Left vocal cord paralysis, lung function and exercise capacity in young adults born extremely preterm with a history of neonatal patent ductus arteriosus surgery—A national cohort study

Background: Left vocal cord paralysis (LVCP) is a known complication of patent ductus arteriosus (PDA) surgery in extremely preterm (EP) born neonates; however, consequences of LVCP beyond the first year of life are insufficiently described. Both voice problems and breathing difficulties during physical activity could be expected with an impaired laryngeal inlet. More knowledge may improve the follow-up of EP-born subjects who underwent PDA surgery and prevent confusion between LVCP and other diagnoses. Objectives: Examine the prevalence of LVCP in a nationwide cohort of adults born EP with a history of PDA surgery, and compare symptoms, lung function, and exercise capacity between groups with and without LVCP, and vs. controls born EP and at term. Methods: Adults born EP (<28 weeks' gestation or birth weight <1,000 g) in Norway during 1999–2000 who underwent neonatal PDA surgery and controls born EP and at term were invited to complete questionnaires mapping voice-and respiratory symptoms, and to perform spirometry and maximal treadmill exercise testing. In the PDA-surgery group, exercise tests were performed with a laryngoscope positioned to evaluate laryngeal function. Results: Thirty out of 48 (63%) eligible PDA-surgery subjects were examined at mean (standard deviation) age 19.4 (0.8) years, sixteen (53%) had LVCP. LVCP was associated with self-reported voice symptoms and laryngeal obstruction during exercise, not with lung function or peak oxygen consumption (VO2peak). In the PDA-surgery group, forced expiratory volume in 1 second z-score (z-FEV1) was reduced compared to EP-born controls (n = 30) and term-born controls (n = 36); mean (95% confidence interval) z-FEV1 was −1.8 (−2.3, −1.2), −0.7 (−1.1, −0.3) and −0.3 (−0.5, −0.0), respectively. For VO2peak, corresponding figures were 37.5 (34.9, 40.2), 38.1 (35.1, 41.1), and 43.6 (41.0, 46.5) ml/kg/min, respectively. Conclusions: LVCP was common in EP-born young adults who had undergone neonatal PDA surgery. Within the PDA-surgery group, LVCP was associated with self-reported voice symptoms and laryngeal obstruction during exercise, however we did not find an association with lung function or exercise capacity. Overall, the PDA-surgery group had reduced lung function compared to EP-born and term-born controls, whereas exercise capacity was similarly reduced for both the PDA-surgery and EP-born control groups when compared to term-born controls.publishedVersio

Total hip replacement in young adults with hip dysplasia: Age at diagnosis, previous treatment, quality of life, and validation of diagnoses reported to the Norwegian Arthroplasty Register between 1987 and 2007

Background and purpose: Dysplasia of the hip increases the risk of secondary degenerative change and subsequent total hip replacement. Here we report on age at diagnosis of dysplasia, previous treatment, and quality of life for patients born after 1967 and registered with a total hip replacement due to dysplasia in the Norwegian Arthroplasty Register. We also used the medical records to validate the diagnosis reported by the orthopedic surgeon to the register. Methods: Subjects born after January 1, 1967 and registered with a primary total hip replacement in the Norwegian Arthroplasty Register during the period 1987–2007 (n = 713) were included in the study. Data on hip symptoms and quality of life (EQ-5D) were collected through questionnaires. Elaborating information was retrieved from the medical records. Results: 540 of 713 patients (76%) (corresponding to 634 hips) returned the questionnaires and consented for additional information to be retrieved from their medical records. Hip dysplasia accounted for 163 of 634 hip replacements (26%), 134 of which were in females (82%). Median age at time of diagnosis was 7.8 (0–39) years: 4.4 years for females and 22 years for males. After reviewing accessible medical records, the diagnosis of hip dysplasia was confirmed in 132 of 150 hips (88%). Interpretation: One quarter of hip replacements performed in patients aged 40 or younger were due to an underlying hip dysplasia, which, in most cases, was diagnosed during late childhood. The dysplasia diagnosis reported to the register was correct for 88% of the hips

