heart rate, heart period, vagal tone, oxygen saturation, respiration) and behavioral responses in newly born 40 preterm infants from intensive care unit of S University Hospital in Seoul. Data has been collected from October, 1997 to March, 1999. The infants were between 27-33 weeks gestational age at birth, and were free of congenital defects. The subjects entered the protocol when they were medically stable (determined by initiation of feeding and discontinuation of all respiratory support) but still receiving neonatal intensive care. The infants' physiologic parameters were recorded a 10 - minute before, during, and after bathing. Continuous heart rate data were recorded on a notebook computer from the infant's EKG monitor. The data were digitized off-line on software(developed by Lee and Chang in Wavelet program) which detected the peak of the R wave for each heart beat and quantified sequential R-R intervals in msec(i.e. heart periods). Heart period data were edited to remove movement artifact. Heart period data were quantified as : 1) mean heart period; 2) vagal tone. Vagal tone was quantitfied with a noninvasive measure developed by Porges(1985) in Mxedit software. To determine behavioral status, tools were developed by Scafidi et al(1990) were used. Collected data were analyzed with the SPSS program using paried t-test, ANOVA, and Pearson correlation. The result were as follow. 1. The results of the ANOVAs indicated that vagal tone were signifcantly lower during bathing than baseline and post-bathing. There were significant differences in heart period and heart rate levels across the bathing. But the mean oxygen saturations and respirations were no differences. Also, there were no significant differences on behavioral sign, motor activity, behavioral distress, weight changes, morbidity, and hospitalization period. 2. To evaluate the relation between vagal tone and subsequent parameters, the two groups (the high group had 19 subjects and low group had 21subjects) were divided by the mean baseline vagal tone. Vagal tone measured prior to bathing were significantly associated with respiration before bathing, vagal tone during bathing, and the magnitude of change in both vagal tone. But, other subsequent reactivities were no differences in two groups. 3. Correlations were also calculated between vagal tone and the subsequent physiological reactivities from baseline through after- bathing. Correlations were significant between baseline vagal tone and baseline heart rate, between baseline vagal tone and baseline heart period, between baseline vagal tone and oxygen saturation after bathing. In summary, the bathing in this study showed a stressful stimulus on premature infants through there was significance in the physiological parameters. In addition, our study represents the documentation that vagal tone reactivity in response to clearly defined external stimulation provides an index of infant's status.

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초등학생 심장병 집단검진을 통한 선천성 심장병 유병률 (Prevalence of Congenital Heart Disease from the Elementary Student Heart Disease Screening Program)

  • 이홍주;김명희;정조원;김성호;최보율
    • Journal of Preventive Medicine and Public Health
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    • 제34권4호
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    • pp.427-436
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    • 2001
  • Objective : To estimate the prevalence of congenital heart disease from the 1990 student heart disease screening program. Methods : The heart disease screening program for elementary students was conducted in Kyonggi-do, in 1998. The subjects of the present study comprised the 40,402 students who attended the schools in the catchment area of a collaborative university hospital and who participated in the primary examination. The congenital heart disease (CHD) patients were initially identified through a questionnaire about prior medical history, and further through diagnostic tests & medical examinations in the secondary & the tertiary examinations. Certain assumptions were used in the estimation of the number of CHD cases among non-participants of the secondary & tertiary examinations. The overall prevalence of CHD was estimated by adding the CHD detection rates of the participants and the estimated prevalence of the non-participants. Results : Among the 40,402 primary participants, 1,655 were referred further, of whom 79.1% (1,309) participated in the secondary examination. Of these, 121 were referred to the tertiary examination, with a participation rate at this last stage of 80.2%. The positive predictive value (PPV) of the screening tools was the highest when the results of both EKG and the questionnaire were positive. Because 85.9% of the detected cases had a past history of CHD, PPV was higher when the selection criteria in the questionnaire included past CHD history than when it didnt. The CHD defection rate among the participants was 1.76 cases/1,000 and the presumed number of cases among the non-participants was 31; giving an estimated final CHD prevalence of 2.52 cases/1,000 (95% CI : 2.06-3.06). Among the identified cases of CHD, VSD (52.8%) was the most common, followed by PDA (9.7%), TOF (9.7%) & PS (9.7%). Conclusion : Because the characteristics of the non-participants differed from those of the participants, the estimation of prevalence was influenced by the participation rate. Of the detected cases, 85.9% had a past history of diagnosis or operation for CMD. These findings suggested that the prevalence estimated in this study may be an underestimation of the actual condition. Therefore, a birth cohort study is required in order to more accurately estimate the prevalence and the effects of the program.

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