Paradoxical herniation after decompressive craniectomy provoked by mannitol: A case report

INTRODUCTION

The paradoxical herniation is a rare but life-threatening complication of cerebrospinal fluid (CSF)drainage in patients with large decompressive craniectomies. This result is due to a combined effect of brain gravity, atmospheric pressure, and intracranial hypotension[1,2]. Paradoxical herniation has been reported after craniotomy in the context of CSF hypovolemia, usually ascribed to intraoperative or postoperative CSF drainage or spinal CSF fistula[1,3-5]. Currently, only a few cases have been reported that paradoxical herniation may occur in the absence of CSF drainage, which is somewhat different from our case[6-8]. We describe a unique case of spontaneous paradoxical herniation after intravenous infusion of mannitol and decompressive craniectomy.

But as I sat there in my quiet house, eating my dinner, the phone started ringing. First, a woman from our church called; she identified herself as Sue15 and offered to give me rides back and forth16 to the hospital. She even insisted on driving three hours each way to pick up Kate at college and bring her home for the weekend. Then someone else called and said she d stop by with a meal. I didn t even know these people! Finally, just before I went to bed, the phone rang again. It was my mom.

CASE PRESENTATION

Chief complaints

A 48-year-old male was admitted to our hospital with sudden weakness in his left limb.

History of present illness

Two hours before his arrival, he suddenly developed weakness in his left limb and was unable to stand while working. The neurological examination showed a Glasgow Coma Scale score of 8 and a left hemiparesis. An urgent head computed tomography (CT) examination revealed massive right temporal hemorrhage (Figure 1A and B). The patient underwent an emergent frontoparietal decompressive craniectomy with a duraplasty, and the hematoma was completely evacuated. A mass of abnormal blood vessels founded in the hematoma cavity during the operation was removed. Postoperative pathological examination revealed malformed blood vessels (Figure 1E and F). On the 1st d after the operation, the patient was fully awake with normal communication and directional movement of the right limb but had left hemiplegia. The head CT shows that the hematoma has been completely cleared,and the midline is almost in the middle (Figure 1C and D). The CT angiography shows normal cerebral vessels (Figure 1G and H). Although the flap pressure was not high, 50 g of 20% mannitol was given every 8 h to reduce local edema. On the morning of the 7th d after the operation, after an intravenous drip of 50 g of mannitol in the Fowler’s position, the neurological function of the patient continued to deteriorate, the right pupil dilated to 4 mm and the left to 2 mm. An urgent head CT revealed increasing midline shift, transtentorial herniation, and brainstem compression (Figure 2).

History of past illness

The patient had no history of systemic diseases.

Physical examination

Neurologic examination revealed a Glasgow Coma Scale score of 8, with no eyes open. The pupils dilated to 4 mm on the right and 2 mm on the left, with left hemiplegia.

Laboratory examinations

The authors declare that they have no conflict of interest.

Imaging examinations

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FINAL DIAGNOSIS

The patient was ultimately diagnosed with paradoxical herniation.

So far, there are only a few cases of paradoxical herniation without CSF drainage. If a patient begins rebleeding after intracranial hematoma removal, reoperation should be performed to remove the hematoma and cranioplasty; if paradoxical herniation occurs after surgery, the possibility of CSF loss during multiple operations should be considered[8]. Another report of paradoxical herniation that occurred 1 h after removing acute subdural hematoma was suspected to be due to the loss of a large amount of CSF during the operation; the patient improved after an immediate cranioplasty[6].Interestingly, a patient with massive cerebral infarction caused by middle cerebral artery embolization developed paradoxical herniation 8 d after decompressive craniectomy, and the neurological function in the upright position continued to deteriorate. These symptoms were resolved by placing the patient in the Trendelenburg position and hydrating the patient quickly, considering the location changes of the patient[7].

