Ultraschall Med 2023; 44(05): 503-511
DOI: 10.1055/a-1820-8101
Original Article

Surface Cranial Ultrasound: The Natural Heir to X-Ray for the Screening of Skull Deformities in Infants

Kranialer Ultraschall der Schädeloberfläche: Nachfolger der Röntgen-Untersuchung bei Schädeldeformitäten von Säuglingen
Laura Maria Pogliani
1   Pediatric Department, Ospedale di Legnano Nuova Sede, Legnano, Italy (Ringgold ID: RIN424691)
,
Gian Vincenzo Zuccotti
2   Department of Biomedical and Clinical Sciences, Università degli Studi di Milano, Milan, Italy (Ringgold ID: RIN9304)
3   Pediatrics, Ospedale Luigi Sacco-Polo Universitario, Milan, Italy (Ringgold ID: RIN18602)
,
Maddalena Reggiori
3   Pediatrics, Ospedale Luigi Sacco-Polo Universitario, Milan, Italy (Ringgold ID: RIN18602)
,
Alessandra Erbetta
4   Neuroradiology, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy (Ringgold ID: RIN9328)
,
Michele Lacerenza
5   Physics, Politecnico di Milano, Milan, Italy (Ringgold ID: RIN18981)
,
6   Neurosurgical Department, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milano, Italy (Ringgold ID: RIN9328)
7   Neurological Surgery, University of Virginia School of Medicine, Charlottesville, United States (Ringgold ID: RIN12349)
,
Marika Furlanetto
6   Neurosurgical Department, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milano, Italy (Ringgold ID: RIN9328)
,
6   Neurosurgical Department, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milano, Italy (Ringgold ID: RIN9328)
,
6   Neurosurgical Department, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milano, Italy (Ringgold ID: RIN9328)
› Author Affiliations

Abstract

Purpose Volumetric tomography (3D-CT) is currently considered the gold standard for the diagnosis of craniosynostosis, but its use as the first-line examination for cranial deformities is a topic of debate, because of skull X-ray radiation and low sensitivity and specificity. Cranial ultrasound is an emerging noninvasive radiation-free alternative, but its diagnostic accuracy still needs confirmation.

Materials and Methods The present prospective study included 350 infants with skull deformities, who underwent cranial ultrasound as the first-line examination, followed by 3D-CT if the echography results was positive or unclear. If the results were negative, infants underwent physical treatment and follow-up. To evaluate ultrasound reliability, we focused on cases that underwent both the index test and the gold standard and performed a double-blind comparison of the echography and 3D-CT results.

Results Ultrasound documented patent sutures in 293 infants and 9 had inconclusive results. The 293 ultrasound-negative infants were followed clinically: all improved, except 28 that underwent 3D-CT. In all of these cases, 3D-CT confirmed the ultrasonography results (no false negatives). 48 infants showed premature suture closure and underwent 3D-CT: 47 were confirmed (true positive), 1 was false positive. The sensitivity was 100%, the specificity was 99.7%, the positive and negative predictive values were 97.9% and 100%, respectively, the accuracy was 99.7%, and the diagnostic test evaluation was conclusive.

Conclusion The study documented the high sensitivity and specificity of echography for the diagnosis of craniosynostosis in a referral center, with better results being achieved before 6 months of age. Major limitations are the loss of diagnostic significance as the child grows and the learning curve needed. The advantages are avoidance of radiation and chance to evaluate the brain at the same time.

Zusammenfassung

Ziel Die volumetrische Tomografie (3D-CT) gilt derzeit als Goldstandard für die Diagnose von Kraniosynostosen, ist jedoch für die Erstuntersuchung von Schädeldeformitäten aufgrund der Strahlenbelastung und der geringen Sensitivität/Spezifität umstritten. Die kraniale Sonografie ist eine aufkommende, nicht-invasive, strahlungsfreie Alternative, aber ihre diagnostische Genauigkeit muss noch bestätigt werden.

