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Conservative and surgical treatment of pediatric asymptomatic lumbosacral lipoma: a meta-analysis

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Abstract

The aim of this study is to compare the outcomes of surgical and conservative treatments of pediatric asymptomatic lumbosacral lipomas, and to address whether the patients can benefit from prophylactic surgeries. The literature reports of surgical and conservative treatments of child asymptomatic lumbosacral lipomas were reviewed and collected, and a meta-analysis of the reports regarding the incidence of sphincter and lower limb dysfunctions was performed. A total of five literatures were collected, containing a total of 403 patients, among which 124 patients received conservative treatments with 32 (25.81%) cases developing neurological dysfunctions during follow-up, and 279 received prophylactic surgical treatments with 30 (10.75%) patients developing neurological dysfunctions in follow-up, the difference being statistically significant (P ≤ 0.05). For pediatric asymptomatic lumbosacral lipomas of the three major subtypes, the limited source of literature so far suggests that prophylactic surgery is superior to conservative strategy in preventing the patients from neurological deterioration. Larger patient cohorts, randomized studies, and longer length of follow-ups are needed for further corroboration.

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Authors and Affiliations

  1. Department of Neurosurgery, Tongji Hospital of Tongji Medical College, Huazhong University of Science and Technology, No.1095 Jie Fang Avenue, Hankou, Wuhan, 430030, People’s Republic of China

    Yang Xiong, Liu Yang, Wang Zhen, Dong Fangyong, Wan Feng & Lei Ting

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  1. Yang Xiong
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  2. Liu Yang
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  3. Wang Zhen
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  4. Dong Fangyong
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  5. Wan Feng
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  6. Lei Ting
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Correspondence to Wan Feng.

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Comments

D. Douglas Cochrane, Vancouver, BC, Canada

In this article Xiong et al. have tried to clarify a long-standing controversy surrounding the management of complex lumbosacral lipomas. They reviewed the published literature that reported patients treated operatively or followed without surgical intervention. Based on their analysis, the authors conclude that prophylactic spinal cord untethering for lumbosacral lipoma is of benefit for infants and children.

To answer the question of the effectiveness of operative and non-operative management for patients with lumbosacral lipoma with rigor, the following issues need to be considered and reported in future studies.

Specificity of clinical descriptions—the neuroradiological and surgical anatomy

Lumbosacral lipoma is not a single clinical or pathological entity. Chapman’s description was the first attempt to define the surgical anatomy of these lesions [1]. The eloquence of the original descriptions has resulted in their wide adoption and their use for stratifying clinical populations and surgical results. With improved neuroimaging, this group of malformations have proven to be more heterogeneous than previously thought resulting in additional descriptive terms such as lipomyelomeningocle, lipomyelocystocele, lipomyelocele being added to the clinical, radiological and surgical lexicon [2–4]. Additionally, dysplastic elements of bone, muscle and cutaneous structures can be seen in individual patients [5]. Further complicating descriptions of these malformations is the fact that within each descriptor, individual patients vary greatly in the definability and complexity of the lipoma neural interface [6].

Do these terms add to our understanding of the condition?

From a clinical and surgical anatomical perspective, they do. They are descriptive of the underlying disordered anatomy but they also describe features that are likely to be important for the clinical assessment of these infants and children pre and following intervention.

Given that surgical anatomy varies amongst patients with lumbosacral lipoma and with it the effectiveness of surgical untethering, can we continue to use the simple but elegant categories described by Chapman as a primary stratification factor in describing treatment or observational results?

Standardized and auditable clinical and radiographic descriptions of these various malformations must be developed and agreed upon by investigators. It is only with such clarity that “like can be compared with like” with respect to treatment outcomes.

What are the features of symptomatic and asymptomatic patients?

Not all infants born with a lumbosacral lipoma will be neurologically or urologically “normal” at birth [7]. It appears that the more complex the anatomical malformation of the conus, the more likely it is that the infant will have a clinical deficit that is evident at or shortly after birth. So called “congenital deficits” challenge the traditional concept that infants are normal at birth and that all evolving neuro or urological deficits are due solely to cord tethering, and that the deficit can be prevented by “cord untethering”. The evolution of a “deficit” with time and the failure to prevent or reverse it with a technically adequate untethering suggests additional underlying pathological causes.

Congenital deficits follow an evolutionary course affected by growth and development in the infant and child. The failure of a foot or limb to grow normally might suggest symptomatic cord tethering but it may be due to the pre-existing malformative deficit. Other than a detailed and often repeated examination in infant by experts, such deficits may be missed and when subsequently detected ascribed to tethering when that is not the case. Outcome evaluations must control for these pretreatment clinical features lest they bias the outcome assessment for whatever treatments are being studied [8, 9].

What constitutes deterioration?

Prior to intervention

Assuming that a clear and reproducible clinical baseline can be established by a multidisciplinary team, deterioration can be detected. What remains unclear in the literature is the threshold that authors have applied to determine deterioration. This threshold is not standardized and varies amongst observers and care settings. Clinical examination by experts in the motor assessment of the infant and toddler and judicious use of urological testing can detect those who are normal and who deteriorate and those who have a pre-existing “congenital deficit” who are deteriorating not because growth, and development but because of new “pathology”. Deterioration due to a new pathological process is most commonly seen with physiological fat accumulation occurring during the second 6 months of the first year in those infants who have large intraspinal lipomas contained within intact laminae and those who develop a progressing enlarging syrinx rostral to the lipoma neural interface [4].

