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| 2. |
- Aring, Eva, 1959, et al.
(författare)
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The FASD Eye Code: a complementary diagnostic tool in fetal alcohol spectrum disorders
- 2021
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Ingår i: Bmj Open Ophthalmology. - : BMJ. - 2397-3269. ; 6:1
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Tidskriftsartikel (refereegranskat)abstract
- Objective To create an easy-to-use complementary ophthalmological tool to support a fetal alcohol spectrum disorder (FASD) diagnosis. Methods and Analysis The FASD Eye Code was derived from 37 children with FASD evaluated along with 65 healthy age-matched and sex-matched controls. Four ophthalmological categories, which are abnormalities commonly found in children with FASD, were ranked independently on a 4-point scale, with 1 reflecting normal finding and 4 a strong presence of an abnormality: visual acuity, refraction, strabismus/binocular function and ocular structural abnormalities. The tool was validated on 33 children with attention deficit/hyperactivity disorder (ADHD), 57 children born moderate-to-late premature (MLP) and 16 children with Silver-Russell syndrome (SRS). Among children with ADHD none was born prematurely or small for gestational age (SGA) or diagnosed with FASD. Among children born MLP none was SGA, had a diagnosis of ADHD or FASD, or a history of retinopathy of prematurity. Children with SRS were all born SGA, half were born preterm and none had FASD. Children with FASD were re-examined as young adults. Results An FASD Eye Code cut-off total score of >= 10 showed an area under the curve (AUC) of 0.78 (95% CI 0.69 to 0.87), with 94% specificity and 43% sensitivity, in discriminating between FASD and controls, MLP and ADHD, corresponding to a positive likelihood ratio (LR+) of 7.5. Between FASD and controls, an AUC of 0.87 (CI 0.80 to 0.95), with 100% specificity and 43% sensitivity, was found; between FASD and SRS, an AUC of 0.60 (CI 0.45 to 0.75) was found, with 88% specificity and 43% sensitivity. A cut-off score of >= 9 showed a specificity of 98% and a sensitivity of 57% for FASD versus controls, corresponding to an LR+ of 36.9. Scores in individuals with FASD were stable into young adulthood. Conclusion The FASD Eye Code has the potential to serve as a complementary tool and help to strengthen an FASD diagnosis.
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- Ayoub, L., et al.
(författare)
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Visual and ocular findings in children with fetal alcohol spectrum disorders (FASD): validating the FASD Eye Code in a clinical setting
- 2023
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Ingår i: Bmj Open Ophthalmology. - : BMJ. - 2397-3269. ; 8:1
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Tidskriftsartikel (refereegranskat)abstract
- ObjectiveFetal alcohol spectrum disorders (FASD) is an umbrella term covering a spectrum of medical conditions caused by prenatal alcohol exposure. The FASD Eye Code is a new complementary ophthalmological diagnostic tool created to corroborate the complex FASD diagnosis. The aim of this work was to validate the FASD Eye Code by testing it on a second group of children diagnosed with FASD in a clinical setting.Methods and analysisA clinical study was carried out in a group of 21 children (13 males, 8 females, mean age 13.3 years) investigated for suspected FASD and a healthy sex-matched and age-matched control group (n=21). The participants underwent a detailed ophthalmological examination including visual perception problems (VPPs) assessment. Clinical examination results were compiled, and total scores were calculated according to the FASD Eye Code protocol (range 4-16).ResultsThe median total score in the FASD group was 8. Eight individuals in the FASD group and none of the controls obtained a total score of >= 9 corresponding to 38% sensitivity and 100% specificity with an area under the curve of 0.90. A cut-off total score of >= 8 showed 52% sensitivity and 95% specificity. One individual in the FASD group versus 12 controls had a total score of 4, representing normal findings. No significant difference between the two groups regarding VPPs was seen.ConclusionThe FASD Eye Code can be used as a complementary diagnostic tool for FASD to assist in diagnosis and to detect ophthalmological abnormalities in individuals with suspected FASD.
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- Gyllencreutz, Emelie, et al.
