Abstract

Background: Cephalohematomas (CH) are benign neonatal fluid collections that arise between the periosteum and skull due to birth trauma, and usually resolve spontaneously without intervention. CH may rarely become infected.

Case Description: The authors report a case of sterile CH requiring surgical evacuation in a persistently febrile neonate treated with intravenous (IV) antibiotics for Escherichia coli urosepsis. Diagnostic tap of the CH yielded no pathogens, but given the persistence of fevers, surgical evacuation was performed. The patient demonstrated clinical improvement postoperatively.

Conclusion: A systematic review of literature was conducted through a MEDLINE search using the keyword “cephalohematoma.” Articles were screened for cases of infected CH and their subsequent management. Clinicopathological characteristics and outcomes of the present case were reviewed and compared to those in the literature. Infected CH were reported in 25 articles describing 58 patients. Common pathogens included E. coli and Staphylococcal species. Treatment included a course of IV antibiotics (10 days–6 weeks) and often included percutaneous aspiration (n = 47) for diagnostic and therapeutic purposes. Surgical evacuation was performed in 23 cases. To the authors’ knowledge, the present case is the first documented report in which evacuation of a culture-negative CH resulted in resolution of the patient’s clinical symptoms of sepsis that persisted despite appropriate antibiotic treatment. This suggests that patients with CH should be evaluated through diagnostic tap of the collection if there are signs of local or persistent systemic infection. Surgical evacuation may be indicated if percutaneous aspiration does not result in clinical improvement.

Keywords: Birth trauma, Cephalohematoma, Infection, Pediatric neurosurgery

INTRODUCTION

Cephalohematomas (CHs) occur in 1–4% of live-births, often associated with birth trauma.[ 3 , 12 , 24 ] CH is usually self-resolving with minimal sequelae, but bacterial infection may occur in rare instances.[ 3 , 22 , 24 , 26 ] Herein, we report the case of an infant with Escherichia coli sepsis and a preexisting CH who had persistent fevers despite treatment with intravenous (IV) antibiotics. To the author’s knowledge, this is the only reported case in which clinical signs of systemic infection resolved only after surgical incision and debridement (I&D) of a sterile CH.

METHODS

A systematic MEDLINE search was conducted using the keyword “cephalohematoma.” The reference lists from these publications were examined for relevant literature and historical reports [ Figure 1 ]. Reports were excluded if they described non-infectious complications of CH, if the articles were not published in English literature, or if full articles were unavailable to review.

Figure 1:

PRISMA flow diagram. Citations identified and evaluated on literature review of infected cephalohematomas. Moher D, Liberati A, Tetzlaff J, Altman DG, The PRISMA Group (2009). Preferred Reporting Items for Systematic Reviews and Meta-Analyses: The PRISMA Statement. PLoS Med 6(7): e1000097. doi:10.1371/ journal.pmed1000097

Clinicopathological characteristics and outcomes of the present case were compared to those in the literature. The patient’s chart was reviewed for demographic data, clinical presentation, hospital course, relevant imaging, and follow-up using our electronic medical record system. Clinical case review was deemed not human subjects research and exempt from approval by our Institutional Review Board.

CASE DESCRIPTION

A male infant was born at term through uncomplicated vaginal delivery without instrument-assistance or fetal scalp electrodes. A left-sided CH was noted post-partum. At 3 weeks old, he presented to an outside institution with 1-day of fever, emesis, and diarrhea. He was started empirically on IV ampicillin and gentamicin, and infectious workup later revealed pan-sensitive E. coli bacteremia, urinary tract infection, and meningitis. After initial clinical improvement, he was transitioned to monotherapy with IV ampicillin. Shortly thereafter, he became febrile again, so cultures were redrawn and a head ultrasound performed, which was largely unrevealing. The patient was transferred to our institution for further management [ Table 1 ].

Table 1:

Notable laboratory findings and events for our presented case throughout the course of his hospitalizations (abnormal values in red).

