A report on three cases of chronic adult hydrocephalus occurring several months postlumbar spine surgery complicated by cerebrospinal fluid leakage

Abstract

Background:

Chronic adult hydrocephalus (CAH) is believed to have an incidence rate between 0.2 and 5.5 cases/100,000 individuals. Although the underlying mechanisms remain largely unknown, several potential contributing factors have been identified, such as meningitis, cranial injuries, intradural tumors, malformations of the posterior fossa, neurological conditions, and complications arising from intracranial surgeries. These factors may lead to a delayed onset of symptoms associated with hydrocephalus, which include cognitive decline, mobility difficulties, and urinary incontinence.

Case Description:

We report three cases of patients who developed CAH within a timeframe of 10–14 months following spinal surgery, which was complicated by cerebrospinal fluid (CSF) leakage. The close temporal relationship and the lack of other identifiable causes in their medical histories may suggest a possible causal link. While definitive evidence directly connecting complications from lumbar surgery to the onset of CAH is lacking, the observed temporal association and absence of alternative explanations in the patients’ medical histories indicate a potential correlation.

Conclusion:

This observation highlights the necessity for further research into the connection between spine surgical procedures impacting the meninges and the subsequent emergence of normal pressure hydrocephalus, as well as the possible influence of CSF dynamics and neuroinflammatory processes in the development of CAH.

INTRODUCTION

Chronic adult hydrocephalus (CAH) is estimated to occur at an incidence rate ranging from 0.2 to 5.5/100,000 individuals.[1,3,12,14,15] The prevalence of this condition tends to increase with advancing age.[3,9,12,22,23] Numerous hypotheses regarding the pathophysiology of CAH have been proposed, including dynamic, vascular, neuroinflammatory, and genetic abnormalities, though none have been definitively established. Typically, the condition manifests after the age of 60 years, with the classic clinical presentation known as the Hakim triad.[1-3,9,12,14,22,23] Various potential causes have been suggested, including meningitis, cranial trauma, intradural tumors, malformations of the posterior fossa, neurological disorders, and sequelae from intracranial surgeries, particularly those involving narrowing of the aqueduct of Sylvius.[4-8,18,30] This report presents three cases of patients who developed chronic hydrocephalus within 10–14 months following spinal surgery, complicated by cerebrospinal fluid (CSF) leakage. While we lack concrete evidence to establish a direct correlation between the onset of CAH and the meningeal breach complications from their lumbar surgeries, the temporal proximity and the absence of other identifiable causes in their medical histories may imply a potential causal relationship.

CASE 1

A 59-year-old man, slightly overweight and without any significant history of trauma or prior surgeries, worked as a delivery driver and presented with a herniated left posterolateral L4-L5 disc. He underwent endoscopic surgery, during which a tear in the meninges occurred. This ouverture was subsequently sealed using a dural sealant, and the patient was discharged the day after the procedure. One week postsurgery, he reported a clear, non-purulent fluid oozing from the surgical site, but he did not experience any associated symptoms such as headaches or buzzing. Both lumbar and cerebral magnetic resonance imaging (MRI) scans were conducted; the cerebral MRI appeared normal, with ventricles within acceptable limits, while the lumbar MRI revealed an open meningeal breach. On the 7^th day following the initial surgery, the patient was re-admitted, and the breach was sutured successfully without complications. He was discharged the next day. Follow-up appointments at 1 and 2 months postsurgery, along with a new MRI scan, indicated complete healing with no abnormalities and confirmed that the breach was watertight. He resumed his professional duties 2½ months following the surgical procedure, experiencing no pain during this period. Eleven months later, he consulted his physician, who subsequently referred him to a neurologist due to emerging difficulties with walking and worsening headaches. An MRI scan indicated triventricular dilatation, which was identified as the cause of adult-onset hydrocephalus [Figures 1a-d]. An inquiry into his medical history did not uncover any additional incidents in the 8 months postsurgery. He underwent a lumbar puncture along with an assessment for hydrocephalus. The lumbar puncture significantly alleviated his symptoms, including urinary issues, headaches, and mobility challenges.

Figure 1:

(a) A sagittal section of the magnetic resonance imaging scan in T1 (b-d) alongside axial sections in T2, Illustrating ventricular enlargement and transependymal absorption. (c) Green line: frontal horn width, Red line: internal skull width at the same level (biparietal diameter)

CASE 2

A 64-year-old male, an employee and a smoker, presented with no prior medical or surgical history. Following his retirement, he sought consultation due to severe paresthesia in the lower limbs and lumbar pain. Diagnostic evaluation indicated a narrowed lumbar canal at the L3-L4 level. He subsequently underwent a lumbar recalibration procedure, during which a CSF leakage occurred but was successfully sutured without further complications. The patient was discharged the next day. Throughout the recovery period, he reported no issues, and the surgical site healed without incident. His mobility improved, lower back pain diminished, and paresthesia resolved. However, 10 months later, he experienced difficulties with ambulation, urinary urgency, and cognitive issues. An MRI scan identified triventricular hydrocephalus [Figures 2a-c]. A lumbar puncture significantly alleviated his symptoms.

Figure 2:

(a and b) Sagittal sections of the magnetic resonance imaging scan in T1 and (c) in Flair image, illustrating ventricular enlargement and transependymal absorption.

