Letters to the Editor 79 evidence-based prospective studies establishing time of onset, JG Galaznik determining prevalence and refining our understanding of etiology Student Health Center, University of Alabama, Tuscaloosa, of these chronic subdural effusions, the allegations against AL, USA caregivers, the finger pointing at obstetricians and the blame-game E-mail: [email protected] in general will continue to be played in the courts, and the victims will be the infants, their families and the social service and References legal systems called on to resolve the issue based on ‘generally accepted presumption’, not evidence-based medical science. 1 Galaznik JG. A case for an in utero etiology of chronic SDH/effusions of infancy. J Perinatol 2011; 31: 220–222. 2 van Rijn RR, Hobbs D, Bilo RAC. Letter to editor in response to: Galaznik JG. A case I have given testimony in civil and criminal proceedings involving for an in utero etiology of chronic SDH/effusions of infancy. J Perinatol 2011; 31: cases of alleged physical abuse of infants and small children. 220–222.

A case for an in utero etiology of chronic SDH/effusion of infancy (letter)

Journal of Perinatology (2012) 32, 79–81; doi:10.1038/jp.2011.77 comparing their growth with each other is no more meaningful than comparing the growth patterns of siblings from different We read with interest the Perinatal/Neonatal Case Presentation pregnancies. As twin A and twin B are genetically different, their involving a 3-month-old infant with new onset seizures who growth, relative to each other, contributes little to our reportedly was discovered to have a chronic subdural hematoma understanding of the intracranial process in twin A. (CSDH)1 after having ‘new onset partial complex seizures’. The Several important pieces of information are notably absent in infant, a dichorionic–dizygotic twin (twin A), was noted to have a the presented clinical materials. The author reports in passing that large head circumference and bulging fontanelle. The infant was the evaluation of twin A revealed retinal hemorrhages. No other noted to have retinal hemorrhages and had his CSDH drained of information is included about the pattern, location or extent of the ‘xanthochromic fluid with some fresh blood’, which was ‘under retinal hemorrhages. A more precise description of the retinal very high pressure’. The author concluded that the infant’s findings is warranted. The author, who acknowledges serving as intracranial findings were the result of an in utero ‘process’ as an expert witness in previous child abuse cases, certainly must opposed to a postnatal traumatic event. Although the case appreciate that retinal hemorrhages are highly associated with presented is intriguing, we have concerns about the case vignette inflicted brain injury in this age group.2–5 Given this and the interpretation of the data presented that make well-recognized association, the absence of other examinations it difficult for us to draw any meaningful conclusions. to evaluate for additional injury in this patient is perplexing. The vignette presented is apparently from a case that the author The diagnostic workup for infants with abnormalities that could reviewed for a legal process and was not an infant whom he be caused by physical abuse usually includes a skeletal survey.6,7 provided care for. The author reports that a review of the prenatal We question the failure to obtain a radiographic skeletal survey in records supports the infant’s CSDH being from an in utero origin. this patient. Without more complete information about the The support for this conclusion is based upon comparison of the retinal and skeletal findings, non-accidental trauma cannot be intrauterine growth pattern and head shape of the twins. It is excluded. unclear how this conclusion can be supported. The author reports In the discussion, the author reports that in the absence of a ‘superb series’ of prenatal ultrasounds that demonstrate an a prenatal MRI, the in utero intracranial abnormality cannot be accelerating intrauterine growth of twin A as compared with twin adequately assessed. However, apparently a ‘superb’ set of B. The author reports a ‘mathematical analysis’ that includes ultrasound examinations was obtained. The author provides no a ‘‘head/body’ disproportion’ comparison index. As the description of the intracranial findings on these examinations, only mathematical analysis presented is not the one clinically used in measurements of head size and description of skull shape. An assessing fetal growth, the value of this metric in clinical practice intracranial process in the cranium of twin A, large enough to is unclear. The author reports that the twin B is ‘healthy by all occupy ‘25% of the intracranial volume’, should have been clearly accounts’ but includes no data to support claim. As the author visible on a routine prenatal ultrasound. Prenatal ultrasound is points out, the twins are dizygotic (fraternal) and, therefore, an excellent modality for identifying intracranial collections,

