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Paediatric and Perinatal Postmortem Imaging: the Need for a Subspecialty Approach

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Paediatric and Perinatal Postmortem Imaging: the Need for a Subspecialty Approach Pediatr Radiol (2015) 45:483–490 DOI 10.1007/s00247-014-3132-8 REVIEW Paediatric and perinatal postmortem imaging: the need for a subspecialty approach Owen J. Arthurs & Rick R. van Rijn & Andrew M. Taylor & Neil J. Sebire Received: 13 March 2014 /Revised: 24 June 2014 /Accepted: 16 July 2014 /Published online: 30 August 2014 # The Author(s) 2014. This article is published with open access at Springerlink.com Abstract Paediatric postmortem imaging is distinct and different Introduction from adult postmortem imaging due to differences in disease aetiology, pathology and imaging approaches, which require a In our opinion, paediatric postmortem imaging is very differ- particular skill set to maximise its yield and clinical utility. Prac- ent from adult postmortem imaging, due to differences in titioners need to have expertise in several aspects of radiology, disease aetiology, pathology and imaging approaches, which including both plain radiographs and cross-sectional imaging require a particular skill set to maximise its yield and clinical modalities, knowledge of specialist techniques, and familiarity utility. Practitioners need to have combined expertise in sev- with the unique range of pathologies in this patient population, eral aspects of radiology, including both plain radiographs and including perinatal pathology. Here we outline the training re- cross-sectional imaging modalities, together with a knowl- quirements that should be considered to establish such a service. edge of specialist techniques and a familiarity with the unique range of pathologies in this patient population, including perinatal pathology. Here we outline the training requirements Keywords Autopsy . Postmortem . MRI . Children . that should be considered to establish such a service. Note that Perinatal . Foetal throughout this manuscript, we use internationally agreed O. J. Arthurs (*) terminology for postmortem imaging [1]. Department of Radiology, Great Ormond Street Hospital for Children NHS Foundation Trust, London WC1N 3JH, UK What is the clinical need? e-mail: [email protected] R. R. van Rijn The numbers of foetal, stillborn and infant deaths in any Department of Radiology, country are relatively small in comparison to overall adult ’ Emma Children s Hospital - Academic Medical Centre, deaths (around 1–2% of total United Kingdom deaths are Amsterdam, The Netherlands stillbirths, infants and children), but this group still represents A. M. Taylor a significant clinical issue. This article discusses only the Cardiorespiratory Unit, impact of paediatric postmortem imaging, acknowledging Great Ormond Street Hospital for Children, the importance of this small group on parental bereavement, London, UK genetic counselling and planning of future pregnancies. N. J. Sebire The perinatal mortality rate (PMR) is defined as the death of a Department of Pathology, foetus >24 weeks or early neonatal death <7 days per 1,000 live Great Ormond Street Hospital for Children, births (Table 1). Even allowing for slightly different definitions, London, UK PMR shows a significant variability across Europe, ranging from : O. J. Arthurs N. J. Sebire 4.6 per 1,000 in Germany to 12.35 per 1,000 in Latvia (2004 UCL Institute of Child Health, London, UK data; Fig. 1 [2]). For full-term live births these figures are lower, ranging from 1.2 per 1,000 in Luxembourg to 5.1 per 1,000 in A. M. Taylor Centre for Cardiovascular Imaging, UCL Institute of Cardiovascular Latvia [2]. This equates to around 36,000 annual perinatal deaths Science, London, UK across Europe. In addition, there are a significant number of 484 Pediatr Radiol (2015) 45:483–490 Table 1 Definition of types of paediatric death – Does a stillbirth with normal antenatal US imaging and Term Definition normal karyotype benefit from detailed postmortem im- aging using CT or MRI, or will other investigations, such Late foetal loss Delivered showing no signs of life between 20+0 as placental histology, provide the likely cause of death in and 23+6 weeks of pregnancy. the majority of cases? Termination of Induced delivery, with or without fetocide. – Does conventional postmortem MRI of very small foe- pregnancy tuses in the first and early second trimester yield diagnos- Stillbirth Delivered showing no signs of life after 24+0 weeks of pregnancy. tically useful information? – Neonatal death Death of a live born baby occurring within In what proportion of cases originally diagnosed as SIDS 28 days of birth. could postmortem CT or postmortem MRI provide addi- Early neonatal death Death of a live born baby occurring within 7 days tional clinically relevant information by determining a of birth. cause of death? Perinatal death Stillbirths and early neonatal deaths. Post-neonatal or Death occurring from the 28th day to 1 year