Oxygen desaturation and time burden during tracheobronchial endoscopy for suspected foreign body in toddlers.

Tracheobronchial endoscopy with general anaesthesia for suspected foreign body aspiration exposes toddlers to acute hypoxemia. Better quantification of intraoperative hypoxemia could help identify and manage the most severe patients. We explored the hypoxic burden approach to account for both duration and depth of desaturation episodes during the procedure and determined risk factors for high hypoxic burden. We retrospectively analysed tracheobronchial endoscopies performed from September 2015 to September 2018 in children ≤ 36 months for suspected foreign body aspiration, in two French university hospitals. The hypoxic burden (area under 90% of the SpO2/time curve) was calculated. The median of non-zero burdens was used to delineate a subgroup with high hypoxic burden. Risk factors for high hypoxic burden were identified using multivariable analysis. Of 96 procedures, 56 (58%) were associated with at least one SpO2 value < 90%. Of them, the median [interquartile] hypoxic burden was 25 [5-87] %.min. Bradycardia < 100 bpm occurred in 11 procedures (11%). Initial admission to general hospitals (OR 0.23, 95% CI 0.06-0.86) and airway anaesthesia with topical lidocaine (OR 0.15, 95% CI 0.03-0.62) were associated with a reduced risk of high hypoxic burden. High hypoxic burden was associated with an increased risk of postoperative invasive ventilation (OR 32, 95% CI 1.7-617) and of hospital stay > 24 h (OR 4.0, 95% CI 1.6-10). No postoperative neurological sequelae were found. The hypoxic burden approach, when applied in tracheobronchial endoscopy for suspected foreign body aspiration in toddlers, enabled the quantification of hypoxemia and the search for specific risk factors.

Gas exchange parameters for the prediction of obstructive sleep apnea in infants.

STUDY OBJECTIVES: Sleep laboratory polysomnography is the gold standard for obstructive sleep apnea (OSA) diagnosis in infants, but its access remains limited. Oximetry-capnography is another simple and widely used tool that can provide information on the presence of desaturations and alveolar hypoventilation. However, its reliability is debated. This study aimed at examining its use in determining OSA severity in infants. METHODS: This retrospective study was conducted in a sleep unit in a tertiary hospital in infants < 4 months old with clinical signs of OSA or Pierre Robin sequence who underwent a 1-night polysomnography coupled with oximetry-capnography. RESULTS: Among the 78 infants included (median [interquartile range] age: 61 [45-89] days at polysomnography), 44 presented with Pierre Robin sequence and 34 presented with isolated airway obstruction. The clinical, sleep, and respiratory characteristics were not significantly different between the 2 subgroups. In the entire cohort, 63.5% had severe OSA. The median obstructive apnea-hypopnea index was 14.5 (7.4-5.9) events/h, peripheral oxygen saturation (SpO2) was 97.4% (96.5-98.1%), and transcutaneous carbon dioxide pressure (PtcCO2) was 41.1 mmHg (38.3-44.9). The optimal threshold to predict an obstructive apnea-hypopnea index > 10 events/h was 6 events/h for an oxygen desaturation index ≥ 3% (sensitivity, 95.7%; specificity, 51.9%) and 2 events/h for an oxygen desaturation index ≥ 4% (sensitivity, 95.7%; specificity, 48.1%). CONCLUSIONS: Whereas transcutaneous capnography does not appear to be sufficient in predicting severe OSA in infants < 4 months old with Pierre Robin sequence or clinical signs of OSA, oximetry may be a useful alternative for the screening of severe OSA in infants in the absence of polysomnography. CITATION: Gyapay R, Ioan I, Thieux M, et al. Gas exchange parameters for the prediction of obstructive sleep apnea in infants. J Clin Sleep Med. 2024;20(7):1059-1067.

Early Adjuvant Medication With the mTOR Inhibitor Sirolimus in a Preterm Neonate With Compressive Cystic Lymphatic Malformation.

Cystic lymphatic malformations result from an abnormal embryological development of the lymphatic structures. Here we report on a case of a preterm female baby, born at 34 weeks of gestation, with a voluminous cervicofacial cystic lymphatic malformation responsible for an airway obstruction. An mTOR inhibitor, sirolimus, was started from the first day of life, and was combined with iterative sclerotherapy procedures. This case illustrates a safe and successful early administration of sirolimus in a preterm neonate.

