Original article
Sub-aponeurotic fluid collections: A delayed-onset self-limiting cerebrospinal fluid fistula in young infants

https://doi.org/10.1016/j.ejpn.2007.10.010Get rights and content

Abstract

Background

Transient sub-aponeurotic fluid collections are rarely recognized lesions that may be frequently related to traumatic labor and occur within weeks after birth. Their etiology has remained uncertain.

Methods and results

Here we report on five new cases with typical clinical findings and normal neuroimaging. A detailed analysis of the lesion aspirates was performed in three patients, always revealing β2-transferrin and high concentrations of β-trace protein.

Conclusions

This indicates that cerebrospinal fluid leakage contributes to this rare condition. All lesions disappeared spontaneously within 2–5 months so that conservative treatment seems appropriate.

Introduction

Skull masses in young infants show a heterogeneous etiology, including congenital (dermoid, epidermoid cyst, encephalocele, lipoma, lymphangioma, naevus), traumatic (sub-aponeurotic hemorrhage (SH), subcutaneous caput succedaneum, subperiostal cephalhematoma), neoplastic (teratoma, neuroblastoma) and vascular (arterial and venous malformation) lesions.1, 2, 3, 4 Whereas most lesions are well described, the etiology of sub-aponeurotic fluid collections (SFC), a disorder recently specified by Hopkins et al., has remained uncertain.5 Like SH, these soft, non-tender and fluctuating swellings may be linked to birth trauma, for example, ventouse-assisted delivery. SFC, however, do not contain large amounts of blood, are not present from birth but develop within few weeks of life. A minor form of SH, disruption of scalp lymphatic drainage and cerebrospinal fluid (CSF) leakage are discussed as etiological factors.5

Section snippets

Patient reports

Five previously healthy children developed occipital fluctuating and painless soft tissue swellings without skin reddening 7 (n=1) and 8 weeks (n=4) after birth, respectively.

Four of the lesions were of large size, one patient showed just a small lesion. Patients 1, 3 and 5 had been delivered by vacuum extraction. In patient 2 cesarean section was performed when external cephalic version failed repeatedly, in patient 4 when vacuum extraction had failed twice and a loud “bang” was noticed. In

Laboratory findings

All aspirates had a serosanguineous appearance (Table 1). Whereas microbiological studies proved normal, the CSF markers β-trace protein (βTP; immunofixation analysis) and β2-transferrin (immunonephelometry) were found in all aspirates (Fig. 2). β2-Transferrin is physiological only in CSF, aqueous humor and perilymph fluid. βTP (Prostaglandin D synthase) represents about 3% of total CSF protein, showing levels that are approximately 35 times higher than in serum.7 Therefore, for example, the

Discussion

Whereas isolated patients with lesions resembling SFC have repeatedly been reported in the literature, the characteristics of this lesion have been delineated only recently by Hopkins et al., summarizing the clinical findings of seven patients.5 Six of these patients had developed SFC between 3.5 and 18 weeks after birth, four of them had been delivered by vacuum extraction. In one patient, microbiological analysis of an aspirate proved normal. In all reported cases, SFC disappeared after 2–24

References (8)

There are more references available in the full text version of this article.

Cited by (18)

View all citing articles on Scopus
View full text