Successful Non-Operative Treatment of Congenital ‘Pin-Pong’ Fracture in a Newborn Delivered by Caesarean Section

NJ Ismail; Aliyu Muhammad Koko; B.B Shehu; Lasseini A

doi:10.31487/j.AJSCR.2020.01.02

Successful Non-Operative Treatment of Congenital

ARTICLE

Successful Non-Operative Treatment of Congenital ‘Pin-Pong’ Fracture in a Newborn Delivered by Caesarean Section

A B S T R A C T

Anterior encephalocele is unusual neurosurgical condition seen worldwide. We managed two cases of anterior encephalocele: a five-day old neonate that presented with fronto-ethmoidal swelling noticed since birth. Clinical and radiological evaluations (computerised tomography scan) were in keeping with fronto-ethmoidal encephalocele. The patient had excision and duroplasty via an extracranial-lesional approach. There was excellent post-operative recovery and good cosmetic appearance. The second case was a 6-month-old infant who was diagnosed with ethmoidal encephalocele and congenital hydrocephalus, had ventriculoperitoneal shunt, excision and repair of encephalocele and cranioplasty using autogenous bone graft had remarkable post-operative recovery and was discharged home.

KEYWORDS

Fronto-ethmoidal encephalocele, surgical outcome, neonate

INTRODUCTION

Fronto-ethmoidal (anterior) encephalocele is a congenital malformation characterised by protrusion of intracranial contents through a defect in the skull at the junction of the frontal and ethmoidal bones [1, 2]. The incidence of encephalocele has been reported as 1 in 5,000 to 10,000 live births [3-5]. The incidence of anterior encephalocele compared to occipital encephalocele is higher in tropical regions like Africa and Asia [1]. While the ratio of occipital (posterior) to sincipital (anterior) in Europe and America varies from 2.5:1 to 15:1 [6].

The exact cause of anterior encephalocele is unknown but risks factors include; genetic aberration and environmental factors like aflatoxin, ochratoxin, found in stored farm products have been implicated [3, 4, 7]. Various theories concerning pathogenesis of fronto-ethmoidal encephalocele have been proposed, but the most accepted theorized by Sternberg: states that, the development of fronto-ethmoidal encephalocele could be related to a disturbance in the closure of the rostral neuropore during end of 3rd week of intrauterine life. This disturbance leads to sustained adhesions between neuroectoderm and surface ectoderm at the site of final closure between the nasal fields [8]. Fronto-ethmoidal encephalocele has been classified by Suwanwela and Suwanwela into naso-frontal, naso-ethmoidal and naso-orbital [9]. Neuro-imaging like computerised tomography scan of the brain aids to demonstrates the site, the size of the defect, the characteristics of the tissues within the lesion and the presence of associated anomalies of the brain [10].

The goals of surgery are: Excision of non-functional herniated brain tissue and water-tight dural closure, early prevention of rupture of thin sac and associated complications like infections, bleeding and drainage of cerebrospinal fluid and cranio-facial reconstruction to achieve acceptable cosmesis. Timing of surgery has been a subject of debate, some recommend delay surgery till child is 6-10 months old or when the baby’s weight is 6-8kg [11, 12]. While others suggest early surgery to minimise the pressure effect of the lesion on the facial growth [11].

In our setting, we opted for early surgical intervention in the first case neonatal period to reduce the risks of spontaneous rupture and meningitis, eliminates mother’s/ family distress of having an abnormal child; which may affect the parental care, love and overall survival of the child. The objective of this report was to highlight the management outcome of two cases of rarely reported anterior encephalocele seen in our centre.

Case Presentation

I Case 1

A 5 days old female neonate presented with midline fronto-ethmoidal swelling noticed since birth (Figure 1). There was no associated swelling in any other part of the body, no difficulty in breathing or refusal to breast feeds. There was history of poorly treated maternal febrile illness, mother has not had antenatal care and lives in a rural area about 350KM away from a neurosurgical centre. The child was delivered at home unsupervised and was reported to cry at birth, no history of delivery of a child with any congenital anomaly in the family. Both parents appeared worried about the health of their child.