On arrival, he was noted to have a large, benign-appearing CH, and a reassuring physical examination, despite being febrile with a rising C-reactive protein (CRP). Magnetic resonance imaging (MRI) of the brain did not reveal any acute intracranial processes or definitive signs of CH infection [ Figure 2 ]. Over the following days, the patient was transitioned to IV ceftriaxone for an up-trending fever curve [ Figure 3 ]. A sterile bedside tap was performed with return of 2 mL of bloody aspirate; cultures and gram stain were negative. The patient remained intermittently febrile and after a multidisciplinary discussion, he was taken for an I&D. Intraoperatively, no gross purulence or abnormalities of the underlying bone were identified. Histopathological examination confirmed a diagnosis of CH without infection or organisms. All surgical cultures and gram stain were negative. The patient tolerated the procedure with no complications.

Figure 2:

(a-h) Magnetic resonance imaging (MRI) study of the patient’s brain on presentation to our institution. Select axial T2-weighted non-contrast (a), T1-weighted non-contrast (b), T1-weighted post-contrast (c), gradient echo (d), diffusion-weighted imaging (e), apparent diffusion coefficient (f), sagittal T1-weighted non-contrast (g), and coronal T1-weighted non-contrast (h) images from an MRI study of the patient’s brain. Images demonstrate a large heterogeneous extracranial fluid collection overlying the left parietal skull consistent with a cephalohematoma. The collection does have some enhancement along its margins, but does not demonstrate restricted diffusion. No significant surrounding subcutaneous edema is identified. No acute intracranial findings or abnormalities noted.

Figure 3:

Documentation of patient’s vital signs during his admission at our institution. Patient’s body maximum temperature in °C (a) and median recorded heart rate (beats per minute; bpm) (b), as charted per nursing documentation for each 24-h period throughout the course of the patient’s admission. Patient continued to be intermittently febrile (>38°C or 100.4°F; red line) and tachycardic (>160 bpm; red line) until after the patient was taken to the operating room for surgical I&D of the cephalohematoma, on his 7^th day of admission to our institution (grey rectangle).

Our patient became afebrile on postoperative day (POD)1 and remained clinically improved for the remainder of his hospitalization [ Figure 3 ]. After surgical drain removal and PICC line placement, he was discharged on POD6 with a 21-day course of IV ceftriaxone. The patient was seen in clinic on POD12 and noted to be doing well with no reaccumulation of the CH.

Literature review

A MEDLINE search was conducted using the keyword “cephalohematoma.” Full-text, English-language articles were reviewed for management and outcomes of infected CH. This resulted in 25 publications, documenting 58 cases [ Table 2 ].[ 1 - 4 , 6 , 7 , 9 , 10 , 12 - 21 , 23 - 29 ]

Table 2:

Literature review of previously reported cases of infected CH.

The majority of cases 52% (n = 30) had one or more concurrent infections,[ 1 - 4 , 6 , 7 , 12 , 13 , 15 , 17 , 18 , 21 , 24 , 25 , 27 , 28 ] while 48% (n = 28) did not have any.[ 1 , 3 , 9 , 10 , 14 , 16 , 19 , 20 , 23 , 26 , 29 ] E. coli was the most common organism isolated (n = 32; 55%),[ 1 , 3 , 4 , 7 , 9 , 12 , 13 , 15 , 17 , 21 , 23 , 25 - 29 ] followed by polymicrobial infections (n = 8; 14%).[ 2 , 10 , 18 ] Only one case reported no growth in the CH culture despite Gram-negative rods noted on gram stain.[ 20 ]

All patients were treated with IV antibiotics; some followed by a course of oral antibiotics. Duration varied from 10 days to 6 weeks, depending on concerns for osteomyelitis or other concurrent infections. The majority were treated with needle aspiration alone (n = 34/58).[ 1 - 3 , 24 , 26 ] Surgical I&D was performed in 23 cases,[ 1 , 2 , 4 , 6 , 7 , 10 , 12 , 14 - 21 , 23 , 25 , 27 - 29 ] sometimes after needle aspiration (n = 13). One case was successfully treated with IV antibiotics alone.[ 9 ] The most common complications of infected CH were osteomyelitis (n = 10),[ 1 - 3 , 6 , 9 , 16 , 19 , 20 , 24 , 25 ] and overlying scalp necrosis or abscess (n = 9).[ 2 , 9 , 12 , 17 , 18 , 20 , 26 , 28 ] The mortality rate of cases reported in our literature review was 8.6% (n = 5/58; range 0–11%).[ 1 , 3 , 18 ]