Material und Methoden Die vorliegende prospektive Studie umfasste 350 Säuglinge mit Schädeldeformitäten, die als Erstuntersuchung einem kranialen Ultraschall unterzogen wurden, gefolgt von einer 3D-CT, wenn die sonografischen Befunde positiv oder fraglich waren. War der Befund negativ, wurden die Kinder ärztlich behandelt und nachuntersucht. Um die Zuverlässigkeit der Sonografie zu bewerten, konzentrierten wir uns auf Fälle, die sowohl dem Index-Test als auch dem Goldstandard unterzogen wurden, und verglichen unter doppelter Verblindung Sonografie- und 3D-CT-Befunde.

Ergebnisse Bei 293 Säuglingen wurden sonografisch offene Nähte nachgewiesen, bei 9 Kindern war der Befund nicht eindeutig. Die 293 Säuglinge mit unauffälligem sonografischen Befund wurden klinisch nachverfolgt: Bei allen, verbesserte sich das klinische Bild, mit Ausnahme von 28, die mit 3D-CT untersucht wurden. In all diesen Fällen bestätigte die 3D-CT den Ultraschallbefund (keine falsch-negativen). 48 Säuglinge zeigten einen verfrühten Nahtverschluss und wurden einer 3D-CT unterzogen: 47 Fälle wurden bestätigt (richtig-positive), 1 Fall war falsch-positiv. Die Sensitivität betrug 100 %, die Spezifität 99,7 %, der positive und negative Vorhersagewert 97,9 % bzw. 100 %, die Genauigkeit 99,7 %. Die Evaluation des diagnostischen Tests war schlüssig.

Schlussfolgerung Die Studie dokumentiert die hohe Sensitivität und Spezifität der Sonografie bei der Diagnose der Kraniosynostose in einem Spezialzentrum, mit besseren Ergebnissen vor dem 6. Lebensmonat. Die wichtigsten Einschränkungen sind die geringere Aussagekraft bei zunehmendem Alter und der erforderliche Lernprozess; Vorteile sind die Vermeidung der Strahlenbelastung und die Möglichkeit, das Gehirn gleichzeitig zu untersuchen.

Supporting information



Publication History

Received: 22 September 2020

Accepted after revision: 13 March 2022

Article published online:
27 June 2022

© 2022. Thieme. All rights reserved.

