Tethered Cord Prevalence among Patients with Prenatal or Postnatal Myelomeningocele Repair

doi:10.21203/rs.3.rs-4294292/v1

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Tethered Cord Prevalence among Patients with Prenatal or Postnatal Myelomeningocele Repair

https://doi.org/10.21203/rs.3.rs-4294292/v1

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published 02 Oct, 2024

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Background:

Myelomeningocele (MMC) is a severe form of spina bifida characterized by spinal cord extrusion into a cerebrospinal fluid (CSF) filled sac which may lead to lifelong disability. Repair of these lesions have classically occurred shortly after birth, but more recently, prenatal myelomeningocele repair techniques have been elucidated. This study aimed to investigate the outcome of surgery, particularly with subsequent spinal cord tethering, in patients with prenatal myelomeningocele closure and those with postnatal repair surgery.

Materials and methods:

In this retrospective study which assessed patients from April 2002 to April 2020, the data of 18 total patients with MMC were reviewed. Nine patients from each group were included, closed prenatally or postnatally, respectively. Demographic information including age and sex, birth week, infant comorbidities, presence of dermoid or lipoma, cutaneous stigmata, total number and timing of detethering procedures, presence of Chiari malformation, and need for VPS was obtained.

Results:

Eighteen patients including 9 infants closed prenatally and 9 infants with myelomeningoceles closed postnatally were reviewed. The mean age was 4 ± 3 years and 6.22 ± 2.4 in prenatal and postnatal retrospectively. There was a significant relationship between the number of detethering procedures (p.value = 0.03) and the need for a ventriculoperitoneal shunt (VPS) (p.value = 0.01) between the groups, with the prenatal closure group having lower rates of each. There was no significant difference between the groups in regard to the mean age at the detethering procedure (p = 0.4), sex (p = 0.09), birth week (p = 0.8), comorbidities (p = 0.8), presence of intraspinal dermoid or lipoma (p = 0.09), presence of cutaneous stigma (p = 0.08), Chiari (p = 0.6), Fatty filum (p = 0.08), syrinx (p = 0.4), bone anomaly (p = 0.4), and spina bifida neurological scale (p = 0.66).

Conclusion:

There was a significant relationship between the two groups in terms of the number of detethering procedures, and the need for VPS. Our data represents a possible difference in the need for detethering surgeries and ventriculoperitoneal shunt placements between patients with prenatal and postnatal myelomeningocele closures.

Myelomeningocele

Tethered spinal cord

spina bifida

detethering

Spina Bifida is one of the most common central nervous system (CNS) congenital anomalies in children. Myelomeningocele (MMC) is a severe form of spina bifida characterized by spinal cord extrusion into a cerebrospinal fluid (CSF) filled sac which may lead to lifelong disability (1). It is complicated by hydrocephalus, motor and cognitive disorders, bowel and bladder dysfunction, lower extremity weakness and numbness, the need for ventriculoperitoneal shunt, and subsequent spinal cord tethering which may require release surgery. The prevalence of MMC is estimated to be 0.8 to 1/1000 and 0.2 to 0.4 live births in the worldwide, and US respectively. and the death rate is about 10% (1–3). Most cases of MMC are diagnosed in utero and while postnatal closure has been the standard of care classically, in recent years, the potential for prenatal closure of these lesions has been extensively studied (4).

With postnatal surgical repair, the extrusion of the spinal cord at birth may lead to irreversible central and peripheral nerve damage, as well as complications during birth and surgical repair, including CSF leak, meningitis, and further neurological injury (5). Prenatal spina-bifida (PNS) closure can potentially protect from these complications and preserve neurological function (6–8). As the ever-expanding literature on prenatal closure further elucidates the immediate risks and benefits compared to postnatal closure, few reports have looked at the differences in long-term outcomes between these groups of patients, particularly in the realm of post-closure spinal cord tethering (1, 6–8).

In 1997, the first case of human prenatal MMC repair by hysterotomy was done. Prenatal closure of myelomeningoceles (MMC) is a relatively new treatment that is effective in improving hindbrain herniation and reducing the need for postnatal shunting for hydrocephalus. It also has positive effects on motor function and neurodevelopmental outcomes. However, despite these benefits, there is an increased need for secondary spinal cord tethering surgery before the age of 12 months and a higher incidence of dermoid cysts in infants who undergo prenatal surgery (9–12).

