| 1. |
- Fransson, Emma, et al.
(författare)
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Diagnostic Efficacy of Rectal Suction Biopsy with Regard to Weight in Children Investigated for Hirschsprung's Disease
- 2022
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Ingår i: Children. - : MDPI AG. - 2227-9067. ; 9:2, s. 1-9
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Tidskriftsartikel (refereegranskat)abstract
- BACKGROUND/AIM: Diagnostic efficacy, defined as the percentage of rectal suction biopsy (RSB) specimens sufficient enough to determine the absence of ganglia cells in Hirschsprung's disease (HD) diagnosis, has been reported to be low, requiring repeated biopsies. The aim was to explore whether RSB diagnostic efficacy was influenced by the child's weight and to ascertain whether RSB efficacy differed between aganglionic and ganglionic tissue.MATERIALS AND METHODS: Efficacy analyses were conducted in a national HD-center's register on children 0-15 kg, examined between 2011-2019. First-time RSB diagnostic efficacy was correlated to the children's weight and final HD diagnosis.RESULTS: Among the 84 children who had first-time RSB, the overall diagnostic efficacy was 85% (71/84). The efficacy was higher among children weighing less than the identified cut-off of 9.0 kg (89% in 0-9.0 kg versus 62% in 9.01-15.0 kg, p = 0.026). Among children diagnosed with HD, 96% (26/27) weighed 0-9.0 kg. In this weight group, the diagnostic efficacy was lower in aganglionosis compared to ganglionosis (77%; 20/26 versus 96%; 43/45), p = 0.045).CONCLUSIONS: The RSB diagnostic efficacy was significantly higher in children weighing less than 9.0 kg and was less in aganglionic compared to ganglionic tissue. Therefore, weight can be useful to predict RSB diagnostic efficacy.
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| 2. |
- Hallabro, Nilla, et al.
(författare)
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Mapping of referral patterns for undescended testes – Risk factors for referral of children with normal testes
- 2023
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Ingår i: Journal of Pediatric Urology. - : Elsevier BV. - 1477-5131. ; 19:3, s. 1-320
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Tidskriftsartikel (refereegranskat)abstract
- Background: Accurate referral of boys with suspected undescended testes (UDT) is of importance to preserve fertility and reduce risk of future testicular cancer. While late referral is well studied, there is less knowledge about incorrect referrals, hence, referral of boys with normal testes. Objective: To evaluate the proportion of UDT referrals that did not lead to surgery or follow-up, and to assess risk factors for referral of boys with normal testes. Study design: All UDT referrals to a tertiary center of pediatric surgery during 2019–2020 were retrospectively assessed. Only children with suspected UDT in the referral (not suspected retractile testicles) were included. Primary outcome was normal testes at examination by a pediatric urologist. Independent variables were age, season, region of residence, referring care unit, referrer's educational level, referrer's findings, and ultrasound result. Risk factors for not needing surgery/follow-up were assessed with logistic regression and presented as adjusted odds ratios with a 95% confidence interval (aOR, [95% CI]). Results: A total of 378 out of 740 included boys (51.1%) had normal testes. Patients >4 years (aOR 0,53, 95% CI [0,30-0,94]), referrals from pediatric clinics (aOR 0.27, 95% CI [0.14–0.51]) or surgery clinics (aOR 0.06, 95% CI [0.01–0.38]) had lower risk of normal testes. Boys referred during spring (aOR 1.80, 95% CI [1.06–3.05]), by a non-specialist physician (aOR 1.58, 95% CI [1.01–2.48]) or referrer's description of bilateral UDT (aOR 2.34, 95% CI [1.58–3.45]), or retractile testes (aOR 6.99, 95% CI [3.61–13.55]) had higher risk of not needing surgery/follow-up. None of the referred boys that had normal testes had been re-admitted at the end of this study (October 2022). Discussion: Over 50% of boys referred for UDT had normal testes. This is higher or equal to previous reports. Efforts to reduce this rate should in our setting probably be directed towards well-child centers and training in examination of testicles. The main limitation of this study is the retrospective design and the rather short follow-up time, which however should have very modest effect on the main findings. Conclusion: Over 50% of boys referred for UDT have normal testes. A national survey regarding the management and examination of boys testicles has been launched and directed at well-child centers to further evaluate the findings of the current study.[Formula
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| 3. |
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| 4. |
- Hambraeus, Mette, et al.
