Downloaded from http://fn.bmj.com/ on July 13, 2015 - Published by group.bmj.com
ADC-FNN Online First, published on June 30, 2015 as 10.1136/archdischild-2015-308529 Images in neonatal medicine
An unexpected cause of stridor in a newborn CASE REPORT A non-dysmorphic term boy presented with inspiratory stridor shortly following delivery. Following clinical examination and thoracic x-ray, no external features or other clinical findings revealed any obvious explanation. Laryngomalacia was suspected. A combined rhinopharyngo—oropharyngo—and laryngoscopy revealed a cystic mass stretching from nasopharynx, to oropharynx and distally beyond the postcricoid structures, filling out 2/3 of the oropharyngeal space. The neck mass increased in size and became externally visible at day 9. MRI showed a multilocular cystic mass measuring 5.8×5.2×4.81 cm to the right side of hypopharynx stretching in the pre-vertebral and retropharyngeal direction (figures 1 and 2). The boy underwent picibanil sclerotherapy (OK432), which 8 months after birth resulted in near total resolution of the lesion.
DISCUSSION Cystic hygroma occurs in approximately 1/6000 live births, and situ of these lesions are predominantly in the neck region.1 2 They are usually detected prenatally or are externally visible after birth. In this patient, no external clinical signs gave rise to suspicion of a congenital abnormality. As this condition may lead to a rapid clinical deterioration, a progressive attitude towards early diagnostic measures should be undertaken in order to find the cause and initiate adequate therapeutics. As intervention in order to secure airway is of high priority and may cause unsuspected difficulties, a planned team approach with capability of potential surgical intervention should be available.
Figure 2 Cor T2 showing cystic hygroma of the neck (arrowheads) and opening to larynx (arrow).
Oddvar Uleberg,1,2 Bjørn Myklebust,3 Jana Rydland,4 Ragnhild Støen5,6 1
Department of Emergency Medicine and Pre-hospital Services, St Olav’s University Hospital, Trondheim, Norway 2 Department of Circulation and Medical Imaging, Norwegian University of Science and Technology (NTNU), Trondheim, Norway 3 Department of Paediatrics, Levanger County Hospital, Levanger, Norway 4 Department of Diagnostic Imaging, St Olav’s University Hospital, Trondheim, Norway 5 Department of Laboratory Medicine, Children’s and Women’s Health, Norwegian University of Science and Technology (NTNU), Trondheim, Norway 6 Department of Paediatrics, St Olav’s University Hospital, Trondheim, Norway Correspondence to Dr Oddvar Uleberg, Department of Emergency medicine and Pre-hospital services, Department of Circulation and Medical Imaging, Norwegian University of Science and Technology (NTNU), Norway, St. Olav’s University Hospital/ AHL, Prinsesse Kristinas gate, Trondheim N-7006, Norway; [email protected] Twitter Follow Oddvar Uleberg at @uleodd Acknowledgements We would like to gratefully acknowledge the parents for granting us permission to publish this report.
Figure 1 Sag T2 showing a large cystic hygroma (arrowheads) and narrow larynx in front (arrow).
Contributors OU and RS contributed to acquisition of case details. OU wrote the first draft of the manuscript. JR supplied the diagnostic images. OU, BM, JR and RS revised the manuscript. All authors have given final approval of this version to be published.
Uleberg O, et al. Arch Dis Child Fetal Neonatal Ed Month 2015 Vol 0 No 0
F1
Copyright Article author (or their employer) 2015. Produced by BMJ Publishing Group Ltd (& RCPCH) under licence.
Downloaded from http://fn.bmj.com/ on July 13, 2015 - Published by group.bmj.com
Images in neonatal medicine Competing interests None declared. Patient consent Obtained. Provenance and peer review Not commissioned; internally peer reviewed. To cite Uleberg O, Myklebust B, Rydland J, et al. Arch Dis Child Fetal Neonatal Ed Published Online First: [ please include Day Month Year] doi:10.1136/archdischild2015-308529 Received 3 March 2015 Revised 27 May 2015 Accepted 8 June 2015
F2
Arch Dis Child Fetal Neonatal Ed 2015;0:F1–F2. doi:10.1136/archdischild-2015-308529
REFERENCES 1 2
Sannoh S, Quezada E, Merer D, et al. Cystic hygroma and potential airway obstruction in a newborn; a case report and review of the literature. Cases J 2009;2:48. Song TB, Kim CH, Kim SM, et al. Fetal axillary cystic hygroma detected by prenatal ultrasonography: a case report. J Korean Med Sci 2002;17:400–2.
Uleberg O, et al. Arch Dis Child Fetal Neonatal Ed Month 2015 Vol 0 No 0
Downloaded from http://fn.bmj.com/ on July 13, 2015 - Published by group.bmj.com
An unexpected cause of stridor in a newborn Oddvar Uleberg, Bjørn Myklebust, Jana Rydland and Ragnhild Støen Arch Dis Child Fetal Neonatal Ed published online June 30, 2015
Updated information and services can be found at: http://fn.bmj.com/content/early/2015/06/30/archdischild-2015-308529
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Notes
To request permissions go to: http://group.bmj.com/group/rights-licensing/permissions To order reprints go to: http://journals.bmj.com/cgi/reprintform To subscribe to BMJ go to: http://group.bmj.com/subscribe/
ADC-FNN Online First, published on June 30, 2015 as 10.1136/archdischild-2015-308529 Images in neonatal medicine
An unexpected cause of stridor in a newborn CASE REPORT A non-dysmorphic term boy presented with inspiratory stridor shortly following delivery. Following clinical examination and thoracic x-ray, no external features or other clinical findings revealed any obvious explanation. Laryngomalacia was suspected. A combined rhinopharyngo—oropharyngo—and laryngoscopy revealed a cystic mass stretching from nasopharynx, to oropharynx and distally beyond the postcricoid structures, filling out 2/3 of the oropharyngeal space. The neck mass increased in size and became externally visible at day 9. MRI showed a multilocular cystic mass measuring 5.8×5.2×4.81 cm to the right side of hypopharynx stretching in the pre-vertebral and retropharyngeal direction (figures 1 and 2). The boy underwent picibanil sclerotherapy (OK432), which 8 months after birth resulted in near total resolution of the lesion.