Hip dysplasia in young adults

According to data held by the Medical Birth Registry of Norway, approximately 1% of all newborns in Norway are diagnosed annually with hip instability at birth. Abduction treatment (Frejka pillow) for 6-12 weeks is the standard treatment in Norway, with which the majority will develop normal hips. Additionally to those detected by the neonatal screening program, some are late-diagnosed cases (diagnosis >1 month of age). For this group, the treatment is usually more demanding and prolonged. The older the child is at start of treatment, the poorer is the prognosis. A dysplastic hip causes altered mechanical conditions, predisposing for increased wear of the cartilage and development of osteoarthritis of the hip in young adult age. The final treatment option for this condition could be a total hip replacement. In Paper I, we linked two national registries, the Norwegian Arthroplasty Register and the Medical Birth Registry of Norway, using the unique national identification number. The Birth Registry contains information on all newborns in Norway from 1967 and the Arthroplasty Register includes all total hip replacements inserted in Norway from 1987. The study found a 2.6 times increased risk for a total hip replacement in young adulthood for patients reported with hip instability in the newborn period. The absolute risk was however low at only 57 in 105 for patients with hip instability compared to 20 in 105 for those with stable hips. Only 8% of those who underwent a total hip replacement due to hip dysplasia were reported to have had instable hips at birth. In Paper II, we validated the dysplasia diagnosis reported to the Norwegian Arthroplasty Register for subjects born after 1967. Medical records were reviewed and we also investigated age at dysplasia diagnosis, previous treatment and quality of life. We found the dysplasia diagnosis reported to the Arthroplasty Register to be correct in 88% of the hips. Median age at time of diagnosis was as high as 7.8 years: 4.4 years for females and 22 years for males. 75% of the patients had undergone different hip-preserving treatments before their prosthesis, and the dysplasia patients scored poorer in quality of life (EQ-5D) compared to the age-matched general population in Sweden and the UK. In Paper III, we aimed to validate a digital measurement programme for hip dysplasia at skeletal maturity. Ninety-five radiographs were measured by three independent observers in both a newly developed digital measurement programme and manually in AgfaWeb1000. Eleven radiological measurements, all relevant for hip dysplasia at skeletal maturity, were evaluated. We found acceptable inter- and intra-observer reproducibility for most measurements, but with poorer accuracy for measurements with small absolute values. The reproducibility was relatively similar for the two methods used, but the digital measurements were performed much faster. In Paper IV, we used data from the 1989 Hip Project and reported on the prevalence of hip dysplasia in 2081 19-year old Norwegians. The prevalence of hip dysplasia in the cohort varied from 1.7% to 20% depending on the radiological measurement used. A Wiberg’s angle <20° was seen in 3.3% of the cohort: 4.3% in women and 2.4% in men. We found no association between subjects with radiological signs indicating hip dysplasia and body mass index (BMI), Beighton hypermobility score, EQ-5D score or WOMAC score. The overall conclusions of this thesis are as follows: About 25% of all total hip replacements in young adults (< 40 years) are performed due to an underlying hip dysplasia. The dysplasia diagnosis is in general detected late, indicating that clinical testing for hip instability in newborns is an insufficient screening method to detect hips that require a total hip replacement in young adulthood. Several radiographic measurements for hip dysplasia are proposed in the literature. The reproducibility for these measurements varies, but with acceptable results for the more common measurements such as Wiberg’s centre-edge and Sharp’s acetabular angle. The prevalence of hip dysplasia is highly dependent on the radiographic measurements used, but a high prevalence for some of the measurements is found in skeletally mature Norwegians as compared to other studies on Caucasians in the literature

Implant survival and radiographic outcome of total hip replacement in patients less than 20 years old