Management of postcricoid and upper esophageal hemangioma.

To discuss management of postcricoid and upper esophageal hemangiomas in infants. Four children presenting with progressive feeding and/or respiratory disturbance underwent endoscopy including systematic esophagoscopy revealing the hemangioma of the postcricoid area and extending to the lumen of the upper esophagus. In the three children with severe disturbance, systemic steroids were insufficient and open surgical excision brought the hemangioma under control. No significant stenosis occurred despite prolonged progressive oral feeding up to 1 month after surgery. The fourth child whose disturbance was moderate, was managed by intralesional steroids. Various solutions have been proposed for the treatment, i.e. conservative approach or partial or complete destruction or excision, management of postcricoid hemangioma by intralesional steroids or by open surgical excision. Indications will require greater series.

Natural history of adult-onset laryngeal papillomatosis following multiple cidofovir injections.

OBJECTIVES: A prospective study was performed to assess the intermediate and long-term efficacy of intralesional cidofovir therapy associated with surgical excision in laryngeal papillomatosis in adults. METHODS: Endoscopy with intralesional injection of cidofovir 5 mg/mL was performed 3 times at 4-week intervals. The concentration was later increased to 7.5 mg/mL and the interval between injections shortened to 2 weeks. Further treatment was performed at 3 or 6 months, depending on the evolution of the papillomas. After complete remission, the treatment was stopped and the patients were reviewed every 6 months. RESULTS: Nineteen patients completed the protocol, with a mean of 4.5 injections each. Complete remission was obtained in 17 cases (89%) after a mean of 3.8 procedures. Remission was stable after a mean follow-up of 24 months (range, 8 to 57 months). With higher cidofovir concentrations at shorter intervals, patients needed fewer injections to achieve remission (mean, 2.1 versus 4.7 injections). CONCLUSIONS: The effectiveness of intralesional cidofovir therapy in adult-onset recurrent respiratory papillomatosis was impressive. Once obtained, complete remission was stable on intermediate or long-term follow-up. The concentration and the interval between injections seemed to influence the number of injections necessary to achieve remission.

Intermediate-term and long-term results after treatment by cidofovir and excision in juvenile laryngeal papillomatosis.

OBJECTIVES: We assessed the intermediate-term and long-term efficacy of intralesional injection of cidofovir used with surgical excision in juvenile-onset recurrent respiratory papillomatosis. METHODS: The protocol was revised during the study, from endoscopy at 4-week intervals with intralesional injection of cidofovir at 5 mg/mL, to a 2-week interval and a 7.5-mg/mL dosage. Partial surgical excision of hypertrophic papillomas was performed before the initiation of injection. Further injections at 4-week (or 2-week) intervals were performed until complete remission. RESULTS: Sixteen patients received a mean 8.9 injections. Complete remission was obtained in 12 patients (75%) after a mean 7.2 endoscopic treatments. Remission was stable at a mean 33.6 months' follow-up. Five of these 12 patients received 5.2 injections and remained disease-free at a mean 39.3 months' follow-up. Seven of the 12 had 1 relapse; they needed complementary treatment to become disease-free, and remained so thereafter over a mean 27.3 months' follow-up. The other 4 of the 16 patients (25%) continued to present active disease. CONCLUSIONS: Active endoscopic treatment until complete remission led to a higher-than-expected complete remission rate on intermediate-term to long-term follow-up, with or without relapse. Transient relapse was associated with a long delay in initiating cidofovir treatment.

Cidofovir plasma assays after local injection in respiratory papillomatosis.

