Cranial nerve palsy secondary to cerebrospinal fluid diversion

Clinical Neurology and Neurosurgery 143 (2016) 19–26

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Cranial nerve palsy secondary to cerebrospinal fluid diversion Guichen Li a , Xiaobo Zhu b , Yang Zhang b , Jinchuan Zhao b , Zhiguo Han c , Kun Hou b,∗ a

Department of Neurology, The First Hospital of Jilin University, Changchun, Jilin, China Department of Neurosurgery, The First Hospital of Jilin University, Changchun, Jilin, China c Department of Surgery, Changchun Orthopaedics Hospital, Changchun, Jilin, China b

a r t i c l e

i n f o

Article history: Received 20 November 2015 Received in revised form 1 February 2016 Accepted 7 February 2016 Available online 9 February 2016 Keywords: Cerebrospinal fluid diversion Cerebrospinal fluid hypovolemia Cranial nerve palsy External lumbar drainage Lumbar puncture Ventriculoperitoneal shunt

a b s t r a c t Objective: Cranial nerve palsy (CNP) secondary to cerebrospinal fluid (CSF) diversion is less familiar to us as a result of its rarity in incidence and insidiousness in presentation. This study aims to further expound the pathophysiological mechanism, clinical presentation, diagnosis, management and prognosis of CNP. Methods: From June 2012 to February 2015, 5 of 347 consecutive patients with CNPs secondary to different CSF diversion procedures were treated at our institution. A systematic PubMed search of published studies written in English for patients developing CNPs after CSF diversion procedures from January 1950 to June 2015 was conducted. Results: Overall, 29 studies and 5 patients of the current series totaling 53 CNPs met the inclusion criteria. CN II, III, IV, V, VI, VII and VIII were got involved in 2 (3.8%), 2 (3.8%), 5 (9.4%), 1 (1.9%), 44 (83.0%), 4 (7.5%) and 1 (1.9%) patients respectively. Thirty-eight patients (71.7%) developed CNPs following inadvertent lumbar puncture, 8 (15.1%) following lumbar drainage, and 7 (13.2%) following ventriculoperitoneal shunt. Forty-eight (90.6%) patients got resolved completely. Conclusions: The proposed mechanism of CNP after CSF diversion procedure is CSF hypovolemia and subsequent downward displacement of the brain and traction and distortion of the vascular and peripheral neural structures. As a result of its distinct anatomic characteristics rather than long intracranial course, CN VI is most commonly affected. With early recognition and timely conservative management, most patients could get favorable recovery. © 2016 Elsevier B.V. All rights reserved.

1. Introduction

2. Material and methods

Cerebrospinal fluid (CSF) diversions as lumbar puncture (LP), external lumbar drainage (ELD), external ventricular drainage (EVD), and CSF shunting are common procedures in daily neurosurgical practice. Complications secondary to CSF diversion procedures are diverse in presentation. Transtentorial herniation, intracranial hemorrhage, shunt malfunction and meningitis are relatively common and fatal complications [1–5]. But cranial nerve palsy (CNP) secondary to CSF diversion is less familiar to us as a result of its rarity in incidence and insidiousness in presentation [6]. In this study we would like to present 5 cases of CNPs after different CSF diversion procedures. To further expound this rare entity, we conducted an extensive review of the literature. The pathophysiological mechanism, clinical presentation, diagnosis, management and prognosis of this unique entity would be discussed in detail.

2.1. Definition of CNP secondary to CSF diversion

∗ Corresponding author at: Department of Neurosurgery, The First Hospital of Jilin University, 3302 Jilin Road, Changchun 130031, China. Fax: +86 043184808174. E-mail address: [email protected] (K. Hou). http://dx.doi.org/10.1016/j.clineuro.2016.02.010 0303-8467/© 2016 Elsevier B.V. All rights reserved.

When CNP secondary to CSF diversion was considered, the following criteria should at least be met simultaneously.

(1) One or more of the CSF diversion procedures such as LP, ELD, EVD, and CSF shunting was/were performed before the symptom onset of CNP. Because procedures as spinal anesthesia and myelography often involve CSF depletion, it was also considered one kind of CSF diversion in this study. (2) CNP occurred before the CSF diversion modality was removed or when it was still exerting effect. (3) No other definite causes responsible for the genesis of CNP were identified by the radiological, laboratory and clinical investigations.