Journal of Perinatology Letters to the Editor 80 particularly one that is actually enlarging the fetus’ head to be present. These additional injuries rendered their argument circumference. As no intracranial ultrasound findings are for an innocuous cause of the infant’s death unsupported.17 described, we conclude that no abnormal collections were seen Incomplete information, incomplete understanding of the on the prenatal ultrasound. As no images were included in the precision of prenatal ultrasound, novel clinical metrics and vignette, the reader is unable to confirm this. misinterpretation of the medical literature render the reader unable Given the clinical information presented, the author argues that to draw any meaningful conclusions from the presented case. What the child’s CSDH could have been from an in utero ‘process’ can be concluded is that the infant presented in the vignette that was unseen on ultrasound, remained quiescent and, did not have evidence of a significant in utero intracranial unprompted, rapidly became symptomatic. The author appears to collection as a cause for his postnatal findings and concerns use the term CSDH and effusion synonymously, despite they being for non-accidental trauma are justifiable. separate, distinct entities. Subdural effusions (often called hygromas) represent an accumulation of cerebro spinal fluid without blood products or other tissues and may be posttraumatic Conflict of interest in origin.8–10 Subdural hygromas are not reported to rebleed The authors declare no conflict of interest. spontaneously, one feature distinguishing them from CSDH. CSDHs are a distinctly different entity. Although they are also C Greeley1 and S John2 usually posttraumatic, they have a well-described inner and outer 1Department of Pediatrics, Center for Clinical Research and neomembrane architecture.11,12 Silent rebleeding in a CSDH is well 13,14 Evidence-Based Medicine, University of Texas Health Science described, but symptomatic rebleeding of a CSDH in an infant, Center, Houston, TX, USA and 2Imaging and Pediatrics, without significant trauma, has not been described. The author University of Texas Health Science Center, Houston, TX, USA cites Till15 as support for the contention that the retinal E-mail: [email protected] hemorrhages, altered behaviors and seizures in the presented child were from rebleeding of a (preexisting) CSDH. Till’s work dates back to a time when skull radiographs and References pneumoencephalography were the only imaging tools available for evaluating subdural collections and has limited usefulness in 1 Galaznik JG. A case for an in utero etiology of chronic SDH/effusion of infancy. J Perinatol 2011; 31: 220–222. this era of advanced brain imaging. Till describes a cohort of 116 2 Morad Y, Wygnansky-Jaffe T, Levin AV. Retinal haemorrhage in abusive head trauma. infants with ‘subdural effusions or hematoma’. The infants Clin Experimental Ophthalmol 2010; 38: 514–520. described in Till had any form of subdural collection, including 3 Binenbaum G, Mirza-George N, Christian C, Forbes BJ. Odds of abuse associated with acute SDH. Additionally, 54 of the 116 infants described by retinal hemorrhages in children suspected of child abuse. J AAPOS 2009; 13: 268–272. Till had ‘retinal or subhyloid hemorrhages’, but this number 4 Bhardwaj G, Chowdhury V, Jacobs MB, Moran KT, Martin FJ, Coroneo MT. A systematic ‘would have been undoubtedly higher if more time had been spent review of the diagnostic accuracy of ocular signs in pediatric abusive head trauma. 15 Ophthalmology 2010; 117(5): 983–992.e17. examining the fundi of these babies.’ Till also reported that 13 5 Maguire S, Pickerd N, Farewell D, Mann M, Tempest V, Kemp AM. Which clinical infants had skull fractures. As for the cause of the subdural features distinguish inflicted from noninflicted brain injury? A systematic review. collections, according to Till, ‘It must be admitted, however, Arch Dis Child 2009; 94: 860–867. that there is no satisfactory explanation in many cases, although 6 Meyer JS, Gunderman R, Coley BD, Bulas D, Garber M, Karmazyn B et al. ACR F trauma is an important factor in the majority.’ It appears that appropriateness criteria on suspected physical abuse child. J Am Coll Radiol 2011; 8: 87–94. the citation used by Galaznik to support the findings of the 7 Kellogg ND, and the Committee on Child Abuse and Neglect. Evaluation of suspected presented vignette (CSDH, retinal hemorrhages and seizure) as not child physical abuse. Pediatrics 2007; 119(6): 1232–1241. being caused by abuse consisted predominantly of infants who, 8 Vezina G. Assessment of the nature and age of subdural collections in nonaccidental by current conventional standards, would be seen as likely victims head injury with CT and MRI. Pediatr Radiol 2009; 39(6): 586–590. of physical abuse (retinal hemorrhages, skull/skeletal fractures, 9 Wilms G, Vanderschueren G, Demaeral PH, Smet MH, Van Calenbergh F, Plets C et al. acute SDH and trauma). CT and MRI in infants with pericerebral collections and macrocephaly: benign enlargement of the subarachnoid spaces versus subdural collections. The presented vignette contains too many discrepancies and Am J Neuroradiol 1993; 14: 855–860. omissions to allow for meaningful interpretation. In a similar 10 Kristof R, Grimm J, Stoffel-Wagner B. Cerebrospinal fluid leakage into the manner, the author recently reported16 a child fatality in which subdural space: possible influence on the pathogenesis and recurrence frequency of incomplete clinical information altered the correct interpretation. chronic subdural hematoma and subdural hygroma. Journal of Neurosurg 2008; Galaznik and his co-authors16 argued that an infant’s fatal injuries 108: 275–280. 11 Schachenmayr W, Friede RL. The origin of subdural neomembranes: part i, fine were not the result of abuse but of ‘dysphagic coughing’ and structure of the dura-arachnoid interface in man. Am J Pathol 1978; 92: 53–68. subsequent resuscitation efforts. Galaznik and his co-authors did 12 Friede RL, Schachenmayr W. The origin of subdural neomembranes: part ii, fine not include in the published vignette injuries that were confirmed structure of neomembranes. Am J Pathol 1978; 92: 69–84.

