Child Abuse & Neglect 58 (2016) 173–179
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Child Abuse & Neglect
Research article
Abusive head trauma in Spanish language medical literature M. Townsend Cooper Jr. a,∗ , Edgardo Szyld b , Paul M. Darden a a The University of Oklahoma Health Sciences Center, Department of Pediatrics, General and Community Pediatrics, 1200 Children’s Ave, Suite 12400, Oklahoma City, OK 73104, USA b The University of Oklahoma Health Sciences Center, Department of Pediatrics, Neonatal-Perinatal Medicine, 1200 Everett Drive, ETNP7504, Oklahoma City, OK 73104, USA
a r t i c l e
i n f o
Article history: Received 8 November 2015 Received in revised form 21 June 2016 Accepted 1 July 2016 Available online 18 July 2016 Keywords: Abusive head trauma Latin America Spanish language
a b s t r a c t Anecdotal experiences raise concerns that abusive head trauma (AHT) is significantly underdiagnosed and perhaps poorly recognized in Latin American settings. With increasing interest in international collaboration in pediatrics, differences in perspectives regarding complex diagnoses should be explored to facilitate a productive exchange of knowledge and ideas. The purpose of this study was to describe the medical literature pertaining to AHT available to physicians who read only in Spanish. In this review, LILACS, SciELO (major Spanish language databases) and Pubmed were searched with appropriate terms and filters in English, Spanish, and Portuguese for Spanish language articles on AHT. Identified articles’ reference lists were then examined for possible additional articles on AHT. All relevant articles were sorted by country and examined for article type and content. Thirty-four unique articles were located for review from 8 of 21 countries. Most of the articles identified were case reports, case series, or educational, and there were no studies regarding overall incidence or prevalence of AHT. Some scientific information contained in the articles varied considerably from that in the English language literature in the areas of etiology and preventive strategies. This survey highlights that the Spanish language literature regarding AHT is sparse and variable. This must be considered when working collaboratively in a global setting. Additionally, identification of this gap presents an opportunity for education and information exchange among global medical communities. © 2016 Elsevier Ltd. All rights reserved.
1. Introduction Abusive head trauma (AHT) is characterized by a constellation of findings including subdural hematoma, retinal hemorrhages, and diffuse brain injury which is indicative of non-accidental trauma (Duhaime, Christian, Rorke, & Zimmerman, 1998). AHT has been found in studies in the United States to be the most common cause of fatal head injury in children under two years of age (Gill et al., 2009). Important milestones in the evolution of the diagnosis include the article by Caffey in 1946 describing the association of long bone fractures and subdural hematomas (Caffey, 1946), the 1971 article by Guthkelch linking subdural hematoma to shaking (Guthkelch, 1971), and the 1974 article by Caffey adding a description of associated retinal hemorrhages (Caffey, 1974). Since that time there have been numerous studies on mechanisms, incidence, and prevention of abusive head trauma in infants (Hymel & Deye, 2011).
∗ Corresponding author. Tel.: +1 405 271 4407; fax: +1 405 271 8709. E-mail addresses:
[email protected] (M.T. Cooper Jr.),
[email protected] (E. Szyld),
[email protected] (P.M. Darden). http://dx.doi.org/10.1016/j.chiabu.2016.07.001 0145-2134/© 2016 Elsevier Ltd. All rights reserved.