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Fig. 1. Preoperative head CT shows subarachnoid hemorrhage concentrated in the left sylvian fissure (a), further CTA reveals an aneurysm at the bifurcation of the left middle cerebral artery (b, arrow). Postoperative CT shows a tight ventricular system and midline shift to the right side (c).

2.2. Case series in our institution A retrospective review of the medical records of the patients who had undergone all kinds of CSF diversion procedures in our institution from June 2012 to February 2015 was performed. Clinical data including primary disease and CSF diversion procedure, cranial nerve inflicted, accompanying chronic disease as diabetes and hypertension, management, outcome, and follow-up of each patient were collected for data interpretation.

diversions in patients with anomalies in the posterior fossa; e.g. Dandy–Walker syndrome and trapped fourth ventricle; endoscopic third ventriculostomy and shunt malfunction were excluded from this study.

3. Results 3.1. Current series

2.3. Literature search A systematic PubMed search of published studies written in English for patients developing CNPs after CSF diversion procedures from January 1950 to June 2015 was conducted. The following key words were used as searching conditions: “cranial nerve palsy,” “shunt,” “shunting,” “drainage,” “drain,” “hydrocephalus,” “lumbar puncture,” and “spinal anesthesia.” The references of all identified articles were also manually searched for additional studies. Because of the complexity in mechanism; CNPs secondary to CSF

From June 2012 to February 2015, 347 consecutive patients had undergone all kinds of CSF diversion procedures in our institution. Among them 5 female patients (4 after external lumbar drainage, 1 after ventriculoperitoneal shunt) developed CNPs (Table 1). The primary diseases were intracranial aneurysms in 4 patients and obstructive hydrocephalus secondary to aneurysmal subarachnoid hemorrhage (SAH) in 1 patient. The inflicted cranial nerves were cranial nerve III (1), VII (2), and VI (2). All the patients recovered completely in a time course from 1 h to 3 months after CNPs onset.

Fig. 2. Physical examination shows right ptosis (a) and motility limitation of the right eye when looking leftward (d), downward (e) and upward (f). No evident abnormality was noted when looking forward (b) and rightward (c).

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Fig. 3. Follow-up 3 months later shows resolution of the right ptosis (a). The right eye motility is recovered in every direction (b–e).

Table 1 Clinical data of the patients with CNP secondary to CSF diversion in current case series. Patient (sex, age)

Primary disease and type of CSF diversion

1 (F, 49)

Warning symptoms

CN inflicted

Time of CNP onset

Management

Outcome

Follow-ups (month)

No Left MCA aneurysm, intraoperative ELD No Right PComA aneurysm, intraoperative ELD

Negative

Right III

3 days postoperatively

Reducing ELD volume

CR 3 months later

24

Negative

Left VII

CR 1 h after onset

24

3 (F, 61)

Left A1 aneurysm, No intraoperative ELD

Negative

Right VII

CR 2 days postoperatively

12

4 (F, 69)

AComA aneurysm, No intraoperative ELD Obstructive Diabetes hydrocephlus after aSAH V-P shunt

Negative

Right VI

CR 2 months later

9

Negative

Right VI

30 min after Clamping ELD, withdrawing the intravenous tracheal intubation hydration, Trendelenburg position Immediately after Clamping ELD, withdrawing the intravenous tracheal intubation hydration, Trendelenburg position 5 days Reducing ELD postoperatively volume 3 days Intravenous postoperatively hydration

CR 1 months later

6

2 (F, 61)

5 (F, 50)

Accompanying diseases

F: female, CSF: cerebrospinal fluid, CN: cranial nerve, CNP: cranial nerve palsy, ELD: external lumbar drainage, CR: complete resolution, MCA: middle cerebral artery, PComA: posterior communicating artery, A1: anterior cerebral artery segment 1, V-P shunt: ventriculoperitoneal shunt, aSAH: aneurysmal subarachnoid hemorrhage.

Fig. 4. Head CT shows diffuse SAH (a), which is verified a right posterior communicating artery aneurysm by CTA (b, arrow). Postoperative CT reveals evidently collapsed ventricular system and subarachnoid space (c).