DISCUSSION Cystic hygroma occurs in approximately 1/6000 live births, and situ of these lesions are predominantly in the neck region.1 2 They are usually detected prenatally or are externally visible after birth. In this patient, no external clinical signs gave rise to suspicion of a congenital abnormality. As this condition may lead to a rapid clinical deterioration, a progressive attitude towards early diagnostic measures should be undertaken in order to find the cause and initiate adequate therapeutics. As intervention in order to secure airway is of high priority and may cause unsuspected difficulties, a planned team approach with capability of potential surgical intervention should be available.
Figure 2 Cor T2 showing cystic hygroma of the neck (arrowheads) and opening to larynx (arrow).
Oddvar Uleberg,1,2 Bjørn Myklebust,3 Jana Rydland,4 Ragnhild Støen5,6 1
Department of Emergency Medicine and Pre-hospital Services, St Olav’s University Hospital, Trondheim, Norway 2 Department of Circulation and Medical Imaging, Norwegian University of Science and Technology (NTNU), Trondheim, Norway 3 Department of Paediatrics, Levanger County Hospital, Levanger, Norway 4 Department of Diagnostic Imaging, St Olav’s University Hospital, Trondheim, Norway 5 Department of Laboratory Medicine, Children’s and Women’s Health, Norwegian University of Science and Technology (NTNU), Trondheim, Norway 6 Department of Paediatrics, St Olav’s University Hospital, Trondheim, Norway Correspondence to Dr Oddvar Uleberg, Department of Emergency medicine and Pre-hospital services, Department of Circulation and Medical Imaging, Norwegian University of Science and Technology (NTNU), Norway, St. Olav’s University Hospital/ AHL, Prinsesse Kristinas gate, Trondheim N-7006, Norway; [email protected] Twitter Follow Oddvar Uleberg at @uleodd Acknowledgements We would like to gratefully acknowledge the parents for granting us permission to publish this report.
Figure 1 Sag T2 showing a large cystic hygroma (arrowheads) and narrow larynx in front (arrow).
Contributors OU and RS contributed to acquisition of case details. OU wrote the first draft of the manuscript. JR supplied the diagnostic images. OU, BM, JR and RS revised the manuscript. All authors have given final approval of this version to be published.
Uleberg O, et al. Arch Dis Child Fetal Neonatal Ed Month 2015 Vol 0 No 0
F1
Copyright Article author (or their employer) 2015. Produced by BMJ Publishing Group Ltd (& RCPCH) under licence.
Downloaded from http://fn.bmj.com/ on July 13, 2015 - Published by group.bmj.com
Images in neonatal medicine Competing interests None declared. Patient consent Obtained. Provenance and peer review Not commissioned; internally peer reviewed. To cite Uleberg O, Myklebust B, Rydland J, et al. Arch Dis Child Fetal Neonatal Ed Published Online First: [ please include Day Month Year] doi:10.1136/archdischild2015-308529 Received 3 March 2015 Revised 27 May 2015 Accepted 8 June 2015
F2
Arch Dis Child Fetal Neonatal Ed 2015;0:F1–F2. doi:10.1136/archdischild-2015-308529
REFERENCES 1 2
Sannoh S, Quezada E, Merer D, et al. Cystic hygroma and potential airway obstruction in a newborn; a case report and review of the literature. Cases J 2009;2:48. Song TB, Kim CH, Kim SM, et al. Fetal axillary cystic hygroma detected by prenatal ultrasonography: a case report. J Korean Med Sci 2002;17:400–2.
Uleberg O, et al. Arch Dis Child Fetal Neonatal Ed Month 2015 Vol 0 No 0
Downloaded from http://fn.bmj.com/ on July 13, 2015 - Published by group.bmj.com
An unexpected cause of stridor in a newborn Oddvar Uleberg, Bjørn Myklebust, Jana Rydland and Ragnhild Støen Arch Dis Child Fetal Neonatal Ed published online June 30, 2015
Updated information and services can be found at: http://fn.bmj.com/content/early/2015/06/30/archdischild-2015-308529
These include:
References
This article cites 2 articles, 0 of which you can access for free at: http://fn.bmj.com/content/early/2015/06/30/archdischild-2015-308529 #BIBL
Email alerting service
Receive free email alerts when new articles cite this article. Sign up in the box at the top right corner of the online article.
Topic Collections
Articles on similar topics can be found in the following collections Oncology (199) Competing interests (ethics) (92) Journalology (100) Surgery (67) Surgical diagnostic tests (63)
Notes
To request permissions go to: http://group.bmj.com/group/rights-licensing/permissions To order reprints go to: http://journals.bmj.com/cgi/reprintform To subscribe to BMJ go to: http://group.bmj.com/subscribe/