Journal of Perinatology Letters to the Editor 81

13 Ito H, Yamamoto S, Komai T, Mizukoshi H. Role of local hyperfibrinolysis 15 Till K. Subdural haematoma and effusion in infancy. Br Med J 1968; 3: in the etiology of chronic subdural hematoma. J Neurosurg 1976; 45(1): 400–402. 26–31. 16 Barnes PD, Galaznik JG, Gardner H, Shuman M. Infant acute life-threatening 14 Ito H, Yamamoto S, Saito K, Ikeda K, Hisada K. Quantitative estimation of hemorrhage eventFdysphagic choking versus nonaccidental injury. Semin Pediatr Neurol 2010; in chronic subdural hematoma using the 51Cr erythrocyte labeling method. 17: 7–12. J Neurosurg 1987; 66(6): 862–864. 17 Greeley CS. Infant Fatality (letter). Semin Pediatr Neurol 2010; 17: 275–278.

Response to ‘A case for an in utero etiology of chronic SDH/effusions of infancy’

Journal of Perinatology (2012) 32, 81–82; doi:10.1038/jp.2011.80 increased risk for the development of subdural hematomas resulting from minor trauma. However, in one series of patients We have read the paper ‘A case for an in utero etiology of chronic with benign enlargement of the subarachnoid space, except for 1 SDH/effusions of infancy’ by Galaznik with interest. Acaseofchild one child who survived a motor vehicle accident, no retinal with a subdural hematoma, which is presumed to have arisen hemorrhages were reported.8 Common clinical reasoning in utero, is presented. We feel that several issues should be addressed. dictates that ‘the rare presentation of a common disorder is First is the fact that a scientific publication, even a case report, more common than a common presentation of a rare disorder’. should provide all available information so that the reader can  At the age of 3 months, twin A is presented in hospital with a form his/her own founded opinion, based on the presented ‘new onset of partial complex seizure’. At presentation, his head findings. This case report lacks much information needed to do so. circumference was 44 cm (>95th percentile). Ophthalmology results showed retinal hemorrhages and computed tomography  No information on the parental history or social background is scans showed bilateral subdural hematomas with normal provided. We believe that no diagnosis of child abuse should be underlying brain development. No further description of the made or dismissed without a proper evaluation of all factors, retinal hemorrhages is provided, it is therefore impossible to including parental history and the psychosocial environment in assess the clinical implication of this finding. The author refers which the child resides.2 to a publication dating from 1968 to explain that at that time,  At 20 weeks of gestational age, prenatal ultrasound showed a the presence of retinal hemorrhages in the absence of well- larger biparietal diameter for twin A compared with twin B, defined head trauma was considered almost pathognomonic for whereas femoral length of twin A was shorter compared with his chronic subdural hematoma.9 He fails to mention that in 46 sib. However, P-values and confidence intervals are not stated. cases (40%), there were clear signs of previous trauma, Serial prenatal ultrasound, presumably part of clinical routine, consisting of bruises and fractures, and that in 54 cases (47%), was performed. Post-natal cranial ultrasonography is not retinal hemorrhages were seen. Furthermore, it is of interest to mentioned. This suggests that macrocrania was clinically not note that Till10 comments on the fact that trauma must have considered to be abnormal to such a degree that it warranted occurred in the 46 cases. In response to a letter to the editor, he further evaluation. The absence of post-natal cranial does indicate that child abuse was considered in a group of ultrasonography is interesting as in our opinion a child with patients. Since 1968, medicine has seen significant changes in serial abnormal antenatal exams should not have been sent the way we not only practice our profession but also approach home without proper evaluation. From the presence of the and evaluate our patients. Applying clinical standards from chronic subdural hematoma at the age of 3 months, it is nearly half a century ago to today’s practice does, in our concluded that the increased biparietal diameter could have opinion, not make sense. been the result of a subdural hematoma arisen in utero.  Any presentation of a child with the reported findings should Although this has been documented in literature, it is a rare raise the suspicion of abusive head trauma, as seems to have finding with, in most case reports, serious outcomes either happened in this case. In the policy statement of the American during pregnancy or after shortly after birth.3–6 This report, Association of Pediatrics on abusive head trauma, close however, explores no differential diagnosis for the macrocrania. collaboration with consultants in subspecialties, among which One common clinical differential diagnosis for macrocrania, radiologists, is advised.2 Both the American Association of which also can be diagnosed prenatally, is benign enlargement Pediatrics and the American College of Radiology have issued of the subarachnoid space.7 This has been related to an guidelines for the radiological evaluation of suspected child

Journal of Perinatology