Although data from prenatal repair studies have demonstrated a dramatic improvement in hindbrain herniation, additional risks of preterm labor, uterine dehiscence, preterm birth, and preterm and fetal death have been reported (13–14). Despite recent advances in prevention, diagnosis, and intervention, neural tube defects remain a major concern of disability, morbidity, and mortality in the US and other parts of the world. Hence, this study aimed to investigate the outcome of surgery, particularly spinal cord tethering, in prenatal fetal closure in comparison with postnatal repair surgery in patients with myelomeningocele.

In this retrospective chart review study, 18 babies operated on for myelomeningocele closure in the Department of Neurosurgery of John Hopkins University of Medical Sciences from April 2002 to April 2020. The study was outlined based on the STROBE guideline for epidemiological observational studies. The context of the study was approved by the ethics committee of Johns Hopkins University of Medical Sciences. Nine patients from each group were either closed prenatally or postnatally. Rates of tethering at the last follow-up were collected in addition to demographic and functional information to determine variable rates of long-term tethering between the two groups.

Inclusion and exclusion criteria

The study included pregnant women who had given informed consent and whose fetuses were diagnosed with MMC. They were referred to the Johns Hopkins University of Medical Sciences for MMC repair surgery between 2002 and 2020. The participants were then followed up prospectively until June 2023. The inclusion criteria were maternal in any gestational week, age between 18 to 35, normal karyotype, S1 and a higher level of lesion, and acceptance of the long-term follow-up. The exclusion criteria were any chronic disease such as Insulin-dependent pregestational diabetes, multiple gestational pregnancies, any previous abortion, any other fetal anomalies, placenta previa, Obesity and BMI ≥ 35, incompetent cervix and/or short cervix < 20 mm, Fetal kyphosis ≥ to 30 degrees, maternal-fetal Rh immunization, positive maternal human immunodeficiency virus or hepatitis-B or known hepatitis-C, any Uterine anomaly, inability to comply with follow-up.

Study methods

Demographic information including age, sex, birth week, infant comorbidities, presence of dermoid or lipoma, Cutaneous stigmata, total tethering procedures, age upon tethering procedure, Chiari malformation presence, and need for VPS, was obtained. Information regarding the type of myelomeningocele closure, the constellation of preoperative symptoms, postoperative symptoms, complications after surgery, spinal level of myelomeningocele, conus medullaris level, and presence of fatty filum, syrinx, or bony anomaly was obtained directly from the patient’s medical records.

Study outcome

The primary outcome was evaluating tethering procedure number as dependent upon prenatal vs. postnatal MMC repair. Secondary outcomes included symptom improvement after detethering procedures, complication rate, and requirement for multiple detethering procedures between groups. We also investigated long-term follow-up including complications, and motor and sensory function. All patients followed-up until June 2023.

Statistical analysis

In this study, the Chi-Square test was used for qualitative, and variables the t-student test was used to compare quantitative variables independently. The analysis of data was performed descriptive statistics tests, and the results were presented as mean ± standard deviation (SD) and frequency in percentage in some variables. In this study, IBM SPSS Statistics for Windows, version 28 (IBM Corp., Armonk, NY, USA) was used to analyze all data, and a p-value ≤ 0.05 was considered significant.

In the present study, 18 patients were reviewed for this study, including 9 infants closed prenatally and 9 infants with myelomeningocele closed postnatally. The mean age in the prenatal closure group was 4 ± 3 years (min = 1, max = 8) and in the postnatal group was 6.22 ± 2.4 years (min = 1, max = 9) (Table 1). There was a significant relationship between two groups based on age (p.value = 0.01).

