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Pediatrics International (2014) 56, 378–381

doi: 10.1111/ped.12255

Original Article

Intracranial hemorrhage in infants with cephalohematoma Heng Mi Kim,1 Soon Hak Kwon,1 Sook Hyun Park,1 Yong Sun Kim2 and Ki Won Oh3 Departments of 1Pediatrics and 2Radiology, Kyungpook National University School of Medicine, Daegu and 3Department of Pediatrics, Ulsan University School of Medicine, Ulsan, Korea Abstract

Background: Intracranial hemorrhage (ICH) is a rare birth injury in term infants. Newborn infants with cephalohematoma (CH) associated with ICH, however, have frequently been found incidentally at Kyungpook National University Hospital; many of them had no neurological symptoms. The aim of this study was to evaluate the clinical manifestations of ICH in newborn infants with CH. Methods: Newborn infants with CH in the neonatal intensive care unit were retrospectively evaluated. During period I (5 years), neuroimaging (brain computed tomography and magnetic resonance imaging) was performed when intracranial abnormalities were suspected. During period II (36 months) neuroimaging was performed when CH > 5 cm in diameter was present. Results: During period I, seven out of 19 infants who underwent neuroimaging had ICH (36.8%) including two epidural hemorrhages (EDH). During period II, 18 out of 27 infants who underwent neuroimaging had ICH (66.7%), including two EDH. There was no significant difference in the clinical manifestations between infants with and without ICH. In 10 cases of CH accompanied with a linear skull fracture, nine had ICH, including all cases of ICH that needed intervention. Conclusions: The association of ICH appears to be common in newborn infants with CH; particularly in infants with CH accompanied with a skull fracture, the rate of ICH was very high, and all cases of EDH requiring intervention were associated with skull fracture. Therefore, evaluation of accompanying skull fracture should be required in infants with CH, and, in cases of skull fracture, neuroimaging should be considered.

Key words cephalohematoma, epidural hemorrhage, intracranial hemorrhage, newborn, skull fracture, subdural hemorrhage.

Intracranial hemorrhage (ICH) is thought to be a rare birth injury in term infants, having an incidence of 5.9/10 000 live births.1 Although the outcome of ICH is dependent on the severity of bleeding and location, prompt detection and appropriate management of hematoma have also been important for improvement of outcome.2 Cephalohematoma (CH) is a not uncommon birth injury of newborns. It occurs in 2.5% of newborns.3 Given that most CH have spontaneous resolution without complications,2,4 CH has been considered to be a benign birth injury that requires no specific evaluation and treatment. As a result, neuroimaging is indicated only when neurological symptoms develop or the possibility of a depressed skull fracture exists.4 Assisted vaginal delivery, which was reported to increase the rate of CH up to 10.8%,5 also increased the frequency of ICH to 8–10/1000 live births6 and many cases of epidural hemorrhage (EDH) in the literature are associated with CH.7–11 Thus, in cases of CH, the ICH might occur more frequently than in infants without CH. Correspondence: Heng Mi Kim, MD, Department of Pediatrics, Kyungpook National University School of Medicine, 2-50 Samduckdong, Jung-gu, Daegu 700-721, Korea. Email: [email protected] Received 23 May 2012; revised 10 October 2013; accepted 5 November 2013.

© 2013 The Authors Pediatrics International © 2013 Japan Pediatric Society

Recently, neuroimaging performed on asymptomatic newborn infants after normal vaginal delivery indicated a high prevalence of ICH.12,13 Therefore, even in the asymptomatic newborn with CH, prevalence of ICH could be high, and CH would be an indicator of ICH in newborn infants. Few studies of the frequency of ICH in newborns with CH, however, have been conducted. The aim of this study was to evaluate the clinical manifestations of ICH in newborn infants with CH.