(författare)
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Differential Activation of Immune Effector Processes in Mature Compared to Immature Sacrococcygeal Teratomas
- 2022
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Ingår i: Fetal and Pediatric Pathology. - : Informa UK Limited. - 1551-3815 .- 1551-3823. ; 41:3, s. 413-425
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Tidskriftsartikel (refereegranskat)abstract
- Objective: This study aims to characterize the molecular signatures of sacrococcygeal teratomas (SCTs). Methods: Three SCTs were analyzed with whole genome genotyping. RNA sequencing of 10 SCTs dominated by mature, immature and neuroglial elements was analyzed. Expression in SCT-samples with different elements were compared to each other and to a reference group of malignant pediatric tumors. Macrophages, T- and B-lymphocytes were detected by immunohistochemistry. Results: No chromosomal imbalances were detected. SCTs showed overexpression of genes involved in neurosignaling, DNA-binding molecules and pathways of early germ cells. Genes associated with immune effector processes were overexpressed in mature compared to immature SCTs, and immune cell infiltration was found predominantly around mature epithelial elements. Conclusion: The broad repertoire of histological elements in SCTs reflects differences in transcriptional regulation rather than differences in gene copy numbers. A paucity of immune response in immature SCTs may be a factor contributing to their uninhibited growth.
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| 5. |
- Hambraeus, Mette, et al.
(författare)
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Functional outcome and health-related quality of life in patients with sacrococcygeal teratoma – a Swedish multicenter study
- 2019
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Ingår i: Journal of Pediatric Surgery. - : Elsevier BV. - 0022-3468 .- 1531-5037. ; 54:8, s. 1638-1643
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Tidskriftsartikel (refereegranskat)abstract
- Background/Purpose: The aims of this study were to evaluate bowel and urinary tract function, to identify predictors for dysfunctional outcome and to evaluate health-related quality-of life (QoL) in patients treated for sacrococcygeal teratomas (SCT). Methods: Medical records of patients with SCT born between 1985 and 2015 treated at three Swedish pediatric surgical centers were reviewed. Questionnaires regarding urinary tract function, bowel function and QoL were sent to patients and parents. Different QoL instruments were used for the different age groups. Results: Totally 85 patients were identified. Four patients died in the neonatal period. Forty-nine patients answered the questionnaires (60%). Median age at follow-up was 8.9 years (range 3.6–28.8). Bowel dysfunction was reported by 36% and urinary tract dysfunction by 46% of the patients. Univariate analysis revealed that urinary tract dysfunction correlated with gestational age (p = 0.018) and immature histology (p = 0.008), and bowel dysfunction correlated with gestational age (p = 0.016) and tumor size (p = 0.042). Low gestational age was an independent predictor for both urinary tract and bowel dysfunction. Good or very good QoL was reported by 56% of children aged 4–7 years, 90% of children aged 8–17 years and 67% of the adults. Conclusion: Although a considerable proportion of bowel and urinary tract dysfunction was found, the reported QoL was good in a majority of the patients with SCT. Low gestational age was found to be a predictor for bowel- and urinary tract dysfunction. Level of Evidence: Level III.
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| 6. |
- Hambraeus, Mette, et al.
(författare)
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Health-related quality of life and scar satisfaction in a cohort of children operated on for sacrococcygeal teratoma
- 2020
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Ingår i: Health and Quality of Life Outcomes. - : Springer Science and Business Media LLC. - 1477-7525. ; 18:1
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Tidskriftsartikel (refereegranskat)abstract
- Aim: The aims of this study were to evaluate health-related quality of life (HRQoL) in children with sacrococcygeal teratoma and to explore the effect of the scar on physical, emotional and behavioral aspects. Methods: A cohort of children operated on for sacrococcygeal teratoma between 2000 and 2013 at Lund University Hospital, Sweden, and their parents were interviewed. HRQoL was evaluated with PedsQL, and scar satisfaction was estimated through Patient Observer Scar Assessment Score (POSA). Results: All eligible children (n = 17) were included (100% response rate). Median age was 7.3 years (range 3.5-16.0). Mean total PedsQL score was 92.3 (range 72.0 to 99.0). Patients with comorbidity scored lower (87.5) than those without (95.0) (p < 0.05). Pain during sitting down was reported by two (20%) patients, and itching was reported by another two patients (20%) aged > 8 years. No children reported that they avoided situations due to the scar, and most (80% of children and 90% of parents) reported absent or only mild negative emotions when considering the scar. Conclusion: Children with sacrococcygeal teratoma had a good overall HRQoL, but comorbidity reduced the outcome. A few children reported scar-related impact on physical, behavioral and emotional aspects.