Table 1

The table shows demographic information of the patients enrolled in the study.
Variables	Case		Control		P.value
Age	4 ± 3(min = 1, max = 8)		6.22 ± 2.4(min = 1, max = 9)		0.01
Sex	Male	3	Male	2	0.09
Sex	Female	6	Female	7	0.09
Birth week	36 ± 3.77 (Min = 27, Max = 39)		37.66 ± 1(Min = 36, Max = 39)		0.9
Comorbidity	None	2	None	1	0.8
	Neurogenic bladder	3	Neurogenic bladder	6
	Club foot	1	scoliosis	2
	Hip dysplasia	1	scoliosis	2
	Hip dysplasia	1	hip dysplasia	1
	Bilateral foot deformity	2	kyphosis	1
			Bilateral foot deformity	1
			Neurogenic bowel	2
Dermoid or lipoma	None	7	None	9	0.09
	Dermoid	2	Dermoid	0
	Lipoma	0	Lipoma	0
Cutaneous stigmata	None	9	None	7	0.08
			dermal sinus tract	1
			hemangioma	1
Detethering Surgery	Yes	3	Yes	5	0.03
Detethering Surgery	No	6	No	4	0.03
Chiari	Yes	8	Yes	8	0.6
Chiari	No	1	No	1	0.6
VPS	Yes	4	Yes	8	0.01
VPS	No	5	No	1	0.01
Fatty filum	Yes	2	Yes	0	0.08
Fatty filum	No	7	No	9	0.08
Syrinx	Yes	5	Yes	6	0.4
Syrinx	No	4	No	3	0.4
Bony anomaly	Yes	6	Yes	5	0.4
Bony anomaly	No	3	No	4	0.4

Of 9 patients in the prenatal group, 3 (33%) were male and 6 (66%) were female and of 9 patients in the postnatal repair group, 2 (22.2%) patients were female, and 7 (77.8%) patients were male. There was no significant relationship between the two groups based on sex (p.value = 0.09), birth week (p.value = 0.8), comorbidities (p.value = 0.8), presence of dermoid or lipoma (p.value = 0.001), cutaneous stigma (p.value = 0.08), Chiari malformation (p.value = 0.6), fatty filum presence (p.value = 0.08), syrinx presence (p.value = 0.4), and associated bone anomalies (p.value = 0.4) (Table 2, 3). The spina bifida neurological scale was 10.33 ± 2.5 (min = 6, max = 14) in prenatal and 9.44 ± 4.76 (min = 4, max = 14) in postnatal group. There was no significant relationship between the prenatal and postnatal group based on the spina bifida neurological scale (p.value = 0.66).

Table 2

The demographic, intra-operative, post-operative, and complications in the prenatal myelomeningocele closure group.
Case number	Age	sex	Chiari	Gestational age (week)	Syrinx	Fatty filum	Bony anomaly	VPS	Number of tethering procedures	Age at surgery ) Month)		Symptoms and signs	Spina bifida neurological scale	conus medullaris Level	Surgery improvement	Complication
Patient 1	1	f	yes	25	Yes	No	Yes	yes	0	-		Bladder dysfunction	6	S1	Yes	None
Patient 2	1	f	yes	26	No	No	Yes	yes	0	-		None	10	L3/4	Yes	None
Patient 3	2	m	No	26	Yes	No	Yes	No	0	-		None	14	L5/S1	Yes	None
Patient 4	2	m	yes	25	Yes	No	Yes	No	0	-		Bladder dysfunction, back pain	11	L3/4	Yes	None
Patient 5	2	f	yes	23	No	Yes	No	No	0	-		None	11	L3	Yes	None
Patient 6	5	f	yes	22	No	No	No	yes	0	-		None	12	S1	Yes	None
Patient 7	7	f	yes	22	Yes	Yes	Yes	yes	3	First surgery	12	Bladder dysfunction	7	L5	yes	Wound Dehiscence
										Second surgery	24	Motor decline			Late improvement	None
										Third surgery	36	Motor decline			Late improvement	None
Patient 8	8	m	yes	24	No	No	No	No	1	First surgery	36	Bladder dysfunction, motor decline	10	L4	yes	None
Patient 9	8	f	yes	25	Yes	No	Yes	yes	2	First surgery	36	Scoliosis progress	12	L4/5	yes	None
										Second surgery	84	Scoliosis progress			-	None
										Second surgery	84	None			-	None