Methods The medical records of infants with CH who were admitted to the neonatal intensive care unit (NICU) of Kyungpook National University Hospital (KNUH) between 1 January 2001 and 31 December 2008 were retrospectively reviewed. The study was divided into two periods: period I, 1 January 2001–31 December 2005 (60 months), and period II, 1 January 2006–31 December 2008 (36 months); during period I, neuroimaging was performed only when a history or symptoms and/or signs were highly suggestive of intracranial abnormalities. During period II, neuroimaging was performed in infants with CH > 5 cm in diameter to rule out the presence of ICH. All computed tomography (CT) and magnetic resonance imaging (MRI) findings were reviewed by a neuroradiologist (Y.K.) and a pediatric neurologist (S.K). There were no changes of staff, equipment and imaging protocols during the study periods. Infants with abnormal

ICH in cephalohematoma

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Table 1 Characteristics of infants with CH

CH/total admission (%) Birthweight (g) Gestational age (weeks) Age on admission (days) M: F Maternal age (years) 5 min Apgar score < 7 Indication for neuroimaging Neuroimaging† (%/CH) ICH total (%/neuroimaging) SDH EDH IVH‡ Neurologic abnormalities (%/ICH)

Period I 47/1797 (2.6) 3269 ± 463 38.8 ± 1.7 6.4 ± 6.5 29:18 30.9 ± 3.5 1 Neurologic abnormalities 19 (40.4) 7 (36.8) 3 (15.8) 2 (10.5) 2 (10.5) 7 (100)

Period II 31/1106 (2.8) 3274 ± 463 39.0 ± 1.4 4.5 ± 4.9 19:12 31.0 ± 4.0 1 Cephalohematoma > 5 cm 27 (87.1) 18 (66.7) 15 (55.6) 2 (7.4) 1 (3.7) 7 (38.9)

P NS NS NS NS NS NS NS 0.0037 0.0005 0.043 NS NS 0.008

† Brain computed tomography and/or magnetic resonance imaging; ‡including one SDH/SAH and one SDH/IVH. CH, cephalohematoma; EDH, epidural hemorrhage; ICH, intracranial hemorrhage; IVH, intraventricular hemorrhage; SAH, subarachnoid hemorrhage; SDH, subdural hemorrhage.

coagulation profile (hemophilia, thrombocytopenia and disseminated intravascular coagulation) and central nervous system (CNS) anomalies were excluded. SPSS version 17.0 (SPSS Chicago, IL, USA) was used for data analysis. Statistical analysis was done using the chi-squared test and Fisher’s exact test. Informed consent by parents was obtained before neuroimaging and the study was approved by the institutional review board of Kyungpook National University Hospital.

Results During the study periods, 78 out of 2903 infants admitted to the NICU had CH (2.7%). The frequency of CH did not differ between periods I and II. CT and/or MRI were performed in 40.4% of infants with CH during period I, and in 87.1% during period II; this increase was the result of practice changes in neuroimaging. Among the infants who underwent neuroimaging, ICH was detected in 25 infants: seven out of 19 infants (36.8%) during period I, and 18 out of 27 infants (66.7%) during period II. The detection rate was significantly higher during period II compared to period I (P = 0.046). Subdural hemorrhage (SDH) was the most common ICH detected during both periods (18/25, 72.0%). It was found more frequently during period II than period I (15.8% vs 55.6%, P = 0.043). EDH was noted more frequently during period I than during period II, but its frequency did not significantly differ between the two periods (10.5% vs 7.4%, P > 0.05). Contrary to the higher detection rate of ICH, fewer neurologic abnormalities were identified in ICH during period II compared with period I (38.9% vs 100%, P < 0.008; Table 1). Among 18 infants with ICH detected during period II, 7 (38.9%) and 10 infants (55.6%) had neurologic (seizure, decreased activity, irritability and tachypnea) and hematologic manifestations (hyperbilirubinemia and anemia), respectively, but compared with infants without ICH, there was no significant difference in these clinical manifestations according to the presence of ICH (Table 2).

