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Rib Fractures in Infancy, Case-Series and Register Case-Control Study From J Pediatr Endocrinol Metab 2021; 34(3): 363–372 Ulf Högberg* and Ingemar Thiblin Rib fractures in infancy, case-series and register case-control study from Sweden https://doi.org/10.1515/jpem-2020-0137 proportion had metabolic risk factors, suggesting false Received March 26, 2020; accepted October 30, 2020; positive cases. published online February 25, 2021 Keywords: abuse; infant; metabolic bone disease; Abstract radiology; rib fractures. Objectives: To assess cases with rib fractures in infants under observation for or with an abuse diagnosis, and to Learning points compare risk factors with that in infants with fractures but no abuse diagnosis. – The case-series showed that a few infants presented Methods: Design was case-series and register case-control with a popping sound, none had notes of admitted or of infants (aged <1 year) with rib fractures. Data was clinical witnessed shaking, none had pain, and physical records for the case-series (n = 26) and from national health examinations did not reveal any findings. Most had registers for the case-controls (n = 28 and n = 31). Outcome callus at time of diagnosis, no systematic radiology measures were maternal and perinatal characteristics, assessments of bone mineralisation were noted. reasons for appointment, examination, diagnoses, blood Analysis of a metabolic panel was scarce. tests, and radiologic findings. – The register case-control study showed that rib frac- Results: The case-series had a median age of 70 days. A tures can be considered as incidental findings; for the majority were detected through a skeletal survey for abuse cases, mainly from skeletal surveys to detect abuse, investigation. Sixteen were boys, three were preterm and and for the controls through X-rays done for other six were small-for-gestational age. Three carers had reasons (neonatal morbidity, lower respiratory tract noticed popping sound from the chest; no infants showed infections, major surgery, and accidents). signs of pain at physical examination. Mean number of – Both the case-series and the register case-control study fractures was 4.2, and 24 had callus. Bone mineralisation had risk factors for metabolic bone disease. The results was scarcely reported. Metabolic panel was not uniformly indicated that there were false positive cases of analysed. The register case-control had a median age of maltreatment. 76 days, sharing risk factors such as maternal overweight/ obesity, male sex, prematurity, and being small-for- gestational age. Cases more often had subdural haemor- Introduction rhage, retinal haemorrhage, or long-bone fractures, controls more often had neonatal morbidity, respiratory infection, or A skeletal survey is recommended in the screening pro- a fall accident. Detection of fracture at time of a major cedure for physical abuse. Symptomatic or incidental surgery (n = 6) and rickets/vitamin D deficiency (n = 5) findings might be rib fractures, which are considered appeared in both groups, but was delayed among the cases. highly indicative of abusive head trauma (AHT) [1–7]. The Conclusions: Rib fractures in young infants, diagnosed as possible non-accidental trauma mechanism causing rib abuse, are usually asymptomatic and healing. A substantial fractures has not been fully established [8], but the most common theory is thoracic compression when the infant is violently shaken [2]. Rib fractures, including multiple *Corresponding author: Ulf Högberg, Department of Women’s and fractures, are rarely reported in association with birth, Children’s Health, Uppsala University, SE-751 85 Uppsala, Sweden; only in one case-series [9] and one population register and Department of Epidemiology and Global Health, Umeå University, study [10]. In both studies, the infants had concomitant Umeå, Sweden, Phone: +46186115246, Mobile: +46703461327, clavicle fractures. In the case-series, the clavicle fracture E-mail: [email protected]. https://orcid.org/0000-0002-2121-7511 was ipsilateral to the rib fractures [9]. Incidental cases of Ingemar Thiblin, Forensic Medicine, Department of Surgical Sciences, Uppsala University, Uppsala, Sweden. https://orcid.org/0000-0002- occult rib fractures have been reported from neonatal 2962-2466 units [11–13], and in infants in critical care for congenital Open Access. © 2021 Ulf Högberg and Ingemar Thiblin, published by De Gruyter. This work is licensed under the Creative Commons Attribution 4.0 International License. 