Table 3

The demographic, intra-operative, post-operative, and complications in the postnatal myelomeningocele closure group.
Case number	Age (year)	Chiari	sex	Fatty filum	Syrinx	Bony anomaly	VPS	Number of tethering procedures	Age at surgery (month)			Symptom and signs	Spina bifida neurological scale	conus medullaris Level	Surgery improvement		Complication
Patient 1	1	yes	f	No	Yes	Yes	yes	1	First surgery		9	None	14	T12/L1	Early improvement		None
Patient 2	4	yes	m	No	Yes	No	yes	1	First surgery		24	None	12	S1	Early improvement		None
Patient 3	6	yes	f	No	Yes	No	yes	1	First surgery		24	None	13	L5	Early improvement		None
Patient 4	6	yes	f	No	Yes	Yes	Yes	1	First surgery		84	Leg pain	4	T2	Early improvement		None
Patient 5	7	yes	f	No	Yes	Yes	Yes	0	-			Bladder dysfunction	4	L3	-		None
Patient 6	7	yes	m	No	No	Yes	Yes	0	-			Bladder and bowel dysfunction	5	L5/S1	-		None
Patient 7	8	yes	f	No	Yes	Yes	Yes	0	-			Bladder dysfunction	14	S2			None
Patient 8	8	yes	f	No	No	No	yes	2	First surgery	24		Bladder dysfunction and spasticity	5	L4	After first surgery	Early improvement	None
Patient 8	8		f				yes	2	Second surgery	72		Bladder dysfunction and spasticity	5	L4	After second surgery	Early improvement	None
Patient 9	9	no	f	No	No	No	no	0	-			Bladder dysfunction	14	S1		-	None

There was a significant relationship between the need for VPS placement in the two groups (p.value = 0.01), and patients in the prenatal group had fewer VPS placements. Of 9 patients in prenatal and postnatal groups, 7 patients in both groups were diagnosed with tethered cord (p.value = 0.7). However, of 9 patients in the prenatal group, 3 (33%) patients underwent detethering procedures more than once vs. 5 (55%) of the postnatal patients. There was a significant relationship between the two groups in terms of the number of detethering procedures (p.value = 0.03). Moreover, the initial age of tethering surgery in the prenatal group was 38 ± 24.49 (min = 12, max = 84) months, and in the postnatal group was 39.5 ± 30.61 (min = 9, max = 84) months. This did not reflect a statistically significant difference (p.value = 0.4); however, the average age was higher in postnatal group patients.

In patients who underwent detethering surgery in both groups, the most reported signs were bladder dysfunction and motor decline. The motor decline was seen more commonly in the prenatal closure group. Spasticity was seen as a preoperative symptom predominantly in the postnatal closure group. There was no significant relationship between the two groups based on comorbidities.

This study indicates that there was a significant relationship between the two groups in terms of the number of detethering surgeries and patients who underwent postnatal surgery had a higher risk of detethering surgery than those who underwent prenatal tethering procedure (p.value = 0.03). However; there was no significant relationship between comorbidity, birth week, dermoid or lipoma presences, Cutaneous stigmata, Chiari, fatty filum, syrinx, and bony anomaly in the prenatal versus the postnatal group.

The primary goal of myelomeningocele closure is to reconstruct the developmental layers that did not form during gestation. This will prevent the layers from reattaching and restore adequate cerebrospinal fluid flow. The treatment for spina bifida involves closing the myelomeningocele, preventing ventriculomegaly and its complications, as well as managing any other neurological, urological, musculoskeletal, or dermatological conditions throughout the patient's life. Fetal surgical repair of MMC is more effective in improving early neurological outcomes compared to postnatal operation (20–21).

The first step in treating neural tube defects is to close them as early as possible. This is because if the spinal cord is exposed to amniotic fluid or air, it can cause damage. In cases where the MMC is repaired after birth, most patients develop hydrocephalus within the first year of life (20–23). Prenatal closure may have an impact on reducing the development of hydrocephalus in the future based on previous studies. In this study, it has been shown that there was a significant relationship between the need for VPS in two groups, and its rate was higher in the postnatal group, suggesting the less frequent development of hydrocephalus with prenatal myelomeningocele closure.

