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ID naloge: 74 Letnik: 1999 Predmet: pediatrija
Navidezno življenje ogrožajoc dogodek
(usmerjena anamneza, funkcijske kardiorespiratorne preiskave in biokemicni pokazatelji)
Avtor: Tanja Milanic in Urška Weber
Mentor: prof. dr. Jože V. Trontelj, dr. med
Uvod: Navidezno življenje ogrožajoc dogodek (NŽOD) pri dojencku je opredeljen kot dogodek, ob katerem se opazovalcu zdi, da je otrok v življenjski nevarnosti, ali celo, da je umrl. Osnovna znacilnost tega dogodka so specificne motnje srcno-dihalnih vzorcev (SDV). Rezultati številnih študij kažejo, da je NŽOD, zlasti v primeru vzrocne neopredeljenenosti, povezan z nastankom nenadne nepricakovane smrti dojencka (NNSD). Tudi pri dojenckih z NNSD so bile namrec pred smrtjo dokazane specificne motnje SDV, katerih vzrok naj bi bila po danes prevladujoci hipotezi o patogenezi NNSD klinicno nema kronicna hipoksija. Slednja je bila dokazana tako pri žrtvah NNSD kot pri dojenckih z NŽOD. Kljub tem skupnim znacilnostim med NNSD in NŽOD pa pri slednjem še vedno ni možno zanesljivo poiskati primerov z visokim tveganjem za NNSD. Za oceno ogroženosti za pojav NNSD je pri teh dojenckih še najbolj zanesljiva metoda snemanje SDV. Glede na to jo lahko uporabimo tudi za ovrednotenje novejših tovrstnih metod. Fleming je nedavno predpostavil, da bi lahko na to oceno dodatno sklepali preko stopnje resnosti NŽOD, mogoce pa bi lahko za NNSD huje ogrožene dojencke v tej skupini poiskali tudi na osnovi ovrednotenja stopnje kronicne hipoksije, katere dober pokazatelj je nivo hipoksantina v telesnih tekocinah.
Namen: V tej nalogi smo se na osnovi omenjenih ugotovitev namenili preveriti naslednje:
1.) Prva hipoteza: Med stopnjo resnosti NŽOD in stopnjo specificne abnormnosti zapisa SDV obstoja statisticno znacilno ujemanje pri dojenckih z vzrocno neopredeljenim NŽOD, medtem ko pri tistih z vzrocno opredeljenim NŽOD takega ujemanja ni.
2.) Druga hipoteza: pri dojenckih z NŽOD obstoja med stopnjo urinskega hipoksantina in stopnjo specificne abnormnosti v zapisu SDV statisticno znacilno ujemanje.
3.) Kolikšen delež vseh dojenckov z NŽOD je pri nas vzrocno opredeljenih oz. neopredeljenih in kateri so najpogostejši vzroki?
Metode: V raziskavo je bilo vkljucenih 74 dojenckov z NŽOD (38 deckov, 36 deklic, povprecna starost 2,03 ± 1,79 meseca). Vsi podatki o preiskovancih so bili zajeti iz popisov hospitalizacij ter izpolnjenih usmerjeno-anamnesticnih vprašalnikov, t.i. taksonomskih profilov NŽOD. Na osnovi slednjih smo ocenili stopnjo resnosti dogodka preko ovrednotenja intenzitete štirih znacilnih znakov NŽOD (dihanje, barva kože, nevrološki status, za prekinitev dogodka potreben ukrep). Stopnjo hipoksije pri teh otrocih smo dolocili na osnovi vrednosti koncentracijskega razmerja med hipoksantinom in kreatininom v urinu. Na stopnjo ogroženosti za morebitno NNSD smo sklepali na osnovi specificne abnormnosti zapisa SDV. Kot vzrocno opredeljene smo smatrali tiste NŽOD, pri katerih je bila v popisu hospitalizacije zaradi NŽOD postavljena vsaj ena diagnoza, poznana kot vzrocni dejavnik za nastanek tega dogodka. Delovni hipotezi smo preverili z Wilcoxonovim testom vsote predznacenih rangov dveh soodvisnih vzorcev, pri cemer je bila stopnja tveganja za veljavnost nicelne hipoteze 0,05. Stopnjo ujemanja med spremenljivkami (popolno ali delno ujemanje, popolno neujemanje) smo ugotavljali po modificirani McNemarovi metodi.
Rezultati: Med stopnjo resnosti vzrocno neopredeljenega NŽOD in stopnjo specificne abnormnosti zapisa SDV smo ugotovili statisticno znacilno ujemanje. Popolno ali delno ujemanje je bilo ugotovljeno pri 77,0 % preiskovancev. V skupini vzrocno opredeljenih NŽOD statisticno znacilnega ujemanja ni bilo. V celotni skupini dojenckov z NŽOD smo med stopnjo urinskega hipoksantina in stopnjo specificne abnormnosti zapisa SDV ugotovili statisticno znacilno ujemanje. Popolno ali delno ujemanje je bilo prisotno pri 77,8 % preiskovancev. Po koncanih diagnosticnih postopkih je bilo 46,0 % vseh preiskovanih primerov NŽOD vzrocno opredeljenih, 54,0 % pa vzrocno neopredeljenih. Najpogostejši vzroki so bili gastroezofagealni refluks, nevrološke motnje in okužbe zgornjih dihal.