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Previously known as “shaken baby syndrome”, recent recommendations call for use of the term “abusive head trauma” (AHT). This is due to ongoing discussion regarding the exact mechanism of injury in these cases, whether shaking alone or a combination of shaking and impact are responsible for the injuries. Because of the variable nature of injury mechanisms, the more inclusive term of abusive head trauma is considered more correct (Christian CW Block R Committee on Child Abuse and Neglect American Academy of Pediatrics, 2009; Duhaime et al., 1998). Studies estimate that incidence of AHT in the United States is between 27.5 and 32.2 cases per 100,000 infants <1 year annually (Ellingson, Leventhal, & Weiss, 2008; Keenan et al., 2003). The composition of the literature regarding AHT varies outside of the United States. Some countries, such as Australia and the United Kingdom, have extensive literature regarding incidence of abusive head trauma. However, even in large countries, there can be a paucity of literature. One such example is Brazil, which as of 2013 did not have any peer reviewed articles detailing annual incidence of abusive head trauma (Lopes, Eisenstein, & Williams, 2013). Articles regarding incidence and prevalence can orient physicians on the need to be alert for a diagnosis. They are also an indicator that the medical community in a particular country is recognizing a diagnosis with sufficient frequency to generate such data. Recently, there has been increased interest among young physicians in becoming educated on Global Health issues, as well as an increasing focus on Global Health in pediatric residency programs (Garfunkel & Howard, 2011). Research has demonstrated wide variation in challenges in the management of abusive head trauma depending on country in countries as diverse as the United Kingdom, Saudi Arabia, and Sweden (Frasier, Kelly, Al-Eissa, & Otterman, 2014). When working abroad, it is important to be aware of the potential differences in perspective and training of international colleagues for optimal collaboration, information exchange, and patient care. Most of the widely cited landmark studies on abusive head trauma (listed above) are in English and originated from the United States or the United Kingdom (Caffey, 1946, 1974; Duhaime et al., 1998; Guthkelch, 1971), and there is a continued lively, in depth discussion of the topic in the English language medical literature. English language Pubmed search using the terms “abusive head trauma” and “shaken baby syndrome” yields 889 unique articles, the first published in 1984. Although in Spanish speaking countries the majority of academic physicians read English well, in our experience working outside of the academic sphere in Latin America this is often not the case. For the monolingual Spanish speaking colleague with whom we might collaborate, questions arise: What is the literature landscape on which they are basing their conclusions and treatment decisions? How different might their perspective be based on what is available only in the Spanish language literature? The aim of this survey is to characterize the medical literature available from the perspective of physicians who read only Spanish regarding the diagnosis of abusive head trauma in infants. 2. Methods The Spanish language medical literature was searched for articles relating to the subject of abusive head trauma or shaken baby syndrome (all dates included). The databases LILACS, SciELO, and Pubmed (Spanish and Portuguese filters) ˜ sacudido, bebé sacudido, traumatismo were searched with the following terms: shaken baby, abusive head trauma, Nino craneoencefálico por maltrato, traumatismo craniano abusivo, trauma craniano violento, bebé sacudido (overlapping terms in English, Spanish, and Portuguese). Although in English the currently recognized term to include all such injuries is abusive head trauma, it is not clear that there is an officially recognized equivalent in Spanish. For this reason, variations on the terms abusive head trauma, “shaken baby” and their translations were included to ensure that the search was inclusive. Additionally, after extensive article review, it was noted that the term “zarandeado” (English: “buffeted”) was used in place of “sacudido” (English: “shaken”) in many of the articles. Accordingly, the above databases were searched for this term without any additional unique yield. We eliminated duplicate results and eliminated articles that were not published in the Spanish language (the searches also yielded articles in English and Portuguese). References in each article were also examined for relevant Spanish language publications. Each article identified by either the primary (database) search or secondary (reference) search was reviewed in detail. If the article did not contain a significant discussion of non-accidental head trauma in infants, the article was excluded from review. The depth of the treatment of the subject was noted along with number of cases if appropriate, and the article was classified by country of publication or origin as appropriate. 3. Results There were 89 articles identified in the original database search. 48 articles were duplicates, leaving 41 unique articles. Ten articles were removed due to non-Spanish publication. Of the remaining articles, 4 did not have any pertinent information or were unavailable despite librarian search, leaving 27 relevant articles. Eleven additional unique relevant articles were identified through examining the references of those articles of which 7 could be accessed, for a total of 34 articles detailed below. See Fig. 1 for flowchart. See Table 1 for article/country/population details. There were no articles estimating prevalence of AHT in the Spanish language literature. Most of the papers located were reviews or personal practice papers, which normally would be excluded from a systematic review searching for effect sizes of a parameter. However, because the express purpose of our search was to determine what resources monolingual Spanish readers would find when searching for information on the topic, we included them in the results to construct a more complete picture of available literature.
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89 arcles idenfied through database search
48 duplicates removed
10 removed for language
4 excluded due to content, format, or availability 11 Idenfied through reference search (snowball search)
4 “snowball” arcles not available
34 arcles included in the analysis
Fig. 1. Article selection process.