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 
  • References

  • 1 Mathijssen IMJ. Guideline for Care of Patients With the Diagnoses of Craniosynostosis: Working Group on Craniosynostosis. J Craniofac Surg 2015; 26: 1735-1807
  • 2 Jentink J, Loane MA, Dolk H. et al. Valproic acid monotherapy in pregnancy and major congenital malformations. N Engl J Med United States 2010; 362: 2185-2193
  • 3 Furuya Y, Edwards MS, Alpers CE. et al. Computerized tomography of cranial sutures. Part 1: Comparison of suture anatomy in children and adults. J Neurosurg United States 1984; 61: 53-58
  • 4 Morris LM. Nonsyndromic Craniosynostosis and Deformational Head Shape Disorders. Facial Plast Surg Clin North Am United States 2016; 24: 517-530
  • 5 Choi JW, Lim SY, Shin H-J. Craniosynostosis in Growing Children: Pathophysiological Changes and Neurosurgical Problems. J Korean Neurosurg Soc 2016; 59: 197-203
  • 6 American Academy of Pediatrics AAP Task Force on Infant Positioning and SIDS: Positioning and SIDS. . Pediatrics United States 1992; 89: 1120-1126
  • 7 Ghizoni E, Denadai R, Raposo-Amaral CA. et al. Diagnosis of infant synostotic and nonsynostotic cranial deformities: a review for pediatricians. Rev Paul Pediatr 2016; 34: 495-502
  • 8 Linz C, Collmann H, Meyer-Marcotty P. et al. Occipital plagiocephaly: unilateral lambdoid synostosis versus positional plagiocephaly. Arch Dis Child England 2015; 100: 152-157
  • 9 Moore CL, Copel JA. Point-of-care ultrasonography. N Engl J Med United States 2011; 364: 749-757
  • 10 Brenner D, Elliston C, Hall E. et al. Estimated risks of radiation-induced fatal cancer from pediatric CT. AJR Am J Roentgenol United States 2001; 176: 289-296
  • 11 Domeshek LF, Mukundan SJ, Yoshizumi T. et al. Increasing concern regarding computed tomography irradiation in craniofacial surgery. Plast Reconstr Surg United States 2009; 123: 1313-1320
  • 12 Persing J, James H, Swanson J. et al. Prevention and management of positional skull deformities in infants. American Academy of Pediatrics Committee on Practice and Ambulatory Medicine, Section on Plastic Surgery and Section on Neurological Surgery. Pediatrics United States 2003; 112: 199-202
  • 13 Simanovsky N, Hiller N, Koplewitz B. et al. Effectiveness of ultrasonographic evaluation of the cranial sutures in children with suspected craniosynostosis. Eur Radiol Germany 2009; 19: 687-692
  • 14 Sze RW, Parisi MT, Sidhu M. et al. Ultrasound screening of the lambdoid suture in the child with posterior plagiocephaly. Pediatr Radiol Germany 2003; 33: 630-636
  • 15 Alizadeh H, Najmi N, Mehdizade M. et al. Diagnostic accuracy of ultrasonic examination in suspected craniosynostosis among infants. Indian Pediatr India 2013; 50: 148-150
  • 16 Rozovsky K, Udjus K, Wilson N. et al. Cranial Ultrasound as a First-Line Imaging Examination for Craniosynostosis. Pediatrics United States 2016; 137: e20152230
  • 17 Hall KM, Besachio DA, Moore MD. et al. Effectiveness of screening for craniosynostosis with ultrasound: a retrospective review. Pediatr Radiol Germany 2017; 47: 606-612
  • 18 Proisy M, Riffaud L, Chouklati K. et al. Ultrasonography for the diagnosis of craniosynostosis. Eur J Radiol Ireland 2017; 90: 250-255
  • 19 Safran T, Viezel-Mathieu A, Beland B. et al. The State of Technology in Craniosynostosis. J Craniofac Surg United States 2018; 29: 904-907
  • 20 Mazzola C, Baird LC, Bauer DF. et al. Guidelines: Congress of Neurological Surgeons Systematic Review and Evidence-Based Guideline for the Diagnosis of Patients With Positional Plagiocephaly: The Role of Imaging. Neurosurgery United States 2016; 79: E625-E626
  • 21 Massimi L, Bianchi F, Frassanito P. et al. Imaging in craniosynostosis: when and what?. Child’s Nerv Syst ChNS Off J Int Soc Pediatr Neurosurg Germany 2019; 35: 2055-2069
  • 22 Rozovsky K, Barrowman NJ, Miller E. Centile charts for cranial sutures in children younger than 1 year based on ultrasound measurements. Pediatr Radiol Germany 2018; 48: 701-707
  • 23 Okamoto T, Nuri T, Harada A. et al. Cranial Suture Measurement by 2-point Method in Ultrasound Screening of Craniosynostosis. Plast Reconstr surgery Glob open 2019; 7: e2225
  • 24 Pearce MS, Salotti JA, Little MP. et al. Radiation exposure from CT scans in childhood and subsequent risk of leukaemia and brain tumours: a retrospective cohort study. Lancet (London, England) 2012; 380: 499-505
  • 25 Mathews JD, Forsythe A V, Brady Z. et al. Cancer risk in 680,000 people exposed to computed tomography scans in childhood or adolescence: data linkage study of 11 million Australians. BMJ 2013; 346: f2360
  • 26 Proisy M, Bruneau B, Riffaud L. How ultrasonography can contribute to diagnosis of craniosynostosis. Neurochirurgie France 2019; 65: 228-231
  • 27 Regelsberger J, Delling G, Helmke K. et al. Ultrasound in the diagnosis of craniosynostosis. J Craniofac Surg United States 2006; 17: 623-628
  • 28 Regelsberger J, Delling G, Tsokos M. et al. High-frequency ultrasound confirmation of positional plagiocephaly. J Neurosurg United States 2006; 105: 413-417
  • 29 Krimmel M, Will B, Wolff M. et al. Value of high-resolution ultrasound in the differential diagnosis of scaphocephaly and occipital plagiocephaly. Int J Oral Maxillofac Surg Denmark 2012; 41: 797-800
  • 30 Robinson S, Proctor M. Diagnosis and management of deformational plagiocephaly. J Neurosurg Pediatr United States 2009; 3: 284-295