Background and purpose — Total hip replacement (THR) is not recommended for children and very young teenagers because early and repetitive revisions are likely. We investigated the clinical and radiographic outcomes of THR performed in children and teenage patients. Patients and methods — We included 111 patients (132 hips) who underwent THR before 20 years of age. They were identified in the Norwegian Arthroplasty Register, together with information on the primary diagnosis, types of implants, and any revisions that required implant change. Radiographs and Harris hip score (HHS) were also evaluated. Results — The mean age at primary THR was 17 (11–19) years and the mean follow-up time was 14 (3–26) years. The 10-year survival rate after primary THR (with the endpoint being any revision) was 70%. 39 patients had at least 1 revision and 16 patients had 2 or more revisions. In the latest radiographs, osteolysis and atrophy were observed in 19% and 27% of the acetabulae and 21% and 62% of the femurs, respectively. The mean HHS at the final follow-up was 83 (15–100). Interpretation — The clinical score after THR in these young patients was acceptable, but many revisions had been performed. However, young patients with developmental dysplasia of the hip had lower implant survival. Moreover, the bone stock in these patients was poor, which could complicate future revisions

Severe exercise-induced laryngeal obstruction treated with supraglottoplasty

Introduction: Exercise induced laryngeal obstruction (EILO) is relatively common in adolescents, with symptoms often confused with exercise induced asthma. EILO often starts with medial or inward rotation of supraglottic structures of the larynx, whereas glottic adduction appears as a secondary phenomenon in a majority. Therefore, surgical treatment (supraglottoplasty) is used in thoroughly selected and highly motivated patients with pronounced symptoms and severe supraglottic collapse. Aim: To investigate efficacy and safety of laser supraglottoplasty as treatment for severe supraglottic EILO by retrospective chart reviews. Methods: The EILO register at Haukeland University Hospital, Bergen, Norway was used to identify patients who had undergone laser supraglottoplasty for severe supraglottic EILO, verified by continuous laryngoscopy exercise (CLE) test, during 2013-2015. Laser incision in both aryepiglottic folds anterior to the cuneiform tubercles and removal of the mucosa around the top was performed in general anesthesia. Outcomes were questionnaire based self-reported symptoms, and laryngeal obstruction scored according to a defined scheme during a CLE-test performed before and after surgery. Results: Forty-five of 65 eligible patients, mean age 15.9 years, were included. Post-operatively, 38/45 (84%) patients reported less symptoms, whereas CLE-test scores had improved in all, of whom 16/45 (36%) had no signs of obstruction. Most improvements were at the supraglottic level, but 21/45 (47%) also improved at the glottic level. Two of 65 patients had complications; self-limiting vocal fold paresis and scarring/shortening of plica ary-epiglottica. Conclusion: Supraglottoplasty improves symptoms and decreases laryngeal obstruction in patients with severe supraglottic EILO, and appears safe in highly selected cases.Major funding institutions: Haukeland University Hospital and University of Bergen.publishedVersio

Severe exercise-induced laryngeal obstruction treated with supraglottoplasty

Introduction: Exercise induced laryngeal obstruction (EILO) is relatively common in adolescents, with symptoms often confused with exercise induced asthma. EILO often starts with medial or inward rotation of supraglottic structures of the larynx, whereas glottic adduction appears as a secondary phenomenon in a majority. Therefore, surgical treatment (supraglottoplasty) is used in thoroughly selected and highly motivated patients with pronounced symptoms and severe supraglottic collapse. Aim: To investigate efficacy and safety of laser supraglottoplasty as treatment for severe supraglottic EILO by retrospective chart reviews. Methods: The EILO register at Haukeland University Hospital, Bergen, Norway was used to identify patients who had undergone laser supraglottoplasty for severe supraglottic EILO, verified by continuous laryngoscopy exercise (CLE) test, during 2013–2015. Laser incision in both aryepiglottic folds anterior to the cuneiform tubercles and removal of the mucosa around the top was performed in general anesthesia. Outcomes were questionnaire based self-reported symptoms, and laryngeal obstruction scored according to a defined scheme during a CLE-test performed before and after surgery. Results: Forty-five of 65 eligible patients, mean age 15.9 years, were included. Post-operatively, 38/45 (84%) patients reported less symptoms, whereas CLE-test scores had improved in all, of whom 16/45 (36%) had no signs of obstruction. Most improvements were at the supraglottic level, but 21/45 (47%) also improved at the glottic level. Two of 65 patients had complications; self-limiting vocal fold paresis and scarring/shortening of plica ary-epiglottica. Conclusion: Supraglottoplasty improves symptoms and decreases laryngeal obstruction in patients with severe supraglottic EILO, and appears safe in highly selected cases