Table 1 Articles by Spanish speaking country. Country/territory
Populationa
Number of articles located
Mandated reporting discussed
Physician protection discussed
Argentina Bolivia Chile Colombia Costa Rica Cuba Dominican Republic Ecuador Equatorial Guinea El Salvador Guatemala Honduras Mexico Nicaragua Panama Paraguay Peru Puerto Rico Spain Uruguay Venezuela
43,024,374 10,631,486 17,363,894 46,245,297 4,755,234 11,047,251 10,349,741 15,654,411 722,254 6,125,512 14,647,083 8,598,561 120,286,655 5,848,641 3,608,431 6,703,860 30,147,935 3,620,897 47,737,941 3,332,972 28,868,486
3 0 5 3 4 4 0 0 0 0 0 0 4 0 0 0 0 1 9 0 0
No n/a Yes Yes Yes No n/a n/a n/a n/a n/a n/a Yes n/a n/a n/a n/a No No n/a n/a
No n/a No No Yes No n/a n/a n/a n/a n/a n/a No n/a n/a n/a n/a No No n/a n/a
a
Central Intelligence Agency (2015).
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3.1. Argentina Three articles are available in the Argentinian literature. From 2004, there is a case series of two cases with typical presentations (Esteban et al., 2004). There is an article from 2010 that is a case presentation in a diagnostic challenge section of a pediatric journal (Címbaro Canella, Clemente, González, Indart, & De Lillo, 2010), and there is a large review article regarding recognition, diagnosis, and prevention of shaken baby syndrome from 2012 (Ward & King, 2012). An additional article from Argentina was published in the literature from Spain (see Spain section below). 3.2. Chile Five articles are available from Chile. A 1993 article is a case report and has some recommendations on how to recognize shaken baby syndrome (Perla & Eugenia, 1993). A 2007 piece is a review article and appears to have been prompted by the creation of special family courts whose jurisdiction included child abuse (Coria De La, Canales, Avila, Castillo, & Correa, 2007). An article from 2007 in a neurosurgery journal details neurologic sequelae of different types of child abuse, including abusive head trauma (Giménez-Pando, Dujovny, & Díaz, 2007). A 2009 article is a case series (6 cases) designed to bring this diagnosis to the attention of the medical community (López, 2009). A 2011 article reviews the radiographic findings in abusive head trauma (Eberhard, Pose, Mora, & Francisa-Vergara, 2011). 3.3. Colombia Three articles are available from Colombia. A 2009 article in a Colombian ophthalmology journal describes the ocular lesions of abusive head trauma in detail and instructs physicians on the logistics of how to report abuse (Sánchez Montenegro & Amaya Hernández, 2009). An article published in a nursing journal in 2010 details a potential educational intervention for parents/caregivers to reduce shaken baby syndrome (Montsalve-Quintero, 2010). Much of this article focused on a connection between types of play (e.g. throwing the child into the air) as a cause of shaken baby syndrome, etiologies that are not supported in the English language literature. An article in 2013 is a series from one hospital regarding head injury in general that includes abusive head trauma (Ramírez Restrepo & Aguirre Arango, 2013). 3.4. Costa Rica There are four articles in the Costa Rican literature. From 2002 there is an informative article detailing the clinical findings, risk factors, and workup (Porras Velásquez & Pérez Herra, 2002). In 2006, the Costa Rican medico-legal journal published an article including history of the diagnosis of AHT, clinical findings, and autopsy findings. In addition to mentioning the need for a high index of suspicion in making the diagnosis, it describes the medicolegal framework within the Costa Rican system, and specifically details that reporters of child abuse are both obligated to report and protected from liability if reporting under Costa Rican law (Bonilla Montero, Vargas Sanabria, & Mora Guevara, 2006). A 2008 article is again a discussion of the presentation and legal framework (Alvarado Guevara & Garro Vargas, 2008), and a 2010 article also addresses the medicolegal framework and specifically the protection of mandated reporters by the law, even including a transcript of the pertinent law in the text of the article (Madrigal-Ramírez, 2010). 3.5. Cuba Four articles are found in the Cuban literature. In 2004, a case series from one hospital was published regarding child maltreatment. In this series there were injuries consistent with abusive head trauma (AHT), but “shaken baby syndrome” was not specifically discussed (Fernández Couce & Perea Corral, 2004). Also in 2004 there was a community survey done to determine the incidence of shaking as reported by caregivers (Río-Victoria, 2004). This study was somewhat difficult to interpret, as shaking during playing was included in the survey. A 2008 study regarding an educational program to reduce shaken baby syndrome also mentioned play as a potential etiology (Fonseca-Marbelis, Osorio-Leyva, Santos-Padilla, & RegisAngulo, 2008). A 2011 article about ocular lesions in child abuse mentions retinal hemorrhages in shaken baby syndrome (Beatriz, 2011). 