(författare)
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Ophthalmologic Findings in Fetal Alcohol Spectrum Disorders : A Cohort Study From Childhood to Adulthood
- 2020
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Ingår i: American Journal of Ophthalmology. - : Elsevier. - 0002-9394 .- 1879-1891. ; 214, s. 14-20
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Tidskriftsartikel (refereegranskat)abstract
- PURPOSE: To investigate whether ophthalmologic findings in children with fetal alcohol spectrum disorders (FASD) persist into young adulthood.DESIGN: Prospective cohort study.METHODS: Thirty children (13 female) adopted from eastern Europe to Sweden in the 1990s and diagnosed with FASD by a multidisciplinary team at the median age of 7.9 years were followed up by the same team 13-18 years later. Visual acuity (VA), refraction, stereoacuity, strabismus, ocular media, and fundus were investigated.RESULTS: Median VA in right/left eye (OD/OS) was 20/32/20/32 (0.2/0.2 logMAR) in childhood and 20/22/20/20 (0.05/0.0 logMAR) in adulthood. Median (range) refraction OD/OS was +0.88/+1.25 (-8.75 to +4.75/-9.38 to +5.25) spherical equivalent diopter (D) in childhood and -0.25/-0.25 (-12 to +2.75/-13.25 to +2.63) in adulthood. Astigmatism (≥1 D) was the most common refractive error, in 13 (40%) and 14 (47%) subjects, respectively. Defective stereoacuity (>60 arc second) was noted in 20 subjects (67%) in childhood and 22 (73%) in adulthood. Heterotropia occurred in 12 subjects (40%) in childhood and 13 (43%) in adulthood. Increased tortuosity of the retinal vessels was found in 8 (27%) subjects in childhood vs 11 (37%) in adulthood. Optic nerve hypoplasia was recorded in 3 children and in 4 young adults.CONCLUSIONS: Ophthalmologic findings such as refractive errors, strabismus, and fundus abnormalities are frequent in children with FASD and persist into early adulthood. The facial features characteristic of FAS diminish with age, making a dysmorphology evaluation in adulthood less reliable. An ophthalmologic examination is an important part of the evaluation of FASD in childhood as well as in young adulthood.
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| 7. |
- Gyllencreutz, Emelie, et al.
(författare)
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Thinner retinal nerve fibre layer in young adults with foetal alcohol spectrum disorders
- 2021
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Ingår i: British Journal of Ophthalmology. - : BMJ Publishing Group Ltd. - 0007-1161 .- 1468-2079. ; 105:6, s. 850-855
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Tidskriftsartikel (refereegranskat)abstract
- BACKGROUND/AIMS: Ophthalmological abnormalities such as ptosis, strabismus, refractive errors and optic nerve hypoplasia have been reported in foetal alcohol spectrum disorders (FASD). The purpose of this study was to investigate whether retinal thickness, retinal nerve fibre layer (RNFL) and optic disc area (ODA) differ between individuals with FASD and healthy controls.METHODS: Best-corrected visual acuity (BCVA) in terms of logarithm of the minimum angle of resolution (logMAR), refraction, and fundus variables measured by optical coherence tomography were obtained from 26 young adults with FASD (12 women, median age 23 years) and 27 controls (18 women, median age 25 years).RESULTS: The total thickness of the peripapillary RNFL was significantly lower in the FASD group than in controls; median (range) in the right/left eye was 96.5 (60-109)/96 (59-107) µm in the FASD group and 105 (95-117)/103 (91-120) µm among controls (p=0.001 and p=0.0001). Macular RNFL and retinal thickness measurements from the FASD group were also lower in most of the nine ETDRS areas, except for the central parts. Median (range) BCVA in the best eye was 0.00 (-0.1-0.3) logMAR in the FASD group and 0.00 (-0.2-0.0) logMAR in controls (p=0.001). No significant differences between the groups were found regarding ODA or refraction.CONCLUSION: Significant differences in peripapillary and macular RNFL, retinal thickness and BCVA were found in this group of young adults with FASD compared with healthy controls. However, there were no differences in the size of the optic disc.
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| 8. |
- Gyllencreutz, Emelie, et al.
(författare)
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Visual perception problems and quality of life in young adults with foetal alcohol spectrum disorders
- 2022
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Ingår i: Acta Ophthalmologica. - : Wiley. - 1755-375X .- 1755-3768. ; 100:1
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Tidskriftsartikel (refereegranskat)abstract
- Purpose To investigate visual perception problems (VPPs), health-related quality of life (HRQoL) and vision-related quality of life (VRQoL) in young adults with foetal alcohol spectrum disorders (FASD) and to compare the results with healthy controls. Methods Thirty young adults with FASD (13 female; mean age 23 years) and 29 controls (20 female; mean age 25 years) participated. Five areas of VPPs were assessed by a structured history-taking. In the FASD group, VPPs were investigated both in childhood (mean age 8 years) and in early adulthood in a prospective follow-up. Health-related quality of life (HRQoL) was investigated with the Pediatric Quality of Life Inventory (TM) (PedsQL) and VRQoL with the 25-item Visual Function Questionnaire (VFQ-25). Results Visual perception problems (VPPs) in at least one area were reported by 16/30 FASD participants (53%) and 1/29 controls (3%) (p = 0.0001, Fisher's exact test), with a similar rate in the same individuals in childhood as in early adulthood (8/27 and 15/27, respectively p = 0.09, McNemar's test). PedsQL total score was lower in the FASD group (n = 20; median: 83; 95% confidence interval (CI) 76-88) compared with controls (n = 29; median: 91; 95% CI 90-95; p = 0.0001, Mann-Whitney U-test). VFQ-25 subscale general vision indicated lower VRQoL in the young adults with FASD (n = 19; median: 80; 95% CI 80-100) compared with controls (n = 29; median: 100; 95% CI 100-100; p = 0.003). Conclusion Young adults with FASD in the present study had more VPPs and worse VRQoL and HRQoL than healthy controls. In the FASD group, VPPs were reported in childhood as well as in early adulthood.