Severe exercise-induced laryngeal obstruction treated with supraglottoplasty

Introduction: Exercise induced laryngeal obstruction (EILO) is relatively common in adolescents, with symptoms often confused with exercise induced asthma. EILO often starts with medial or inward rotation of supraglottic structures of the larynx, whereas glottic adduction appears as a secondary phenomenon in a majority. Therefore, surgical treatment (supraglottoplasty) is used in thoroughly selected and highly motivated patients with pronounced symptoms and severe supraglottic collapse. Aim: To investigate efficacy and safety of laser supraglottoplasty as treatment for severe supraglottic EILO by retrospective chart reviews. Methods: The EILO register at Haukeland University Hospital, Bergen, Norway was used to identify patients who had undergone laser supraglottoplasty for severe supraglottic EILO, verified by continuous laryngoscopy exercise (CLE) test, during 2013–2015. Laser incision in both aryepiglottic folds anterior to the cuneiform tubercles and removal of the mucosa around the top was performed in general anesthesia. Outcomes were questionnaire based self-reported symptoms, and laryngeal obstruction scored according to a defined scheme during a CLE-test performed before and after surgery. Results: Forty-five of 65 eligible patients, mean age 15.9 years, were included. Post-operatively, 38/45 (84%) patients reported less symptoms, whereas CLE-test scores had improved in all, of whom 16/45 (36%) had no signs of obstruction. Most improvements were at the supraglottic level, but 21/45 (47%) also improved at the glottic level. Two of 65 patients had complications; self-limiting vocal fold paresis and scarring/shortening of plica ary-epiglottica. Conclusion: Supraglottoplasty improves symptoms and decreases laryngeal obstruction in patients with severe supraglottic EILO, and appears safe in highly selected cases

Severe exercise-induced laryngeal obstruction treated with supraglottoplasty

Introduction: Exercise induced laryngeal obstruction (EILO) is relatively common in adolescents, with symptoms often confused with exercise induced asthma. EILO often starts with medial or inward rotation of supraglottic structures of the larynx, whereas glottic adduction appears as a secondary phenomenon in a majority. Therefore, surgical treatment (supraglottoplasty) is used in thoroughly selected and highly motivated patients with pronounced symptoms and severe supraglottic collapse. Aim: To investigate efficacy and safety of laser supraglottoplasty as treatment for severe supraglottic EILO by retrospective chart reviews. Methods: The EILO register at Haukeland University Hospital, Bergen, Norway was used to identify patients who had undergone laser supraglottoplasty for severe supraglottic EILO, verified by continuous laryngoscopy exercise (CLE) test, during 2013-2015. Laser incision in both aryepiglottic folds anterior to the cuneiform tubercles and removal of the mucosa around the top was performed in general anesthesia. Outcomes were questionnaire based self-reported symptoms, and laryngeal obstruction scored according to a defined scheme during a CLE-test performed before and after surgery. Results: Forty-five of 65 eligible patients, mean age 15.9 years, were included. Post-operatively, 38/45 (84%) patients reported less symptoms, whereas CLE-test scores had improved in all, of whom 16/45 (36%) had no signs of obstruction. Most improvements were at the supraglottic level, but 21/45 (47%) also improved at the glottic level. Two of 65 patients had complications; self-limiting vocal fold paresis and scarring/shortening of plica ary-epiglottica. Conclusion: Supraglottoplasty improves symptoms and decreases laryngeal obstruction in patients with severe supraglottic EILO, and appears safe in highly selected cases