3.6. Mexico Four articles are found in the Mexican literature. A 1995 article is case report of an infant with abusive head trauma (Delgado Fernández, 1995). A 2001 article describes a case of abusive head trauma, reviewing typical features and the diagnostic dilemma (Perea-Martínez, Loredo-Abdalá, Trjo-Hernández, & Monroy, 2001). A 2004 article gives a comprehensive overview of the diagnosis of AHT, sequelae, and some strategies for prevention. As a caution to practitioners, it also lists a series of events that do not cause the lesions of abusive head trauma, but are often cited by caregivers as part of an inaccurate history (Barriga-Marín, Ramos-Gutiérrez, & Barrón-Balderas, 2004). A 2008 article describes physiologic and
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anatomic mechanisms of head trauma in children, and describes non-accidental trauma as one of the causes of head injury ˜ (Garduno-Hernández, 2008). 3.7. Spain Nine articles were identified in the Spanish literature. The first article is a 1999 series of two cases and a description of the diagnosis (Abenia-Uson, Guirado-Giménez, & Rábano, 1999). Next is a 2001 case report regarding a case where exact etiology was unclear, but was judged by the authors to be innocent shaking by the caregivers when the patient (7 months old) was crying or having an episode of perceived apnea (Pascual-Castroviejo, Pascual Pascual, Ruza-Tarrio, Viano, & Garcia-Segura, 2001). This assumption that resuscitation (specifically in this article, the stimulation of an apneic infant by shaking) can cause findings consistent with abusive head trauma is cited in several other articles in this survey, including the next article in the review. This article is a case report of a child from 2003 with presumed complex febrile seizures who had findings consistent with abusive head trauma (Sanchez-Gimeno et al., 2003). An article in 2005 reviewed a case with findings consistent with abusive head trauma but also osteogenesis imperfecta, calling into question the presence of abuse from the point of view of the authors (Cabrerizo de Diago et al., 2005). Two articles from 2006 give a general description of the AHT and strategies for prevention (Aguilar Serrano, Reyes Morillas, Raez Liebanas, & Ruiz Rodriguez, 2006; Rufo-Campos, 2006). A case presentation from 2007 describes stroke in shaken baby syndrome (this article is from a Brazilian author) (Fernandes, Pala, Ferreira, & Jasmin, 2007). An article from 2014 cites two examples of febrile children with seizures who had findings consistent with abusive head trauma (Fernandez-Menendez et al., 2014). An article from Argentinian authors (but in the Spanish literature) in 2015 details a case study and different methods of neuroimaging (Grana, Nazar, de Luca, Casalini, & Eyheremendy, 2015). 3.8. United States (Puerto Rico) Puerto Rican literature contains one article which describes a parental education program designed to reduce the incidence of shaken baby syndrome (Mirabal Rodriguez, Pascual Marrero, Diaz Ortiz, Rios, & Marrero Rivera, 2011). 3.9. International literature A 2009 informational bulletin from the World Health Organization published in multiple languages (including Spanish) mentions shaken baby syndrome in the context of a review of prevention programs internationally (Mikton, 2009). 4. Discussion Literature on abusive head trauma and “shaken baby syndrome” available in the Spanish language medical literature is of limited scope, despite there being over 400 million primary Spanish speakers in the world (Central Intelligence Agency, 2015). Our initial search of 3 databases, prior to eliminating duplicates and articles that did not meet our criteria, yielded 89 articles, while a cursory search of one database in English yielded almost 900 articles. After refining our search as previously described, we identified 34 unique, accessible articles. This presents obstacles for clinicians trying to learn about the diagnosis who rely on the Spanish language literature for education. Paucity of literature could be an indication of a diagnostic blind spot for this area in Global Health, although there may be articles published in English from these countries. Nevertheless, even if there are authors from these Spanish speaking countries publishing in English, that literature is still inaccessible to physicians who read only in Spanish. There were no studies detailing incidence of AHT in the Spanish speaking countries, and for several large countries no literature was located. As the medical communities around the globe strive to work together to improve child health, it is important to recognize that we come from a diverse collection of training and backgrounds. Many