In the older toddler or child, deterioration is seen in neuromotor function, commonly in strength or foot posture. The “lipoma foot” is characteristic and is due to either symptomatic tethering or neural compression by the intraspinal fat. Older children tend to show urological dysfunction (urodynamics prior to clinical symptomatology). It is yet to be confirmed whether the anatomy of the malformation, specifically the neural-lipoma interface and the presence of dysplastic roots predict deterioration due to malformation or tethering [8].

To clarify future literature, congenital deficits must be recognized and controlled for in outcome analysis and the threshold for “deterioration” must be standardized.

Following intervention or during observations follow-up

The follow-up of these children after surgical untethering is as important as the assessments in infancy regardless of whether surgery was performed of not. Delayed deterioration due to placode scarring with retethering or arachnoiditis will manifest with pain, motor and or urological deterioration. As with the preoperative determination, the threshold that defines deterioration is highly variable introduces a surgeon/site outcome bias. Without consistency in this determination, time to deterioration analysis will be unreliable.

What constitutes an effective operation?

The underlying pathogenesis of expected or observed clinical deterioration must be addressed by the surgical technique used. In most papers, the goals and the technique used are not described [10, 11]. Traditionally, debulking of the lipoma was the goal of operations where the mass effect of the lipoma resulted in neural compression. To untether fully requires maximal lipoma resection with reconstruction of the subarachnoid space. Excepting Pang’s work [12–14], standardized methods of assessing the technical outcome of the operation are not generally described in published clinical series.

Surgical morbidity is likely to vary greatly amongst surgeons based on their experience and technique. Fortunately, decompression and often aggressive untethering can be done with minimal morbidity by many surgeons. The risks of neurological injury are decreased by neural monitoring and mapping (free running EMG, MEP, lipoma and placode stimulation. As with the definition of deterioration, standardized descriptions of lipoma debulking/resection and untethering are needed [15, 16].

The duration of follow-up

Deterioration after surgical intervention does occur but its timing is variable. To date, the studies that have tried to address the “natural history” suggest that a lengthy follow-up, through childhood and adolescence are likely necessary to define the effectiveness of surgery or the safety of observation [17, 18]. Shorter follow-up introduces a confounder and likely a bias for either treatment (surgical or observation). The magnitude of this biases is not known.

Patients undergoing neurosurgical untethering rarely require no other interventions to address the consequences of the congenital or acquired deficits. Urological and orthopedic interventions are frequent. One wonders whether the outcome that should be measured for conus lipoma surgery should be a composite outcome that includes surgical morbidity, time to “deterioration” (urological and/or neurological) and the need for other operations to address either operative injury or progression on non “congenital” deficits. Certainly from the patient’s perspective, this is what they will see and would want to know.

Defining the effectiveness of treatment approaches for lumbosacral lipoma remains a challenge because of definitional, functional assessment, surgical technique and outcome issues described. Unfortunately, the published literature does not consistently address important confounders and limits its outcome determination to acute surgical morbidity and short term placode retethering.

The conclusions reached by Xiong and colleagues reflect their interpretation of the literature with its limitations however it is not clear that their conclusions are supported by the selected data. The authors appeal for larger patient cohorts and randomized studies for further corroboration. To undertake a randomized trial for this condition, the definitional and assessment issues mentioned will need to be addressed. Achieving this clarity will be an important step forward in trying to define the safety and effectiveness of observational or surgical treatment. Equipoise is required for the ethical participation in a randomized trial. Whether it exists [11, 18–21] amongst surgeons should be the subject for a future study.

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[12]Pang D, Zovickian J, Oviedo A (2009) Long-term outcome of total and near-total resection of spinal cord lipomas and radical reconstruction of the neural placode: part I-surgical technique. Neurosurgery 65: 511–528; discussion 528–519

[13]Pang D, Zovickian J, Oviedo A (2010) Long-term outcome of total and near-total resection of spinal cord lipomas and radical reconstruction of the neural placode, part II: outcome analysis and preoperative profiling. Neurosurgery 66: 253–272; discussion 272–253

[14]Pang D, Zovickian J, Wong ST, Hou YJ, Moes GS (2013) Surgical treatment of complex spinal cord lipomas. Childs Nerv Syst 29: 1485–1513

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[16]Valentini LG, Visintini S, Mendola C, Casali C, Bono R, Scaioli W, Solero CL (2005) The role of intraoperative electromyographic monitoring in lumbosacral lipomas. Neurosurgery 56: 315–323; discussion 315–323

[17]Dorward NL, Scatliff JH, Hayward RD (2002) Congenital lumbosacral lipomas: pitfalls in analyzing the results of prophylactic surgery. Childs Nerv Syst 18: 326–332

[18]Wykes V, Desai D, Thompson DN (2012) Asymptomatic lumbosacral lipomas--a natural history study. Childs Nerv Syst 28: 1731–1739

[19]Pang D (2012) Commentary to the article: asymptomatic lumbosacral lipomas--a natural history study, by Wykes V, Desai D, and Thompson DNP. Childs Nerv Syst 28: 1741–1742

[20]Chumas PD (2000) The role of surgery in asymptomatic lumbosacral spinal lipomas. British journal of neurosurgery 14: 301–304

[21]Kulkarni AV, Pierre-Kahn A, Zerah M (2004) Conservative management of asymptomatic spinal lipomas of the conus. Neurosurgery 54: 868–873; discussion 873–865

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Xiong, Y., Yang, L., Zhen, W. et al. Conservative and surgical treatment of pediatric asymptomatic lumbosacral lipoma: a meta-analysis. Neurosurg Rev 41, 737–743 (2018). https://doi.org/10.1007/s10143-016-0796-6

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