OBJECTIVE: To assess cidofovir plasma concentration after intralesional airway administration for recurrent respiratory papillomatosis. DESIGN: Prospective study. SETTING: Tertiary care teaching hospital. PATIENTS AND METHOD: The study comprised 21 patients (10 children and 11 adults). Plasma samples were collected at 10 and 45 minutes (T10, T45) or at 10 and 60 minutes (T10, T60) after injection. The measurements of cidofovir were performed using a high-performance liquid chromatographic method. RESULTS: Plasma samples were collected at T10 and T45 on 19 occasions from the children and on 17 from the adults. A linear relationship was found between plasma concentration and dose in children (mean dose 1.2 mg/kg; mean cidofovir plasma levels 0.91 and 0.81 microg/mL) but not in adults (mean dose 0.2 mg/kg; mean plasma levels 0.21 and 0.31 microg/mL). The same relationships were found between dose and area under the concentration/time curve (AUC). Four plasma samples were taken in children at T10 and T60: mean dose 1.2 mg/kg and mean plasma concentrations 1.11 and 1.24 microg/mL. Maximum plasma concentration averaged 34% (SD 11%) in children and 62% (SD 33%) in adults, with equivalent plasma level after intravenous infusion of the same dose. CONCLUSIONS: The cidofovir plasma levels were below those leading to toxicity. The levels and the AUC were dose dependent in children but not in adults. Diffusion from the injected site was greatest in a few adults and unpredictable. Because of the great individual variation in diffusion in adults, cidofovir should be used at less than the recommended intravenous dose to prevent any risk of systemic toxicity.

Controlled risk of stenosis after surgical excision of laryngeal hemangioma.

OBJECTIVE: To evaluate the risk of subglottic stenosis after surgical excision of congenital subglottic hemangioma. DESIGN: Retrospective analysis and case series. SETTING: Tertiary care teaching hospital. PATIENTS: A total of 13 pediatric patients diagnosed with subglottic hemangioma with unilateral, bilateral, or circular lesions and more than 50% airway obstruction between 1992 and 2001. INTERVENTION: Open surgical excision was performed as a single-stage procedure either as primary or secondary intention. The cricoid cartilage was left open at the end of the procedure. Postoperative intubation was carried out in a pediatric intensive care unit. MAIN OUTCOME MEASURE: An adequate airway after surgical excision. RESULTS: All patients were successfully extubated. No recurrence was noted. Three patients developed subglottic stenosis, two grade 1 and one grade 2. All 3 showed a favorable outcome and did not require reintubation. One needed endoscopic management of the stenosis. Of these 3 cases, 2 occurred after carbon dioxide laser treatment (out of 3) and 1 after circumferential dissection (out of 3). CONCLUSIONS: Extubation after surgery was successful in all cases of subglottic hemangioma. Risk of subglottic stenosis was limited and occurred only after circumferential dissection, especially if associated with prior traumatic laser damage of the hemangioma.

CT-assisted surgery in choanal atresia.

OBJECTIVE: To limit restenosis after surgical repair of choanal atresia. MATERIAL AND METHODS: A total of 20 children (age range 3 days to 11 years) with choanal atresia underwent surgical repair using a microdebrider via an intranasal approach. Surgery was guided by CT. Mitomycin was administered at the end of the procedure. RESULTS: An average of 1.7 procedures per child were required. CT combined with peroperative endoscopy revealed postoperative neo-osteogenesis and insufficient bone resection. CONCLUSIONS: CT-assisted intranasal repair led to extended bone resection of the atretic plate, the predominant cause of which was restenosis resulting from neo-osteogenesis.

Propranolol in the therapeutic strategy of infantile laryngotracheal hemangioma: A preliminary retrospective study of French experience.

OBJECTIVE: Preliminary assessment of the efficacy of propranolol on subglottic hemangioma in children on a nation-wide scale. METHODS: Multicentric, retrospective study of clinical files of 14 children; pre- and post-treatment endoscopies. RESULTS: Mean age at diagnosis was 2.3 (0.7-4) months. Mean percentage of airway obstruction was 68% (15-90) before propranolol introduction. Propranolol was started at 5.2 (0.7-16) months of age. This treatment was effective in all cases with a mean regression of the stenosis to 22% after 2 weeks and 12% after 4 weeks. Other medical treatments (steroids) could be stopped. In one patient, a side effect of propranolol motivated the switch to another ß-blocker. In four patients, treatment was stopped after 5.2 (1-10) months with a relapse in 2 (50%) cases. One of these two patients developed a resistance to propranolol and required a surgical procedure by external approach. CONCLUSION: This preliminary nation-wide survey confirms propranolol high effectiveness against airways' localization of infantile hemangiomas. Propranolol also allows alleviation or cut-off of previous medical treatments. However, recurrences are possible after early treatment interruption.