of the articles identified are case reports, some of which have entered the literature only within the last few years. Several of the case reports reference articles in Spanish with information regarding diagnosis and management of AHT. These articles were either already located in the original search, or were added during the “snowball” search (see “Methods” section). The country which contributes the largest number of articles is Spain. In Latin America, the origin of the articles, and the number of articles from each country does not appear to be related to the size of the country. In our search, Mexico, the most populous Spanish speaking Latin American country (population approximately 121 million) had four articles. The next largest, Colombia (48 million) had three articles, and Argentina (3rd largest – 43 million) had three articles. The 4th and 5th largest Spanish speaking Latin American countries (Peru – 31 million, and Venezuela – 31 million), did not yield any articles in our search. Chile had the most articles in Latin America with 5 (population 17 million), and Cuba had four articles (population 11 million). Notably, Costa Rica (17th largest – 5 million) also had four articles and was the only country to have physician reporter protections described in their literature. All of these articles would be available to those searching from other countries in a similar manner to our search. However, while helpful, many contain country specific legal information which would not be relevant. Again, there is no incidence or prevalence information available for any country. The information given in the articles was, for the most part, consistent with the English language medical literature which was extensively cited in many of the articles found by the search. One consistent theme in the literature was an emphasis on holistically treating the family to aid in prevention (Barriga-Marín et al., 2004; Címbaro Canella et al., 2010). There were some discrepancies in opinions regarding etiology of lesions consistent with AHT, with articles in the search mentioning
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shaking to awaken children from apnea (Spain) and games including throwing children in the air as possible causes of the lesions of abusive head trauma (Colombia and Cuba). In the first case, this assertion was repeatedly cited in other articles in the review, sometimes expanding the definition to include all resuscitation. The evidence in the English language literature does not support these as etiologies that are consistent with the lesions of AHT. This difference could be clinically significant in both cases, as causal mechanisms could be characterized differently depending on context and physician education. As mentioned above, most articles were case studies or articles with general information. One of the more interesting findings in our review was the substantial and high quality literature from Costa Rica. Though all in the last 13 years (since 2002), it was an impressive treatment of the subject. This literature included copious detail regarding the legal environment from the perspective of the physician reporter. In all of the articles that were located in this review, two of the Costa Rican articles were the only articles to discuss mandated reporting and physician protection from legal consequences. The legal framework was specifically cited in both articles, and one directly quoted from existing Costa Rican law. This is in contrast to Peru, the largest Spanish speaking country to yield no articles in our search, which appears to have mandated reporting, but ˜ no legal protection for physician reporters detailed in their legal code (“Ley No 27337.- Aprueba el Nuevo Código de los Ninos y Adolescentes”). In a diagnosis such as abusive head trauma, which by its very nature usually comes with an inconsistent history that does not fit the clinical situation, the ability of a physician to consider this diagnosis within a supportive legal framework would be key. Our findings suggest that an important point of future investigation could be whether lack of protection from legal liability discourages the investigation of AHT in a large country like Peru. Limitations of this study are related to the search mechanism, terminology, and language. The search only included journal articles, so information in textbooks and other alternative sources was not included, and it is possible that these media are being used by some physicians for education. There is also no reliable method to estimate what fraction of the non-academic physicians in Spanish speaking countries read English well. While it is investigator experience that this fraction can be quite low, there may be variation by country. Finally, it is possible that there are other articles that were not picked up in the search algorithm. However, three