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Accompanying chronic diseases as diabetes or/and hypertension were only indentified in one patient.

ventricular size. No special management was given. She experienced a favorable recovery of the abducens nerve palsy in one month (Fig. 8).

3.2. Illustrative cases 3.2.1. Patient 1 A 49-year-old female was admitted to our center from a local hospital for spontaneous subarachnoid hemorrhage three days previously (Fig. 1a). Physical examination revealed nothing remarkable except neck stiffness. Computerized tomography angiography (CTA) showed a left middle cerebral artery aneurysm (Fig. 1b, arrow). Microsurgical clipping of the aneurysm was planned. A lumbar catheter was placed preoperatively to facilitate intraoperative brain relaxation and postoperative drainage of bloody CSF. The aneurysm was uneventfully clipped via left pterional approach. On day 3 postoperatively, she began to complain of diplopia. Physical examination demonstrated right cranial nerve III palsy (Fig. 2). Immediate CT scan revealed no postoperative intracranial hematoma but a tight ventricular system and midline shift to the opposite side (Fig. 1c). Further conventional angiography revealed no undiscovered aneurysm or vasospasm. As overdrainage was suspected, temporary clipping of the catheter combined with intravenous hydration was initiated. Her external ocular muscles began to recover gradually. She was discharged 2 weeks postoperatively. Three months later her cranial nerve III palsy was completely recovered (Fig. 3). 3.2.2. Patient 3 A 61-year-old female was admitted with a complaint of severe headache for three days. Physical examination revealed nothing remarkable except neck stiffness. She denied history of any other chronic diseases as hypertension or diabetes. CT performed on admission showed diffuse SAH (Fig. 4a), which was verified a right posterior communicating artery aneurysm by later CTA (Fig. 4b, arrow). The aneurysm was microsurgically clipped via right pterional approach under general anaesthesia and tracheal intubation. An ELD was implanted preoperatively. The intraoperative process was unremarkable. But she developed effacement of the right nasolabial groove and tilting of the mouth to the left right after withdrawing the tracheal intubation (Fig. 5a). Immediate CT scan showed evidently collapsed ventricular system and subarachnoid space (Fig. 4c). As excessive drainage of CSF was suspected, clamping the ELD, intravenous hydration and Trendelenburg position were initiated immediately. Her facial paralysis dramatically resolved with a slight shallow right nasolabial groove one hour after withdrawing the tracheal intubation (Fig. 5b). Two days postoperatively, no perceived difference was noticed between the two sides of her face. 3.2.3. Patient 5 A 50-year old female with a history of diabetes mellitus for three years was admitted for sudden headache and decline in mental state. Subsequent CT showed diffuse SAH and ventricular enlargement (Fig. 6a,b), which was demonstrated to be an internal artery aneurysm at the left side. She underwent an uneventful left pterional craniotomy and microsurgical clipping of the aneurysm. Postoperative course was unremarkable and she was discharged 14 days after admission. One month later she was readmitted for diffuse headache and vomiting. CT showed ventricular enlargement and periventricular edema (Fig. 6c). A ventriculoperitoneal shunt via the right occipital horn was performed. Her symptoms of headache and vomiting resolved immediately after surgery. But at day 3 postoperatively, she began to complain of diplopia at right horizontal gaze, which was demonstrated to be incomplete right abducens nerve palsy on physical examination (Fig. 7). CT revealed resolution of the periventricular edema and slight decrease of the

3.3. Searching results The PubMed search identified 29 studies reporting of 48 patients (Table 2) met the inclusion criteria. In all there were 53 (36 females, 68%) patients of CNP secondary to CSF diversion plus 5 cases in our case series. The inflicted patients aged from 5 to 82 years (43.7 ± 12.1). Cranial nerves (CNs) that got involved comprise CN II, III, IV, V, VI, VII and VIII. CN II, III, IV, V, VI, VII and VIII were got involved in 2 (3.8%), 2 (3.8%), 5 (9.4%), 1 (1.9%), 44 (83.0%), 4 (7.5%) and 1 (1.9%) patients respectively. In 34 patients (64.2%) a unilateral isolated cranial nerve was got involved, in 16 patients (30.2%) bilateral cranial nerves were got involved, in 6 patients (11.3%) more than one cranial nerves were got involved. In the 53 patients 32 (60.4%) had right CNPs, while 35 (66.0%) had left CNPs. Thirty-eight patients (71.7%) developed CNPs following inadvertent lumbar puncture, 8 (15.1%) following lumbar drainage, and 7 (13.2%) following ventriculoperitoneal shunt. Postural headache was the commonest warning or accompanying sign of CNPs in 43 (81.1%) patients. The time interval from CSF diversion to CNP onset was from during surgery to 20 days postoperatively (5.5 ± 3.0), excluding one was 2 years postoperatively and the other one the time interval could not be identified. Fortyeight (90.6%) patients got resolved completely. In the identified patients only 15 provided a definite description of whether having any chronic accompanying diseases or not, with 5 had diabetes or/and hypertension.