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| 9. |
- Landgren, Valdemar, 1988, et al.
(författare)
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Attention-deficit/hyperactivity disorder with developmental coordination disorder: 24-year follow-up of a population-based sample
- 2021
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Ingår i: BMC Psychiatry. - : Springer Science and Business Media LLC. - 1471-244X. ; 21:1
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Tidskriftsartikel (refereegranskat)abstract
- BackgroundAlthough the body of research concerning neurodevelopmental disorders is vast, there is a scarcity of longitudinal studies beyond late adolescence, and of studies taking co-existing disorders into account. The present study aimed to investigate outcome in adulthood for children with attention-deficit/hyperactivity disorder (ADHD) combined with developmental coordination disorder (DCD) diagnosed at 6.6years of age.MethodsOut of a screening-based population cohort of 589 individuals, 62 (10 female) diagnosed with ADHD+DCD at mean age 6.6years naive to stimulant treatment were followed into adulthood through national registries. Results were compared to a screen- and assessment negative population matched group from the same cohort (PM group, n=51) and a registry-matched (RM group, n=410) group of the same county and age.ResultsAt 30 to 31years of age, five deaths had occurred; one in the ADHD+DCD group and two each in the comparison groups. In time to event analyses of the composite outcome of any psychiatric disorder, psychotropic prescription, sick pension or criminal sentence, events occurred at a significantly higher rate in the ADHD+DCD group (p=0.0032, vs PM group p=0.0115, vs RM group p=0.0054). The ADHD+DCD group had significantly higher rates of psychiatric diagnoses, prescriptions of psychoactive medications and occurrence of sick pension than both comparison groups. Further, the ADHD+DCD group had significantly lower educational attainment compared to both comparison groups, more years with unemployment, and overall higher welfare recipiency. Rates of pain diagnoses and analgesic prescriptions did not separate the groups.ConclusionADHD+DCD entailed a less favorable outcome in adulthood compared to a non-clinical comparison group and a registry-matched population. Neurodevelopmental disorder diagnosed upon school entry is of prognostic utility with respect to function in adulthood, and warrants early identification and management.
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| 10. |
- Landgren, Valdemar, 1988, et al.
(författare)
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Deficits in attention, motor control and perception childhood to age 30 years: prospective case-control study of outcome predictors
- 2022
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Ingår i: Bmj Open. - : BMJ. - 2044-6055. ; 12:3
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Tidskriftsartikel (refereegranskat)abstract
- Objective Investigate predictors of adverse outcome in children with and without attention-deficit/hyperactivity disorder (ADHD) combined with developmental coordination disorder (DCD) at 6 years of age. Design Prospective population-based cohort study. Setting Western Sweden. Participants From a screening-based population cohort of 589 individuals, 62 (11 female) diagnosed with ADHD+DCD at mean age 6.6 years, and a comparison group of 51 population-matched (10 female) children were followed prospectively. Outcome measures Drawn from a clinical reassessment at age 9 years of 110 of the 113 individuals, neuropsychiatric symptoms, continuous performance test results and measures of motor function were used as predictors of outcome in linear regression models. Participants were followed in national registers up to 30-31 years of age for outcomes in adulthood. Predictors were regressed onto an adverse outcome score (range 0-7) comprising seven binary endpoints, and when applicable onto each continuous outcome separately (low educational attainment, low occupation level, psychiatric disorder, psychotropic medication prescription, sick pension, high dependence on social benefits and criminal conviction). Results Of the 110 individuals, 3 had died. In univariable regression onto the adverse outcome score, the strongest predictors at age 9 years were symptoms of conduct disorder, oppositional defiant disorder, ADHD and motor dysfunction, with an R-2 around 25%, followed by autistic traits (R-2=15%) and depressive symptoms (R-2=8%). Combining these six strongest predictors in a multivariable model yielded an adjusted R-2=38%. Subgroup analyses were similar, except for a strong association of autistic traits with the adverse outcome score in females (n=20, R-2=50%). Conclusion Several neurodevelopmental symptoms, including ADHD severity at age 9 years, accounted for a considerable amount of the variance in terms of adulthood adverse outcome. Broad neurodevelopmental profiling irrespective of diagnostic thresholds should inform research and clinical practice. The study highlights the importance of considering associated comorbidities and problems in ADHD.
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