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| 7. |
- Hambraeus, Mette, et al.
(författare)
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Long-Term Outcome of Sacrococcygeal Teratoma : A Controlled Cohort Study of Urinary Tract and Bowel Dysfunction and Predictors of Poor Outcome
- 2018
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Ingår i: Journal of Pediatrics. - : Elsevier BV. - 0022-3476. ; 198, s. 2-136
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Tidskriftsartikel (refereegranskat)abstract
- Objective: To evaluate urinary tract and bowel function in children with sacrococcygeal teratoma, compare the findings with healthy children, and assess predictors of poor outcome. Study design: This was a controlled cohort study of all patients operated for sacrococcygeal teratoma at a tertiary pediatric surgery center, 2000-2013. Urinary and bowel function were compared with healthy control patients matched for age and sex. Perioperative and histopathologic risk factors were analyzed. Results: In total, 17 patients with sacrococcygeal teratoma and 85 healthy control patients were included in the study. Patients with sacrococcygeal teratoma more often were reported to have uncontrolled voiding (12% vs 0%, P <.01), difficulty in bladder emptying (24% vs 0%, P <.001), and pyelonephritis (18% vs 1%, P <.05). Constipation was more common in patients with sacrococcygeal teratoma (47 % vs 14%, P <.05), but the overall bowel function score was equal in the 2 groups. Children with large tumors and immature histology were more likely to have a dysfunctional outcome (P <.05). Conclusions: Uncontrolled voiding, difficulty in bladder emptying, pyelonephritis, and constipation were more common in patients with sacrococcygeal teratoma than in healthy children. Dysfunctional outcome was more prevalent in children with large and immature teratomas.
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| 8. |
- Hambraeus, Mette, et al.
(författare)
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Sacrococcygeal teratoma: A population-based study of incidence and prenatal prognostic factors.
- 2015
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Ingår i: Journal of Pediatric Surgery. - : Elsevier BV. - 1531-5037 .- 0022-3468.
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Tidskriftsartikel (refereegranskat)abstract
- Sacrococcygeal teratoma (SCT) is a rare congenital tumor associated with high rates of perinatal mortality and morbidity. This study evaluated the incidence, prenatal detection rate, and early predictors of a complicated outcome following diagnosis of SCT.
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| 9. |
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| 10. |
- Hofko Lindkvist, Emma, et al.
(författare)
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National survey of physicians in Swedish child health centres finds insecurity and unawareness regarding management of undescended testicles
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Ingår i: Acta Paediatrica, International Journal of Paediatrics. - 0803-5253.
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Tidskriftsartikel (refereegranskat)abstract
- Aim: To investigate underlying factors for previously reported shortcomings in child health centres' (CHC) referral process of boys with undescended testicles. Methods: A total of 386 physicians working at Swedish CHCs were surveyed regarding their knowledge about undescended testicles and their clinical management. Multivariate regression analyses were performed to identify risk factors of non-adherence to guidelines and self-reported lack of clinical skills. Results: The overall knowledge of the health benefits of undescended testicle surgery was high (89%), while two-thirds were unaware of surgery being recommended <1 year of age. One-fifth of respondents had never received guidance on examination techniques. Male gender (adjusted odds ratio [aOR] 0.51, 95% confidence interval [95% CI] 0.31–0.86), education in paediatrics (aOR 0.37, 95% CI 0.18–0.76) and more experience (aOR 0.02, 95% CI 0.01–0.09) significantly decreased the risk of unfamiliarity with examinations. More experience decreased the risk of stating the incorrect indications for undescended testicle surgery (aOR 0.17, 95% CI 0.03–0.95) and finding examinations difficult (aOR 0.22, 95% CI 0.07–0.72). Medical education outside Nordic countries was a risk factor for unawareness of guidelines (aOR 2.06, 95% CI 1.21–3.51). Conclusion: The knowledge and confidence level of the study population varied widely. The results indicate a need for further theoretical and practical education among Swedish CHC physicians.
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