4. Discussion In general, CNP secondary to CSF diversion was classically deemed as the consequence of CSF leakage and subsequent intracranial hypotension (ICH) [12,14,23]. This speculation was based on the finding that in most of the patients, the opening pressure was low during lumbar puncture. However, later studies found that some patients with so-called ICH had normal CSF pressure [36,37]. Further studies are in favor of the notion that decreased CSF volume (CSF hypovolemia), rather than decreased CSF opening pressure, seems to be the core pathogenetic factor, whereas CSF opening pressure, MRI findings, and clinical features seem to be variables dependent on the CSF volume [36,38,39]. So, in view of the facts listed above, some scholars prefer hypovolemia rather than ICH as the cause of this specific entity [36,38,39]. The expressions of this entity might differ, but they share the common pathophysiological process that CSF depletion compromises its buoyant effect on the intracranial structures which leads to downward displacement of the brain and traction and distortion of the vascular and peripheral neural structures (including CNs). Postural headache and CNPs are the result of this process. In this study, we found that patients with CNPs often (81.1%) have alarming or accompanying postural headache. So we believe that early recognition and management in patients, who have undergone any kind of CSF diversion procedures recently, presenting with postural headache might avoid the progression to CNP to some extent. The relationship between chronic diseases as diabetes and hypertension and CNP secondary to CSF diversion has never been mentioned by the previous studies. In this review, only 15 patients provided a definite description of whether having any chronic accompanying diseases or not, with 5 had diabetes or/and hypertension. So the relationship between diabetes or/and hypertension and CNP secondary to CSF diversion could not be determined now.

Table 2 Clinical data of the patients with CNP secondary to CSF diversion in the literature. Patient (sex, age)

Primary disease and type of CSF diversion

Accompanying diseases

Warning symptoms

CN inflicted

Time interval from CSF diversion to CNP

Management

Outcome

Time to resolution

Bryce-smith (1951) [7]

1 (F, 42)

NA/NM

Nausea, neck pain, headache

Left VI

4 days

NA/NM

CR

3 months

NA/NM

Headache, nausea

Right VI

7 days

NA/NM

CR

6 weeks

Seyfert (1978) [8]

3 (M, 37) 4 (F, 50) 5 (M, 36) 6 (M, 35) 7 (F, 50) 8 (F, 60) 9 (F, 70) 10 (F, 72) 11 (M, 29) 12 (F, 33) 13 (M, 56) 14 (F, 34)

NA/NM NA/NM NA/NM NA/NM NA/NM NA/NM NA/NM NA/NM NA/NM NA/NM NA/NM NA/NM

Headache, nausea, stiff neck Headache, stiff neck Headache Headache Headache Headache, nausea, dizziness Headache, nausea Headache, nausea Headache Headache Headache Headache

Bilateral VI Right VI Left VI Bilateral VI, III Left VI Right VI Right V, Right VI Bilateral VI Bilateral VI Bilateral VI Bilateral VI Right VI

7 days 8 days 9 days 9 days 16 days 4 days 3 days (V), 7 days (VI) 8 days 6 days 2 weeks 8 weeks 3 days

CR CR CR CR CR CR CR CR CR CR CR CR

3 months 5 months 3 months 5 weeks 5 months 6 weeks 3 days (V), 2 months (VI) 12 weeks 5 months 5 months 4 months 6 weeks

NA/NM

Headache

VI

5 days

NA/NM NA/NM NA/NM NA/NM NA/NM Eye patch NA/NM Shunt revision NA/NM NA/NM NA/NM Intravenous hydration, EBP, eye patch NA/NM