All cases of ICH except three EDH were treated conservatively. Two EDH were evacuated surgically and one EDH resolved after aspiration of the associated CH, which communicated with the epidural space through a linear fracture. These cases were transferred to NICU for evaluation of hyperbilirubinemia and rapid enlargement of CH, respectively. On formal neurological testing, these two surgically treated EDH were normal and remained so until intervention. Among 25 infants with ICH, 21 infants including all EDH were followed more than 12 months and had normal developmental outcome (Table 3). Infants were reevaluated according to the presence of skull fracture. Because skull radiography was not done in most of the studied infants, correlation between skull fracture and ICH was assessed in 36 infants who underwent brain CT. Ten infants were Table 2 Clinical characteristics vs presence of ICH in period II

Neurologic Asphyxia Seizure Decreased activity Irritability Tachypnea† Hematologic (%) Hyerbilirubinemia (Total bilirubin) (mg/dL) Anemia No clinical manifestations Congenital anomaly‡ MAS Chorioamnionitis

Neuroimaging (n = 27) ICH No ICH (n = 18) (n = 9) n (%) n (%) 7 (38.9) 3 (33.3) 0 1 1 2 2 0 2 0 2 0 10 (55.6) 2 (22.2) 7 2 (16.9 ± 8.4) (13.7 ± 7.3) 3 0 1 (5.6) 4 (44.4) 0 3 1 0 0 1

P

NS

NS NS NS

† Tachypnea without abnormalities on chest auscultation and radiology. ‡Congenital heart disease, n = 2; hydronephrosis, n = 1. ICH, intracranial hemorrhage; MAS, meconium aspiration syndrome.

© 2013 The Authors Pediatrics International © 2013 Japan Pediatric Society

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Table 3 Clinical course of infants with epidural hemorrhage

Age on admission (days) Birthweight (g) Gestational age (weeks) Apgar score (1 and 5 min) Birth place Mode of delivery Parity Presenting Symptom Hematocrit at admission, % Peak Bilirubin, mg/dL Skull fracture Management Outcome (age, months)

Patient 1 3 3100 40 >7 Outborn Normal vaginal Multiparous Jaundice 31 15 + Surgical drainage Normal (37)

Patient 2 5 3000 40 >7 Outborn Vacuum Multiparous Large CH 29 6.1 + Surgical drainage Normal (30)

found to have fractures (27.8%); all fractures were linear. The frequency of ICH was significantly higher in infants with fracture than in infants without fracture (90% vs 50.0%, P = 0.027). Fractures more likely occurred with larger CH compared to the smaller CH (8.4 ± 1.9 cm vs 6.0 ± 2.0 cm, P = 0.003), in outborn infants compared to inborn infants (12/14 vs 1/9, P = 0.006) and in boys compared to girls (9/10 vs 10/26, P = 0.006). There were no significant differences with regard to low Apgar score at 5 min, complicated deliveries, primiparity, maternal age and cesarean section delivery. In addition, there were no cases of EDH without an associated skull fracture (Table 4).

Discussion We unexpectedly found a high incidence of ICH in infants with CH. In this study, 25 ICH (18 SDH, 4 EDH and 3 IVH) were found on neuroimaging performed on 46 newborn infants with CH (54.3%). SDH and EDH were detected in 39.1% (18/46 on neuroimaging) and in 8.7 % (4/46 on neuroimaging), respectively. The incidence of ICH has been estimated to be 3.6–17 per 10 000 live births,1,14,15 whereas that of SDH is 2.9 per 10 000 live births in spontaneous vaginal deliveries to 21.3 per 10 000 live

Table 4 Clinical characteristics vs presence of skull fracture on CT CT (n = 36) Fracture No fracture n (%) n (%) No. infants Size of cephalohematoma (mm) Intracranial hemorrhage M:F Inborn : Outborn Low Apgar score (4000 g or ≤2500 g Prematurity (7 Outborn Normal vaginal Primiparous Lethargy 48 16.5 + needle aspiration Normal (33)