CASE 3

A 66-year-old female smoker experienced a cauda equina syndrome (saddle anesthesia) and debilitating S1 sciatica following the lifting of a heavy load, resulting in an inability to walk on her toes and right foot. An MRI revealed a large disc herniation that completely filled the spinal canal. She underwent surgery promptly; however, during the procedure, a CSF leakage occurred, which was sealed using a durapatch and a piece of subcutaneous fat. She quickly regained saddle anesthesia and showed significant recovery of the gastrocnemius muscle. Fourteen months later, she developed urinary incontinence, and her husband reported episodes of falls and instability. A subsequent lumbar MRI did not reveal any specific abnormalities; however, a brain MRI indicated triventricular hydrocephalus [Figures 3a-c]. It is noteworthy that a comparison was made with imaging conducted a year and a half before the surgery, during which she underwent spinal and brain MRIs for dizziness, and the ventricles were not dilated.

Figure 3:

An axial section of computed tomography scan shows (a) dilated ventricular system, with transependymal oedema and axial sections in diffusion magnetic resonance imaging (b) indicates the absence of ventricular enlargement approximately 36 months prior to the depiction in (a). (c) Measurement of the callosal angle (green arrow with red lines) shows 60°, an increase compared to normal (less than 40°) on the coronal view.

DISCUSSION

The estimated prevalence of CAH ranges from 2 to 5.5/100,000 individuals.[1,3,12,15] Similar to other neurodegenerative disorders, including Alzheimer’s and Parkinson’s diseases, this prevalence tends to increase with advancing age.[9,24,28,30] In Europe, the estimated number of cases is approximately 17,000.[5,12] A study conducted in Norway indicated that the prevalence escalated from 3.3 cases/100,000 individuals in those under 60 years of age to 93.3 cases/100,000 in individuals over 80.[18] In addition, a Swedish study reported a prevalence rate of 0.2% among individuals aged 70–79, in contrast to 5.9% in those aged 80 and above.[30] Various hypotheses have been proposed regarding the pathophysiology of CAH, including abnormalities in CSF dynamics, which may arise from cardiac systolic dysfunction leading to impaired resorption of the fluid and subsequent ventricular dilation.[24] Other potential contributing factors include vascular issues, as patients with CAH often have histories of hypertension, diabetes, obesity, and physical inactivity. Research by Huovinen et al. revealed that at least one case of CAH was associated with a genetic predisposition in 16% of the cases studied, suggesting a possible genetic component, although no specific gene has yet been identified in relation to CAH.[2,9,29] Some researchers have suggested a role for neuroinflammation, as elevated inflammatory markers have been detected in the CSF of CAH patients, with levels decreasing postsurgery.[16,18-21] The advent of advanced imaging technologies has revealed a hypometabolic area, particularly in the basal ganglia, in positron emission tomography scans of individuals with CAH, which appears to be reversible following treatment.[22,23] Despite these various theories, the definitive causal relationship remains unclear. Regarding the known etiology of CAH, factors such as meningeal hemorrhage, meningitis, surgical sequelae, cranial trauma, intracranial tumors, aqueduct of Sylvius stenosis, and intrarachnoid tumors are noted whenever these conditions are present in patients’ medical histories.[1-4,6,8,9-15,17-20,24-30] Currently, there is no established direct correlation between a lumbar osteomeningeal breach and the development of adult hydrocephalus. While we do not assert that our cases demonstrate a causal relationship, it is noteworthy that the three patients who underwent lumbar spine surgery exhibited the onset of CAH, within a relatively brief timeframe of 10–14 months. This observation prompts consideration of a potential direct association between these two occurrences. One hypothesis, we propose is the presence of blood in the CSF at the time of, and subsequent to, the formation of the meningeal breach. It raises the question of whether this minimal or potentially negligible amount of blood and its associated proteins could have a significant impact. Discussing this matter and providing substantiated arguments is quite challenging. Two meta-analyses have been conducted, one addressing the risk of CSF leakage following intradural spine surgery and the other focusing on extradural spine surgery, reporting incidences of CSF leakage ranging from 2% to 34% and 1% to 15%, respectively. However, neither analysis addresses the occurrence of postoperative hydrocephalus. It is important to note that the follow-up periods in these studies may not be sufficiently extensive. A more prolonged follow-up and extended timeframe are essential to accurately determine any potential link between CAH and the occurrence of a meningeal breach during lumbar spine surgery.

CONCLUSION

CAH encompasses a range of potential etiologies, with no definitive cause currently identified. Based on our observations, we propose a possible association between CAH and CSF leakage occurring after lumbar spine surgery. This report highlights the importance of further research to explore the connection between spinal surgical procedures involving the meninges and the subsequent onset of CAH. In addition, investigations into the role of altered CSF dynamics and neuroinflammatory processes in the development of CAH are essential to enhance our understanding of this complex condition.

Ethical approval:

Institutional review board approval is not required.

Declaration of patient consent:

The authors certify that they have obtained all appropriate patient consent.

Financial support and sponsorship:

Nil.

Conflicts of interest:

There are no conflicts of interest.

Use of artificial intelligence (AI)-assisted technology for manuscript preparation:

The authors confirm that there was no use of artificial intelligence (AI)-assisted technology for assisting in the writing or editing of the manuscript and no images were manipulated using AI.

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