Postnatal surgery may be performed for cosmetic or infection prevention purposes, but it cannot restore lost neurological function. During the last four months of gestation, the fetal surgery technique can improve the neurological function of patients by preventing the disease from further progression. Neonatal mortality rates for MMC are not different between prenatal and postnatal surgery (22–23). However, when prenatal surgery is performed, there is a higher likelihood of placental abruption, separation of the membranes, and preterm rupture of membranes at birth compared to postnatal surgery (22–24). Additionally, prenatal MMC repair reduces hindbrain herniation, and hydrocephalus, and lowers the need for shunt implantation. It improves postnatal walking ability and psychomotor evaluation score, but not mental outcomes compared to postnatal MMC closure (22, 25–26).

TCS in patients with MMC is diagnosed more frequently due to advanced imaging. Deciding when to offer surgical treatment is a careful consideration made by the neurosurgeon, the patient, and their family, especially in asymptomatic patients with incidental findings. Surgical intervention is recommended for those who present neurological or urological symptoms with radiographic correlation (27–29). It is recommended to undergo TCR (tethered cord release) once neurologic or urologic symptoms appear. However, there is a debate about whether surgery is necessary for an asymptomatic patient with an incidental finding. Some argue that prophylactic procedures are necessary because once neurologic or urologic symptoms occur, there is a risk of permanent deficit despite surgical intervention. The literature reports varying urologic and neurologic outcomes post-tethering, depending on the OSD (occult spinal dysraphism) type and preoperative condition. However, earlier intervention generally leads to better outcomes (28–33). While the details of timing for detethering are beyond the scope of this report, we have provided some insight into the timing and need for detethering surgery between prenatally-closed and postnatally-closed patients with myelomeningocele.

The difference between tethering rates of these cohorts may be related to the methodology for closure in the prenatal versus postnatal group, which results in laying a graft over the defect in the former group and having it incorporated into the skin over time to seal the defect. In the latter group, the skin and underlying tissues are sutured closed in the midline of the defect, which may contribute to greater scar tissue formation and inflammation postoperatively. In the prenatal group, malformation may occur at the graft/skin interface laterally off the midline and away from the neural elements. It is possible that this, among other factors, can contribute to the findings of the report. Further work will be required to better understand the propensity to tether in these cohorts in the future.

This study had some limitations which may have impacted the results. First, the main limitation was the limited number of cases investigated. Additionally, the patients were randomly selected for the two groups and were not matched before reviewing, which may have affected the conclusions of the study. Moreover, this is a retrospective study and confounding factors were not controlled. Additionally, there is a subjective component to symptom reporting and decision-making to undergo detethering surgery, and this may have contributed to the differences in primary outcomes between the cohorts. This, however, remains the first study of its kind, to the best of our knowledge, and may provide a foundation for larger works in the future. Hence, further prospective studies with longer follow-up time are recommended to be conducted, to compare the prenatal and postnatal impact on subsequent tethering rates, and they will reveal a greater understanding of the need for detethering procedure after prenatal myelomeningocele closure.

Our research demonstrates that there was a significant relationship between the two groups in terms of the number of tethering surgeries, and the need for VPS placement. There was a lower shunting rate in the prenatally closed group, which supports previous literature, as well as a lower rate of subsequent detethering procedures. There was no significant relationship in terms of the age of the detethering procedure between the two groups, nor was there a difference in comorbidities, birth week, presence of dermoid, lipoma, or cutaneous stigmata, Chiari malformation, fatty filum, syrinx, or bony anomaly in the prenatal versus the postnatal groups. Differences in tethering rates may be related to the differences in surgical closure between the groups and the implications for future management of these patients will require larger studies with greater cohorts with longer-term follow-up.

Funding Declaration: No Funding

Competing Interest declaration: No Competing Interest

Author Contribution

Authors Individual Contributions: Seyed Ahmad Naseri Alavi, corresponding author, Supervision of the research, Idea procurement, Writing, drafting, Analysis of the article, Final approval of the manuscriptRose Fluss, Idea procurement, Final approval of the manuscriptKurt Lehner, Idea procurement, Final approval of the manuscriptBrendan Judy, Idea Procurement, Final approval of the manuscriptMari Groves, Supervision the research, Idea procurement Final approval of the manuscriptAlan Cohen, Writing, drafting, Analysis of the articleAndrew J Kobetes, Writing, drafting, Analysis of the article, Idea Procurement, Final approval of the manuscript

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No competing interests reported.

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Tethered Cord Prevalence among Patients with Prenatal or Postnatal Myelomeningocele Repair

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