Patient 4 2 3590 40 >7 Outborn Cesarean section Primiparous Seizure 41 16.2 + Conservative Normal (40)

births in vacuum and forceps deliveries.6,14–16 Extremely low rates of EDH are reported in the literature, in which EDH constituted only 2.2% of neonatal ICH observed at autopsy;17 further, only 31 cases were identified until 1995.9,11 Compared to the data, infants in this study had a considerably frequent association of ICH. Additionally, more ICH and SDH were detected when the neuroimaging was extended to neurologically asymptomatic infants (period II, 66.7% and 55.6%, respectively) compared to the period when neuroimaging was limited to only neurologically abnormal infants (period I, 36.8% and 15.8%, respectively; P < 0.05). In contrast, the detection rate of EDH did not differ significantly between the two periods (10.5% vs 7.4%, P > 0.05). Common presenting findings of ICH in newborn infants in previous reports were apnea or seizures,6,18 focal neurological deficits, hypotonia or lethargy, and a full anterior fontanel.6,16 In this study, these manifestations were detected only in 38.9% of infants with ICH. Moreover, when comparing these clinical manifestations between infants with ICH and without ICH, no difference was seen. This suggests that the clinical manifestations in the present infants were not likely due to the ICH itself. Because this study was performed on newborns admitted to the NICU, the majority of these manifestations appear to be those of associated illnesses. Also, there is a considerably higher detection rate during the period in which neuroimaging was performed extensively (period II). These findings indicate that most ICH, particularly SDH, which was the most common ICH, were asymptomatic and were incidentally detected on neuroimaging. Little information is available on the incidence of SDH in asymptomatic infants, with conflicting results. Ludwig et al. reported that asymptomatic ICH has been rare, and all neonates with severe intracranial bleeding had neurological abnormalities,19 whereas other studies noted a considerably higher incidence of asymptomatic SDH. Asymptomatic SDH had been detected in 6.1–62% of term infants after uncomplicated deliveries.12,13,20,21 Such asymptomatic SDH resolved completely13 and was also benign.20 In the present study, SDH was detected in 55.6% during period II. All SDH and one EDH in this study were managed conservatively. Three ICH that required evacuation were EDH. Two of the three EDH had no neurological

ICH in cephalohematoma manifestations on admission. The presenting findings were hyperbilirubinemia and rapid enlargement of the CH. This made early diagnosis of EDH difficult, even in cases that required intervention. This prompted us to evaluate the risk factors or indicators of EDH. Frequent association of EDH and skull fracture was identified in the literature.8–11,22 In the present study, nine out of 10 skull fractures in CH were associated with ICH, and three required intervention. Based on these findings, CH complicated with skull fracture appears to be an indicator of associated ICH. Infants with very small EDH and stable clinical course may be managed conservatively; but no randomized trials have compared surgical evacuation with conservative management for newborn infants with EDH. In addition, newborn infants with EDH present with non-specific neurologic symptoms and have the potential to deteriorate quickly. Therefore, the evaluation of skull fractures in infants with CH seems to be necessary, especially in large CH, male and outborn infants. We recommend routine skull radiography in infants with large CH. The majority of ICH in the present study was benign and needed no intervention; hence, routine neuroimaging on CH will not be necessary. The prevalence of ICH complicated with skull fracture, however, seemed to be considerably increased, and required intervention (30%); neuroimaging should be considered in CH complicated with skull fracture. Epidural hemorrhage was evacuated surgically in two cases and by needle aspiration of CH in one case. Although surgical evacuation is the standard management of ICH, needle aspiration was equally effective and easier to perform in the NICU.8–11,22 For this reason why chose needle aspiration as the first-line management in patient 3. The limitations of this study include the small sample size as well as the retrospective study design. In addition, no specific information was available on infants born outside of the hospital. Furthermore, the long-term outcome of such patients is needed to fully understand patient prognosis.

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© 2013 The Authors Pediatrics International © 2013 Japan Pediatric Society

Intracranial hemorrhage in infants with cephalohematoma.

Intracranial hemorrhage (ICH) is a rare birth injury in term infants. Newborn infants with cephalohematoma (CH) associated with ICH, however, have fre...
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