CR

3 months

16 (M, 36) 17 (F, 33)

Cholecystectomy, dura piercing during epidural anesthesia Hysterectomy, dura piercing during epidural anesthesia Root compression, myelography Multiple sclerosis, myelography Root compression, myelography Root irritation, myelography Root irritation, myelography Depression, diagnostic LP Normal pressure hydrocephalus, V-P shunt Hydrocephalus, V-P shunt Posttraumatic headache, myelography Neck pain, myelography Low back pain, myelography Cesarean section, dura piercing during epidural anesthesia NA/NM, dura piercing during epidural anesthesia NA/NM, myelography Cesarean section, spinal anesthesia

NA/NM NA/NM

Headache Headache

VI Right IV, VI

4 days 6/7 days (VI), NA/NM (IV)

CR CR (VI), P/UR (IV)

1 month NA/NM

18 (M, 58)

Knee surgery, spinal anesthesia

Diabetes, hypertension

Right VI

20 days

P/UR

4 months

19 (F, 26)

Childbirth, dura piercing during epidural anesthesia Dizzy spell and headache, LP Childbirth, dura piercing during epidural anesthesia Dura piercing during epidural anesthesia Cervical spondylosis, myelography Low back pain and right-sided sciatica, myelography Low back pain and right-sided sciatica, myelography Low back pain and right-sided sciatica, myelography Low back pain, myelography Intrathecal injection Childbirth, dura piercing during epidural anesthesia Chronic, intractable lower back pain, implantation of an intrathecal drug delivery device Demyelinating neuropathy, diagnostic LP Demyelinating neuropathy, diagnostic LP Epilepsy, diagnostic LP

NA/NM

Headache, urinary incontinence Headache

NA/NM Epidural saline patch, prism glass prism glass

VI

8 days

EBP

CR

3 months

NA/NM NA/NM

Headache Headache

Bilateral VI Right VII, VIII

10 days 7 days

EBP EBP

CR CR

4 months 4 months

NA/NM NA/NM NA/NM

Headache Headache, dizziness, vomiting Headache, vomiting

Bilateral VI Left VI Right VI

7 days (L), 10 days (R) 9 days 12 days

EBP NA/NM NA/NM

CR CR CR

2 months 6 months 4 months

NA/NM

Headache

Left VI

7 days

NA/NM

CR

3 months

Neuralgic shoulder amyotrophy, diagnostic LP Childbirth, dura piercing during epidural anesthesia Aortic dissection, ELD Neurogenic bladder, dura piercing during epidural anesthesia Hallux valgus, dpinal anesthesia Headache, diagnostic LP Obstructive hydrocephalus, V-P shunt Obstructive hydrocephalus, V-P shunt Intracranial aneurysm, LP

Insel (1980) Black (1981) [10] Miller (1982) [11]

2 (F, 49)

[9]

Heyman (1982)

[12]

15 (F, 25)

Moster (1984) [13] King (1987) [14] Richer (1989)

[15]

Dunbar (1994) [16]

20 (M, 31) 21 (F, 20) De Veuster (1994) [17] Bell (1994) [18]

22 (F, 47) 23 (F, 39) 24 (F, 46) 25 (F, 41) 26 (F, 33)

Dumont (1998) [19] Szokol (1999) [20]

27 (F, 47) 28 (F, 38) 29 (F 32)

Velarde (2000) [21]

30 (M, 58)

Thömke (2000) [22] Follens (2001)

31 (M, 61) 32 (M, 33) 33 (M, 37)

[23]

Niedermüller (2002)[24]

34 (M, 43)

Chohan (2003) [25]

35 (F, 27)

Cheung (2003) [26] Arcand (2004) [27]

36 (F, 41) 37 (F, 40)

Kose (2005) [28] Béchard (2007) [29] Pramod (2008) [30] Yaman (2010) [31]

38 (M, 38) 39 (F, 45) 40 (F, 20) 41 (M, 16) 42 (F, 53)

Kim (2010) [32]

43 (F, 54)

Giesemann (2012) [33] Rowe (2012) [34]

44 (M, 82) 45 (F, 21)

Hiraishi (2013) [35]

47 (F, 58)

Rachel (2013) [6]

48 (F, 65)