On examination, we found an active neonate, with a fronto-ethmoidal mass with visible veins, non-tender, soft and cystic, measuring about 20 x10 x 8cm, compressing over the nasal bridge and flattening the nose. Occipito-frontal circumference was 32cm (normal for age) (Figure 1).

Cranial computerised tomography scan showed a huge mixed density mass with an ethmoidal skull defect measuring about 11.7mm and widest diameter of the lesion about 69mm (Figure 2). Full blood counts and serum electrolytes urea and creatinine were normal. Patients had total excision of the mass via a fronto-facial incision around the lesion, water-tight duroplasty and refreshing of the bone edges around the defects to stimulates osteogenesis (Figures 3 & 4). A firm plastic needle cover of 2ml syringe was used as stent in the nostrils to prevent nasal obstruction from post-operative oedema (Figure 4). Antibiotics were given for five days. Patient had good post-operative recovery and acceptable cosmesis to both the Neurosurgeons and the parents (Figure 5). In addition, parents were very happy and discharged home to see clinic for follow up.

Figure 1: Photograph at initial presentation.

Figure 2: Axial CT scan of showing depression.

Figure 3: 3D CT scan image showing depression.

Figure 4: Clinical photograph at 4 months.

Discussion

Congenital pin-pong fracture is an uncommon neurosurgical condition seen mostly in African women as in the index case [13, 15]. It commonly involves the parietal area on the right side similar to the present report [4, 11]. In contrast to some Authors, congenital pin-pong fracture usually affects the frontal area in the Africans and parietal region in Western countries [15]. Several aetiological factors related to both the mother and the baby, but none was identified in our patient. Congenital pin-pong fracture usually presents a cosmetic concern as in the case of our patient who had no symptoms other than aesthetic deformity [3]. However, some patients may have abnormality such as scalp haematoma, intracranial haematoma, brain edema or sinus tears [1]. Several Authors reported different treatment options for pin-pong fracture. The choice of treatment should be guided by the severity of the fracture and the presence of underlying intracranial injury [3, 14]. Those in favour of surgical treatment express concerns that the pressure caused by depression on the brain may lead to brain damage, decrease cerebral blood flow and epileptogenic foci therefore, the need for surgical elevation to minimise these effects [6].

However, many studies have demonstrated that in the absence of neurological deficit or intracranial injury, there is no difference between operative and non-operative treatment measures, in terms of development of future neurological deficit or epilepsy [3, 6]. Because of theses aforementioned facts, we opted for none-operative watchful waiting in our patient who had successful spontaneous elevation at four months of birth. In tandem with our case, studies in literature have demonstrated spontaneous elevation within 4 to 6 months of life [11]. Although, other none-operative modalities like obstetrics vacuum extraction, use of breast pump and Raynor-parsa manoeuvre by applying digital pressure on the margins of the depression have described in literature, we chose none-operative watchful waiting because it’s the least invasive. In neonates, the presence of membranous sutures, patent fontanelles and the low calcium in the skull confer it a plasticity. Because of the plasticity, relative thin skull, frequent crying in new-borns with resultant increase in intracranial pressure had been thought to result in spontaneous elevation of depressed skull [3]. Pin-pong fracture deeper than 5mm were considered significant requiring surgical elevation [1]. In line with this view, the depression was 3.8mm in our patient and as such none-operative watchful waiting was instituted.

Conclusion

Congenital pin-pong fracture is a rare neonatal skull abnormality that can be seen with no identifiable aetiology. Watchful waiting without the need for more invasive surgical elevation may suffice in the absence of neurological deficts attributable to the depression and intracranial injury.

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Article Info

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Case Report

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Copyright

© 2023 Aliyu Muhammad Koko. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Hosting by Science Repository.

DOI: 10.31487/j.AJSCR.2020.01.02

AUTHOR INFO

Author Info

NJ Ismail

Aliyu Muhammad Koko

B.B Shehu

Lasseini A

Corresponding Author

Aliyu Muhammad Koko
Department of neurosurgery, regional centre for neurosurgery Usmanu Danfodiyo University Teaching Hospital Sokoto, Nigeria

FIGURES & TABLES

Figure 1: Photograph at initial presentation.

Figure 2: Axial CT scan of showing depression.

Figure 3: 3D CT scan image showing depression.

Figure 4: Clinical photograph at 4 months.

REFERENCE