DISCUSSION

CH infection is rare and may occur early (<2 weeks of life) due to direct birth trauma or hematogenous seeding, or late (≥3 weeks of life) due to overlying cellulitis.[ 28 ] Clinical manifestations can be local – increasing size, fluctuance, tenderness, erythema, skin abrasion with purulence – or systemic – fever, decreased feeding, irritability. As in the present case, imaging studies may be of limited diagnostic value. MRI can identify associated abscesses or intracranial findings, but may not definitively detect the presence of infection.[ 4 , 13 ] Computed tomography scan may demonstrate bony changes indicative of osteomyelitis, but are not 100% sensitive and bone scan findings may be confounded by local tissue hyperemia.[ 13 , 28 ] In cases without clear local signs of infection, the presence of other sources of infection may lead to a delay in diagnosis of infected CH.[ 1 , 6 , 7 , 13 , 15 , 17 , 19 , 23 - 25 ]

Aspiration and surgical interventions are contraindicated in uncomplicated CH due to risks of contamination with bacteria.[ 13 , 22 ] However, given the lack of adequate noninvasive diagnostic tools, aspiration should be considered in select circumstances: development of local signs of infection, relapse, or delayed resolution of clinical signs systemic infection.[ 13 ] This usually enables identification of a causative pathogen to guide antibiotic therapy and may be therapeutic if enough purulent material is aspirated.[ 3 ] Surgical I&D of infected CH is recommended if insufficient material is able to be aspirated, or in situations of extensive scalp necrosis and abscess formation.[ 1 , 2 , 4 , 6 , 7 , 10 , 12 , 14 - 21 , 23 , 25 , 27 - 29 ] A course of IV antibiotics (1–6 weeks) is indicated, with optimal duration depending on the presence of other concurrent infections.[ 3 ]

The presented case makes a unique contribution to the body of literature because this patient’s CH did not demonstrate evidence of infection on any diagnostic modality. It may be argued that because our patient received IV antibiotics for several days before needle aspiration or I&D, the CH may have been sterilized by appropriate treatment of pan-sensitive E. coli. There has been growing evidence for polymerase chain reaction testing of culture-negative fluid collections that are suspected to have harbored bacterial infections following initiating of empiric antibiotics. This test was not performed for our patient, but may be helpful to reveal a causative organism.[ 8 ] However, our review also includes multiple cases in which patients received appropriate antibiotic treatment for 5–15 days before positive cultures and gross purulence obtained through needle aspiration or I&D.[ 1 , 6 , 7 , 15 , 17 , 23 , 25 , 27 , 29 ] Another possible explanation for this clinical scenario may be that the significant size of the CH and the inflammatory cascade involved in tissue injury and wound healing was sufficient to induce non-infectious pyrexia in this infant.[ 5 , 11 ]

Although the exact mechanism is unclear, the authors believe that CH evacuation was still indicated in this case given our patient’s persistent signs of systemic infection.

CONCLUSION

Optimal management of infected CH often requires a combination of IV antibiotics and evacuation. In the absence of clear local signs of infection, patients demonstrating a relapse or persistent signs of systemic infection should still be considered for definitive CH evacuation, even with negative cultures.

Ethics approval/consent to participate

Due to its retrospective nature, clinical case review was deemed not human subjects research and exempt from approval by the Loyola University Chicago Stritch School of Medicine Institutional Review Board (LU#213917) on July 22, 2020. Therefore, the Loyola University Chicago Stritch School of Medicine Institutional Review Board has waived the need for the patient’s and/or parents’/legal guardian’s written informed consent for publication. All included patient information has been deidentified.

Author contributions

Daphne Li is the first author having performed the majority of data extraction, literature review, data analysis, and writing of the manuscript. John Tsiang contributed to data analysis and drafting of the manuscript. John Tsiang, Kimberly Mackey, and Andrew Bonwit contributed to the critical review of the manuscript. Suguna Pappu is the principal investigator having contributed to the majority of design and review of the manuscript. All authors read and approved the final manuscript.

Declaration of patient consent

Patient’s consent not required as patient’s identity is not disclosed or compromised.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

Disclaimer

The views and opinions expressed in this article are those of the authors and do not necessarily reflect the official policy or position of the Journal or its management. The information contained in this article should not be considered to be medical advice; patients should consult their own physicians for advice as to their specific medical needs.

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