46 (M, 31)

Neck pain, dura piercing during epidural anesthetic injection Normal pressure hydrocephalus, V-P shunt Idiopathic intracranial hypertension, V-P shunt combined with ELD Idiopathic intracranial hypertension, L-P shunt Vestibular schwannoma, intraoperative CSF drainage Pituitary macroadenoma, ELD

Headache, nausea

Left VI

1 day

Dexamethasone

CR

2 weeks

Negative Headache Headache

Left VI Right VI Left VI

5 days 8 days 6 days

NA/NM EBP EBP, eye patch

CR CR CR

3 months 4 months 8 weeks

NA/NM

Headache

Left VI

6 days

EBP

CR

2 weeks

Diabetes Diabetes NA/NM

Headache Headache Headache

Right VI Bilateral VI Left VI, right IV

7 days 9 days 5 days (VI), NA/NM (IV)

CR CR P/UR

4 months 7 months NA/NM

No

Headache

Left VI

4 days

NA/NM NA/NM EBP, multiple surgical intervention Eye patch, prism glass, EBP

CR

4 months

NA/NM

Headache

Left VI

5 days

EBP

CR

3 months

NA/NM NA/NM

Headache Headache, nausea

Right VI Bilateral VI

1 days 10 days

ELD removal EBP

CR CR

NA/NM 36 days

No No No No Hypertension

Headache, nausea, vomiting Headache Negative Negative Headache

Bilateral VI Bilateral VI Left IV Left IV Left VI

4 days 5 days 1 day 1 day Immediately postoperatively

CR CR CR CR P/UR at 6 months

6 months 21 months 1 year 1 year NA/NM

No

Headache

Left VI

4 days

NSAIDs, steroid EBP Inferior oblique recession Inferior oblique recession intravenous hydration, bed rest, NSAID NA/NM

CR

3 months

No NA/NM

Headache Headache

Bilateral IV Bilateral II, VI

NA/NM Immediately postoperatively

NA/NM Multiple surgical precedures

P/UR CR

NA/NM 3 months (VI), NA/NM (II)

NA/NM

Headache

Bilateral II

2 years

Shunt ligation

CR

NA/NM

NA/NM

NA/NM

Left VII

Intraoperatively

NA/NM

CR

9 months

NA/NM

Negative

Left VI

Immediately postoperatively

Steroids, eye patch, prism glass

CR

3 months

M: male, F: female, CNP: cranial nerve palsy, CSF: cerebrospinal fluid, V-P shunt: ventriculoperitoneal shunt, L-P shunt: lumbar-peritoneal shunt, ELD: external lumbar drainage, LP: lumbar puncture, NA/NM: not applicable/not mentioned, CR: complete resolution, P/UR: partial/un-resolution.

23

NA/NM NA/NM NA/NM NA/NM

G. Li et al. / Clinical Neurology and Neurosurgery 143 (2016) 19–26

First author (year)

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Fig. 5. The patient develops effacement of the right nasolabial groove and tilting of the mouth to the left right after withdrawing the tracheal intubation (a). Her right facial paralysis resolves dramatically after clamping of the external lumbar drainage, intravenous hydration and Trendelenburg position (b).

It is well known that CN VI is most commonly affected by injuries from alterations in intracranial pressure and displacement of intracranial contents [18,40]. More than 83% (44/53) of the affected patients in this review presented with abducens nerve palsy. The vulnerability of CN VI might be attributed to its local anatomic characteristics rather than its long intracranial course [6,31,40]. The intracranial portion of the CN VI, which is vulnerable

to injuries from distortion and displacement of the brain, courses from its exit at the brain stem to its penetration of the arachnoid and dura at Dorello’s canal. It is tethered both at its emergence from the medullopontine junction and exit at the Dorello’s canal. Furthermore, it has three angulation points between the dural entrance point and its anastomosis with the periarterial sympathetic plexus. These three angulation points have different angles in the

Fig. 6. CT on admission shows diffuse SAH (a) and ventricular enlargement (b). CT on her readmission one month later shows ventricular enlargement and periventricular edema (c).

Fig. 7. Comparison between the two eyes shows motility limitation of the right eye when looking outward 3 days postoperatively (a, b).