364 Högberg and Thiblin: Rib fractures in infancy heart disease [14], and in association to physiotherapy Table : Definition of codes for diagnoses and accidents for the [15], whereas rib fractures caused by cardiopulmonary register case-control study, Swedish version of the International fi resuscitation (CPR) are rare [16]. Multiple rib fractures statistical Classi cation of Diseases (ICD -SE). might occur in connection with severe thoracic trauma Category Diagnosis Code (ICD-SE) [17], and have been associated with metabolic bone dis- fi eases of infancy, such as nutritional rickets [18], vitamin D Case de nition Rib fracture S. deficiency [19), Metabolic Bone Disease of Infancy (MBDI) Multiple rib fractures S. [20], and osteopathy of prematurity [19, 21, 22]. Symptoms Infant abuse from rib fractures as part of severe thoracic trauma are diagnoses dramatic (pneumothorax, haemothorax, pulmonary Observation for suspected Z.K contusion, and lung lacerations or injury to mediastinum) abuse [17]. However, specific symptoms of pain from rib frac- Battered baby syndrome Y . Maltreatment syndrome T. tures are rarely reported, whether in association with Neglect and abandonment Y prematurity [22, 23] or during physiotherapy [15]. In one Other maltreatment Y case-series, with rib fractures and clinical and medico- Other diagnoses legal referrals, five out of 80 infants cried when they were Retinal hemorrhage H. picked up and, in three cases, parents had noted a Subdural hemorrhage I . ,S . Long bone fractures S.,S.,S,, popping sound [24]. S.,S.,S, We have previously shown that rib fractures in S,S,T,T infants, before the age of six months, are associated with Clavicle fracture S. metabolic bone disease risk factors such as maternal Thoracic injury (pleura, S obesity, male sex, being born preterm, multiple births, lung, bronchus trachea) fi and diagnoses like rickets, vitamin D deficiency, and Super cial injury of T . unspecified body region calcium metabolism disturbances [25]. However, that Rickets E. population register study did not address clinical history Vitamin D deficiency E. and findings among those with a maltreatment diagnosis, Osteogenesis imperfecta Q. nor did it analyse rib fractures with or without a Accidents maltreatment diagnosis. To our knowledge, no previous Transport accidents V - Fall accidents W- nationwide study has addressed the care-seeking history, clinical presentation, or detection of rib fracture in in- fants. The objectives of this study were to describe cases of Case-series rib fractures in infants being observed for abuse or with a diagnosis of maltreatment by medical history, symptoms, Among 337 infants being observed for suspected abuse or with a clinical signs, and radiologic findings and to compare maltreatment diagnosis (Table 1), for whom medical records were exposure of infants with rib fracture who have or do not requested from the paediatric departments, 258 (77%) records were have a maltreatment diagnosis. retrieved. Among the missing records, there were cases (n = 12) in the NPR having both diagnoses of maltreatment and rib fractures. Cases were those having rib fractures diagnosed by x-ray or chest CT (n = 26). Methods Of those, 16 also had an ICD code for rib fracture in the NPR, while 10 did not. One case of rib fractures diagnosed solely by scintigram was excluded (Figure 1). Design Medical records and written accounts of the radiologic exami- nations were reviewed for each patient. Digitalised radiology was The design is a case-series and register case-control study. not available. The following information was retrieved from the Infants (1–365days),bornintheyears1997–2014 records: (1) reasons for seeking care, (2) symptoms, (3) local signs, (4) (N = 1,855,267), with a rib fracture diagnosis (Table 1) were identified localisation, (5) number of fractures per child. in the Swedish National Patient Register (NPR) based on the 10th Specific information for rib fractures was: (a) one side or version of International Classification of Diseases (ICD-10-SE). Data both sides, (b) localisation: posterior, posterolateral, antero-lateral, were linked to the Swedish Medical Birth Registry (SMBR), Death anterior/costochondral), (c) dating: 0–1 week (fresh), 2–4 weeks Cause Register (DCR), and the Register of Children and Young (“old”), more than 4 weeks (“older”). Persons Subjected to Child Welfare Measures (RCWM), as previously Records were checked for any note on findings of abnormal reported [26, 27]. bone mineralisation: (1) periosteal mineralisation abnormality An eligibility flow chart of the case-series and register case- (subperiosteal new bone formation [SPNBF]), (2) growth plate min- control study is presented in Figure 1. eralisation abnormality, (3) ulnar cupping, (4) skull mineralisation Högberg and Thiblin: Rib fractures in infancy 365 Figure 1: Study eligibility flow chart of children born in Sweden 1997 to 2014 (N = 1,855,267) with rib fractures before the age of 1 year identified in the National Patient Register (NPR), Swedish Medical Birth Register (SBMR), Register of Children and Young Persons Subjected
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