separate search engines were employed, and a snowball search was done in addition (searching references of the located articles for additional articles). If the articles were not located by the search algorithm used for this article, they could easily be missed by Spanish speaking clinicians searching the literature as well. Paucity of literature on AHT could reflect a paucity of literature about child abuse in general. The lack of some other types of medical literature has been previously documented in Latin America, which could also reflect an overall literature deficit (Castillo, Pletikosic, & Pizarro, 2009). The contrasting situations of Costa Rica and Peru detailed above invite a comprehensive review across countries of the relationship of legal statutes, reporting rates, and literature content of not only AHT, but other forms of child abuse as well. There may be actual differences in rates of different forms of abuse in different countries, but it is also conceivable that the legal and cultural environment is shaping reporting and diagnosis patterns. For instance, a recent review of five years of data in a major urban pediatric hospital in Peru revealed that 6.8% of all child maltreatment cases were physical abuse in contrast to a 16% fraction in the United States (Escalante-Romero et al., 2012; Hymel & Deye, 2011). Relative rates of other types of abuse (sexual abuse, neglect, etc.) differed from U.S. rates as well, suggesting either a real difference in frequency of types of abuse or a significant difference in diagnostic patterns. In either case, it is reasonable to assume that the content of the medical literature would be affected. Abundant opportunities exist for future investigation into child abuse incidence, variable diagnostic patterns, and physician and community attitudes. 5. Conclusions The Spanish language literature regarding AHT is sparse compared to the English language literature. There are wide differences in the medical literature between individual countries, both in number of articles and in subject matter. There is a lack information regarding incidence of AHT and misleading information in several articles regarding etiology. This relative lack of literature in Spanish could contribute to decreased awareness of the diagnosis among physicians who read only Spanish. It is key to identify such differences so that we can productively collaborate in our increasingly global community and work to identify each other’s blind spots. References Abenia-Uson, P. F.-S., Guirado-Giménez, S., & Rábano, F. J. A. (1999). Encefalopatía aguda y colección pericerebral en el “síndrome del lactante sacudido”. noles de Pediatría, 50, 610–612. Anales Espa˜ Aguilar Serrano, A., Reyes Morillas, M., Raez Liebanas, A., & Ruiz Rodriguez, C. (2006). [The shaken baby syndrome]. Revista de Enfermería, 29(11), 6–10. ˜ sacudido [The shaken baby syndrome]. Medicina Legal de Costa Rica, 25(1), 37–42. Alvarado Guevara, A. T., & Garro Vargas, K. (2008). El síndrome del nino ˜ Zarandeado. Estudios Sobre las Familias, 3, 5–14. Barriga-Marín, J. A., Ramos-Gutiérrez, R., & Barrón-Balderas, R. (2004). Síndrome del Nino ˜ maltratado [Ophthalmologic alterations in the shaken baby syndrome]. Revista Cubana Beatriz, Z. A. (2011). Alteraciones oculares en el síndrome del nino de Pediatría, 117–119. ˜ y el nino ˜ sacudidos: Hallazgos anatomoclínicos [Shaken baby Bonilla Montero, R. A., Vargas Sanabria, M., & Mora Guevara, A. (2006). Síndrome de la nina syndrome: Anatomoclinic features]. Medicina Legal de Costa Rica, 23(1), 103–112. Cabrerizo de Diago, R., Urena-Hornos, T., Conde-Barreiro, S., Labarta-Aizpun, J., Pena-Segura, J. L., & Lopez-Pison, J. (2005). [Shaken baby syndrome and osteogenesis imperfecta]. Revista de Neurologia, 40(10), 598–600. 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Castillo, D., Pletikosic, C. C., & Pizarro, X. A. F. (2009). Estudios clínicos controlados aleatoreos, en la pediatría de Latino América (1996–2005) [Randomized controlled clinical studies in Latin American pediatrics (1996–2005)]. Revista Chilena de Pediatría, 80(5), 420–426. Central Intelligence Agency. (2015). The World Factbook.. Retrieved from https://www.cia.gov/library/publications/the-world-factbook/rankorder/ 2119rank.html Christian CW Block R Committee on Child Abuse and Neglect American Academy of Pediatrics. (2009). Abusive head trauma in infants and children. Pediatrics, 123(5), 1409. Címbaro Canella, R., Clemente, D., González, N., Indart, J., & De Lillo, L. (2010). Descripción del caso presentado en el número anterior: Síndrome del bebé sacudido [Shaken baby syndrome]. Archivos Argentinos de Pediatria, 108(2), 171–172. ˜ sacudido: Artículo de revisión [Shaken baby syndrome: Coria De La, H., Canales, C. F., Avila, L. V., Castillo, P. D., & Correa, P. A. C. (2007). 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