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Fig. 8. Outward motility of the right eye recovers 1 month later.

horizontal and sagital planes [31]. During caudal displacement of the brain tissue, CN VI is stretched where it turns into Dorello’s canal. And it might also suffer from direct compression by the clivus, basilar artery and anterior inferior cerebellar artery [40]. Generally speaking, CSF diversion procedures could be divided into transventricular and translumbar types. In this review, we found that CNPs secondary to transventricular procedures were less common than translumbar ones (7 vs 46). This might be partly due to the fact that translumbar procedures are more frequently performed than the transventricular ones. Furthermore, translumbar procedures might more readily facilitate the development of negative pressure gradient between the intracranial and spinal compartments. And this may cause caudal displacement of the brain. It is worth mentioning that there were 8 cases (4 from of the past reports and the others from our case series) of CNPs secondary to lumbar drainage hitherto (Tables 1 and 2). With the popularity of ELD, more and more cases of CNPs following ELD would be encountered in the future. Taking into consideration of this rare complication beforehand would be in favor of early recognition and management. The diagnosis of CNP secondary to CSF diversion should be firstly based on the history of CNP following one or more CSF diversion procedures recently. According to our review, the time interval from CSF diversion to CNP onset was from 0 to 20 days postoperatively (5.5 ± 3.0). Though nonspecific, postural headache, nausea and vomitting are very important warning signs of excessive CSF depletion. Routine head CT is a preliminary and right at hand screening method if CSF hypovolemia is suspected. Effacement of the intracranial cisterns and collapse of the ventricular system are the commonest findings. Subdural effusion could also be noticed sometimes [1,21]. If head CT is not so informative further MRI should be performed. Apart from the common findings in head CT, brain and brainstem descent, crowding of the posterior fossa and diffuse pachymeningeal enhancement in MRI are more direct and specific signs of CSF hypovolemia [39]. MRI is also helpful in identifying the site of CSF leakage in some patients. Further LP is not a requisite in the diagnosis of CSF hypovolemia. If the site of CSF leakage needs to be identified for an EBP or repair surgery, myelography could be considered. There is no established rule concerning the management of CNP secondary to CSF diversion till now. Fortunately, 90.6% (48/53) of the patients, according to this review, got complete resolution after a variety of treatment strategies. Usually, bed rest, analgesics and intravenous hydration were most commonly recommended to relieve postural headache and compensate CSF depletion [6,12,25,28,31] (Table 1). In patients for whom CNPs were after inadvertent LP, epidural blood patch (EBP) within 24 h might possibly lead to partial improvement and/or earlier resolution of symptoms [29]. Furthermore, early EBP might also have a prophylactic effect in preventing the occurrence of CNP in patients with post dural puncture headache [29]. However, in patients with

lumbar drain, temporary ligation of the drain was also effective according to the past report and our case series [34] (Table 1). If all of the managements aforementioned fail to exert effect further strabismus surgery should be implemented [23,30]. Considering the long time course from symptom onset to complete resolution, no consensus on the timing of strabismus surgery has been reached till now. 5. Conclusions CNP is an uncommon complication of CSF diversion procedures. The proposed mechanism is CSF hypovolemia and subsequent downward displacement of the brain and traction and distortion of the vascular and peripheral neural structures. As a result of its distinct anatomic characteristics rather than long intracranial course, CN VI is most commonly affected. Most of the reported cases were secondary to inadvertent LP and with only a small percentage of them were secondary to lumbar drainage and ventriculoperitoneal shunt. With early recognition and timely conservative management, most patients could get favorable recovery. If complete resolution is not achieved after a certain period of observation, strabismus surgery should be recommended. Conflict of interest The authors declare that they have no conflict of interest. Authors’ contributions Author contributions to the study and manuscript preparation include the following. Conception and design: Guichen Li, Xiaobo Zhu. Acquisition of data: Yang Zhang, Jinchuan Zhao. Analysis and interpretation of data: Xiaobo Zhu, Zhiguo Han. Drafting the article: Guichen Li, Kun Hou. Critically revising the article: Kun Hou. All of the authors read and approved the final manuscript. Consent Written informed consents were obtained from the patients for publication of this manuscript and any accompanying images. Funding No funding. Acknowledgement Guichen Li and Xiaobo Zhu contribute equally to this manuscript and they conjointly share the role of first author.

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