of Investigating these questions may require a coordinated infant deaths age. approach between several different centres or different coun- Childhood deaths Death between ages 1 year and 16 or 18 years. tries to allow sufficient population sampling. The value of imaging in the paediatric setting should also always be taken in the context of other less-invasive sampling deaths in infants and children, with many remaining unexplained methods, including external examination of the body, skeletal (Sudden Unexplained Deaths in Infancy; SUDI). radiographs, photography, pathological assessment of the pla- Whilst a high percentage of parents indicate a strong desire centa, noninvasive swabs for microbiology, genetics chromo- to know why their child died and that an autopsy helped them somal analysis and other biomarkers, as such investigations, cope with their grief, bereaved parents may be reluctant to traditionally part of the autopsy, often provide additional agree to a full traditional autopsy [3, 4]. It is still perceived to diagnostic information [11, 12]. be invasive and unacceptable by many, although there are a variety of reasons for parents’ refusal [3, 4]. Postmortem imaging in children Overall autopsy rates have shown a significant decline across the world [5] with neonatal and infant autopsy rates currently at Evaluating the true value of postmortem imaging is difficult, around 30% or lower [6]. This is well below national standard but direct comparison between postmortem imaging and autop- recommendations of 75% and means that large amounts of sy findings have recently been published [11, 13]. In adults, the information that could be used to counsel parents about future concordance rates of adult postmortem imaging and full autop- pregnancies, and contribute to epidemiological studies regarding sy findings in the only blinded study published to date were infant deaths, is currently not available. Several studies have disappointing, at around 50% [13]. This included 180 cases, shown a 10–25% error or discrepancy rate between what clini- and the major discrepancy rate between cause of death identi- cians think is the cause of death and the findings of a full fied by radiology and autopsy was 32% for postmortem CT and traditional autopsy [7, 8], with error rates as high as 50% on 43% for postmortem MRI. Postmortem CT appears to be a medical certificates in stillbirths [9]. These errors may be attrib- more accurate imaging technique than postmortem MRI for utable to the infrequency with which traditional autopsies are providing a cause of death in adults, with a similar error rate to performed, and there continues to be little informed support traditional autopsy/clinical death certification. Typical errors available for parents about autopsy following stillbirth [10]. included ischaemic heart disease, pulmonary embolism and There is a broad range of perinatal and paediatric diagnoses pneumonia. Whilst there is room for improvement, postmortem and causes of death, which are very different from adult CT and postmortem CT angiography (CTA) are already becom- mortality causes [11]. This further highlights the need to ing the workhorse imaging modality for adult imaging [14–16]. optimise postmortem examinations to address issues specific Recent data confirm that postmortem MRI is likely to to the type of death, which is likely to be encountered in each become the standard for postmortem imaging in children. A age or gestational group. These, in turn, need to be carefully recent benchmark prospective validation study of postmortem formulated into pragmatic clinical guidelines. Some aspects imaging in foetuses and children at a specialist children’s may be intuitive to those medical professionals with a famil- hospital in London found a >90% concordance rate between iarity with this clinical presentation, such as a radiographic noninvasive postmortem assessment (including postmortem skeletal survey being fundamental in paediatric deaths suspi- MRI and ancillary investigations not requiring invasive pro- cious for inflicted or non-accidental injury. cedures, such as placental examination) and conventional full However, an evidence base is needed to address currently autopsy findings in 400 cases (277 foetuses, 123 children) unanswered questions in this population, such as: [11]. Interestingly, this was greatest for foetuses (<24 weeks Pediatr Radiol (2015) 45:483–490 485 Sweden 0.6 2.0 2.6 <27 weeks 28 - 36 weeks >37 weeks Malta 0.8 4.4 3.1 Luxembourg 0.9 3.0 1.1 Germany 1.5 1.8 1.2 Portugal 1.7 2.5 1.6 Spain/Valencia 2.1 2.7 1.7 Ireland 2.1 3.3 2.6 Denmark 2.2 2.3 3.7 Belgium/Flanders 2.3 2.7 1.5 Norway 2.3 1.9 2.4 Finland 2.3 1.9 1.5 Czech Republic 2.3 2.0 1.6 Country/Region Austria 2.6 2.3 1.5 Belgium/Brussels 2.6 3.3 2.2 Estonia 2.9 1.9 3.9 Poland 2.9 4.0 2.7 Lithuania 2.9 3.2 3.6 Slovenia 3.3 3.0 1.9 Latvia 3.5 4.0 4.8 UK/Scotland 3.6 3.3 2.7 UK/Northern Ireland 4.1 3.1 2.1 Netherlands 4.3 3.2 3.0 0471114 Rate per 1,000 total births Fig.
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