Zakljucki:
1.) Pri skupini dojenckov z vzrocno neopredeljenim NŽOD verjetno z usmerjeno-anamnesticnim vprašalnikom, t.i. taksonomskim profilom NŽOD, lahko prispevamo dodatno informacijo v celostno oceno tveganja za pojav NNSD pri teh otrocih. Njegovo izpolnjevanje ima verjetno precejšen pomen, kajti pri nas tudi po koncanih diagnosticnih postopkih dobra polovica primerov NŽOD ostane vzrocno neopredeljenih. To bi lahko pomenilo, da ima otrok s takim dogodkom ob sprejemu približno 50-odstotno možnost, da njegov taksonomski profil vsebuje informacijo o tveganju za pojav NNSD.
2.) Pri vecini dojenckov z NŽOD vrednost koncentracijskega razmerja med hipoksantinom in kreatininom v urinu verjetno lahko prispeva dodatno informacijo v celostno oceno tveganja za pojav NNSD pri teh otrocih.
3.) Pri nas je v skupini dojenckov z NŽOD 46,0 % primerov vzrocno opredeljenih, 54,0 % pa jih po koncani diagnosticni obdelavi ostane vzrocno nepojasnjenih. Najpogostejši vzroki so bili gastroezofagealni refluks, nevrološke motnje in okužbe zgornjih dihal. Ker je odstotek vzrocno neopredeljenih NŽOD pri nas veliko vecji, standardni diagnosticni protokol pa veliko ožji kot v tujini, bi bilo pri dojenckih z NŽOD morda smiselno razširiti usmerjeno-diagnosticni protokol v smislu iskanja in odprave pravih vzrokov dogodka ter racionalizacije zdravljenja v teh primerih.
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[Abstract / English version]
Apparent life-threatening event
(taxonomy profile, cardiorespiratory patterns and biochemical markers)
Author: Tanja Milanic in Urška Weber
Mentor: prof. dr. Jože V. Trontelj, dr. med
Introduction: Apparent life-threatening event (ALTE) is defined as an episode that is frightening to the observer and in some cases, the observer even fears that the infant has died. The event is basically characterized by a disturbance in cardiorespiratory patterns (CRP). According to several studies, ALTE infants are at increased risk of subsequent dying of sudden infant death syndrome (SIDS), particularly when no treatable cause for the ALTE is found. Disturbances in CRP have also been found in SIDS victims and are thought to be due to chronic, clinically silent hypoxia. The latter has been demonstrated by several studies in both SIDS victims and in ALTE infants. Despite of this it is so far impossible to determine which ALTEs are at increased risk to die of SIDS. Today the most widely used method for evaluation of CRP in infants with ALTE is cardiorespirogram (CRG). Currently in ALTE cases CRG recordings provide the basis for the estimation of degree of risk for SIDS. However, it was also proposed that such an evaluation could be made on the basis of some new methods, such as ALTE taxonomic profile or through the markers of chronic hypoxia, for example body fluids hypoxanthine level.
Objective: The aim of the present study was to verify the following issues:
1.) First hypothesis: In infants with ALTE there is statistically significant degree of correspondence between severity of such event and specific abnormality of the CRG recording in etiologically unexplained cases, while in explained ALTEs no such correspondence can be found.
2.) Second hypothesis: In infants with ALTE there is statistically significant degree of correspondence between the level of urinary hypoxanthine and specific abnormality of the CRG recording.
3.) How many of all ALTE cases can be classified as etiologically explained or unexplained, respectively, after the standard diagnostic procedure and what are the major causes of such events?
Methods: The subjects of this study were 74 infants with a history of an ALTE (38 male and 36 female infants, mean age 2,03 ± 1,79 months). All the data were obtained from hospital records and fulfilled taxonomy profiles. The severity of an ALTE was defined according to the intensity of four ALTE signs (breathing, skin colour, neurological status, intervention). The degree of hypoxia in these infants was assesed by the ratio of the concentration of hypoxanthine to creatinine in urine samples. The estimation of potential risk for SIDS was determined according to specific abnormality of the CRG recordings. All those cases cases of ALTE, in which at least one possible cause of such event was found, were assumed explained. Our hypotheses were evaluated with Wilcoxon matched-pairs signed-ranks test. The degree of correspondence between variables (complete or partial correspondence, complete discordance) was analyzed through the method of McNemara.
Results: Statistically significant degree of correspondence (p < 0,05) was observed between severity of an unexplained ALTE and specific abnormality of the CRG recording. Accordingly, in 77,0 % of the unexplained ALTE infants complete or partial correspondence was observed, while in the group of the explained ALTEs no such correspondence could be found. Between the level of urinary hypoxanthine and specific abnormality of the CRG recording we found statistically significant degree of correspondence (p < 0,05). In 77,8 % of cases complete or partial correspondence was found. In our study 54,0 % of ALTEs could not be etiologically explained even after a careful diagnostic procedure, in the rest 46,0 % however, the most frequent causes were gastroesophageal reflux, neurological disorders and upper airway infection.
Conclusions:
1.) In those ALTEs, where no treatable cause is found, i.e. in unexplained ALTEs, important information regarding possible risk for SIDS might be obtained from accurately fulfilled taxonomic profile. This could be of great significance since there is a more than a half ALTE cases that can not be explained even after a careful diagnostic procedure. This might implicate, that for an infant with an ALTE there is about 50 % chance that his/her taxonomic profile carries an important information on the risk for SIDS.
2.) Ratio of the concentration of hypoxanthine to creatinine, measured in urine samples, provides an additional information about possible risk for SIDS in group of ALTE infants.
3.) In ALTE infants 54,0 % of the causes remain unexplained, while in remainder 46,0 % the major causes are gastroesophageal reflux, neurological disorders and infections of upper respiratory tract. A greater proportion of unexplained ALTEs found in our study compared to the foreign ones, possibly due to the narrower diagnostic protocol we use, might be reduced by expanding the existing diagnostic procedures. At the same time, therapeutic approach to the ALTE infants could be rationalized.
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