Early resection of a rare congenital pulmonary airway malformation causing severe progressive respiratory distress in a preterm neonate: a case report and review of the literature.

BACKGROUND: Congenital pulmonary airway malformations (CPAMs) are a heterogenous collection of congenital lung malformations, often diagnosed prenatally. The Stocker Type III CPAM is a rare CPAM sub-type, and, when large, may be associated with hydrops. Furthermore, reports of CPAM management which may include surgical resection in extreme preterm infants are limited. CASE PRESENTATION: We report a case of a female neonate born at 28 weeks of gestation with severe respiratory distress and diffuse pulmonary opacification on the right concerning for a large congenital lung lesion. This lesion was not detected on routine antenatal imaging, and she did not have clinical findings of associated hydrops. Her respiratory status improved dramatically after surgical resection of a mass at 12 day of age. The mass was consistent pathologically with a Stocker Type III CPAM. Lung expansion showed subsequent improvement at 16 months of age. CONCLUSIONS: Our case describes a preterm neonate with severe respiratory distress that was found postnatally to have a large, unilateral congenital lung lesion despite a normal prenatal ultrasound. Additionally, this lesion required excision early in life due to severity of respiratory compromise. This case highlights that rare congenital lung lesions, like this rare sub-type of CPAM, should remain a diagnostic consideration in neonates with severe respiratory distress. Early lung resection for CPAM in preterm infants is not well described and the favorable outcomes of this case help expand perspectives on potential management strategies.

Randomized Phase II Trial of MIBG Versus MIBG, Vincristine, and Irinotecan Versus MIBG and Vorinostat for Patients With Relapsed or Refractory Neuroblastoma: A Report From NANT Consortium.

PURPOSE: 131I-metaiodobenzylguanidine (MIBG) is an active radiotherapeutic for neuroblastoma. The primary aim of this trial was to identify which of three MIBG regimens was likely associated with the highest true response rate. PATIENTS AND METHODS: Patients 1-30 years were eligible if they had relapsed or refractory neuroblastoma, at least one MIBG-avid site, and adequate autologous stem cells. Patients received MIBG 18 mCi/kg on day 1 and autologous stem cell on day 15. Patients randomly assigned to arm A received only MIBG; patients randomly assigned to arm B received intravenous vincristine on day 0 and irinotecan daily on days 0-4; patients randomly assigned to arm C received vorinostat (180 mg/m2/dose) orally once daily on days 1 to 12. The primary end point was response after one course by New Approaches to Neuroblastoma Therapy criteria. The trial was designed with 105 patients to ensure an 80% chance that the arm with highest response rate was selected. RESULTS: One hundred fourteen patients were enrolled, with three ineligible and six unevaluable, leaving 105 eligible and evaluable patients (36 in arm A, 35 in arm B, and 34 in arm C; 55 boys; and median age 6.5 years). After one course, the response rates (partial response or better) on arms A, B, and C were 14% (95% CI, 5 to 30), 14% (5 to 31), and 32% (18 to 51). An additional five, five, and four patients met New Approaches to Neuroblastoma Therapy Minor Response criteria on arms A, B, and C, respectively. On arms A, B, and C, rates of any grade 3+ nonhematologic toxicity after first course were 19%, 49%, and 35%. CONCLUSION: Vorinostat and MIBG is likely the arm with the highest true response rate, with manageable toxicity. Vincristine and irinotecan do not appear to improve the response rate to MIBG and are associated with increased toxicity.

Meniscus-Targeted Injections for Chronic Knee Pain Due to Meniscal Tears or Degenerative Fraying: A Retrospective Study.

OBJECTIVES: Meniscal tears caused by acute trauma or degenerative fraying affect a wide array of individuals. An effective, long-lasting treatment has widely been sought after. Intra-articular corticosteroid injections have been among the methods of controlling pain for more than 60 years. However, such injections tend to produce short-lasting results, with profound effects lasting an average of up to 4 weeks. The purpose of this study was to determine the average duration and magnitude of pain relief after meniscal-targeted injections. METHODS: The electronic medical records of 135 patients were accessed for this retrospective chart review. Patients who had meniscal tears or degenerative fraying and were treated with meniscal-targeted injections were selected. Patients' visual analog scale (VAS) pain scores (before and after treatment) were recorded, along with the percentage of pain relief and duration of pain relief. RESULTS: Ultrasound-guided meniscus-targeted corticosteroid injections for meniscal tears or degenerative fraying produced 5.68 (SD, 5.28) weeks of pain relief on average, with a decrease in pain from initial to follow-up visits of 2.14 (P < .0001) as per the visual analog scale score, and an Integral of Pain Relief score of 3.98. CONCLUSIONS: Our findings indicate a substantial benefit from 20- or 40-mg meniscus-targeted triamcinolone injections, granted the limitations of chart review research and no control group comparison. Results highlight the need for future prospective research comparing meniscus-targeted injections with intra-articular injections to identify a better modality for treating patients with chronic knee pain caused by meniscal tears or degenerative fraying.