TREATMENT

Our case is unique in that paradoxical herniation occurred after a rapid intravenous drip of 50 g of 20% mannitol in the Fowler’s position, without CSF drainage. Importantly, we found that the skin flap significantly sank after mannitol. In this case, the paradoxical herniation caused by brain sag. This may be due to the loss of the close attachment of the dura mater to the skull, the brain to sag from its own gravity and the differences in the intracranial and extracranial pressure. Additionally, patients with low flap pressure cannot handle large doses of mannitol, as this can lead to life-threatening paradoxical herniation. In this case, the patient's life was quickly saved by changing their position, which avoided reoperation in the short term. Interestingly, several hours after the treatment, including supine position,intravenous rehydration, and stopping mannitol, the depressed flaps gradually protruded with the improvement of the nerve function, indicating ICP had increased.

OUTCOME AND FOLLOW-UP

A few hours later, with the sinking skin flap relieving, the neurological function of the patient was significantly improved, and consciousness returned. However, the left side remained hemiplegia, and the right pupil dilated to 4 mm. The next day, the head CT showed resolution of transtentorial herniation, a significant decrease in midline shift, and reappearance of basal cisterns (Figure 3A and B).The neurological function of the patient continued to improve, and the patient was successfully changed from a supine position to the Fowler’s position within a few days. Two weeks later, he was able to stand and walk with the help of others. The right pupil contracted to 3 mm and the left pupil 2 mm, with a restored light reflection. The head CT shows that hematoma and edema have been absorbed entirely(Figure 3C and D). He was eventually transferred to a rehabilitation facility, waiting for the next skull repair surgery. The 2-mo follow-up revealed a good prognosis with mild hemiplegia on the left side(muscle strength grade 4).

DISCUSSION

Now it is by no means certain that ‘Donkey Skin ‘ had not caught a glimpse of the prince, either when his eyes looked through the keyhole, or else from her little window, which was over the road. But whether she had actually seen him or only heard him spoken of, directly she received the queen’s command, she flung off the dirty skin, washed herself from head to foot, and put on a skirt and bodice of shining silver. Then, locking herself into her room, she took the richest cream, the finest flour, and the freshest eggs on the farm, and set about making her cake.

In silent awe20, I let the wordless, poignant21 communication between Katie and Stripe unfold. Acutely sensitive to her well being, Stripe intuitively softened22 his gait at the slightest perception of Katie s imbalance or discomfort23 in the saddle. The tone of her voice induced the same effect, even though she was unable to use verbal commands that the horse was trained to recognize. Surprise, delight, hesitation24, fear-Stripe understood and responded patiently, lovingly-like a great teacher.

Aggressive medical treatment was immediately initiated to lower intracranial pressure (ICP), including 30° Fowler’s position and 20 mg intravenous bolus of furosemide. However, neurological symptoms continued to worsen, and the patient went into lethargy. During the active preparation for the operation, a significant phenomenon was found: regular brain beats could be seen under his obviously sinking flap. Therefore, we considered that the brain herniation was caused by the brain sag due to low ICP, and therefore, instead of operating, the patient was therefore placed in a supine position and quickly replenished with 9% saline intravenously.

Paradoxical herniation, a rare complication of patients who have undergone craniotomy, refers to transtentorial herniation in the context of intracranial hypotension[3,6]. This life-threatening complication has been reported after various types of CSF depletion and drainage, including CSF leakage, lumbar puncture, lumbar drain, CSF shunt, and ventriculostomy in patients undergoing a decompressive craniectomy[4,9-11]. The pathological mechanism of this rarely reported complication is unclear and may be related to atmospheric pressure, cerebral gravity, positional changes, and CSF exhaustion[3,6,7]. Interestingly, there are many reports of sinking skin flap syndromes after large craniectomy, with a series of symptoms, including motor dysfunction, cognitive dysfunction, headache,mood disorders, and sensory disturbance, most of which can be improved by cranioplasty[12,13]. The syndromes have a similar pathological mechanism with paradoxical herniation; however, the difference is that the symptoms are mild, slow to deteriorate, not life-threatening, and usually occur several months after decompressive craniectomy[14,5].

Treatment of paradoxical herniation should include immediately placing the patient in the supine or Trendelenburg position, providing intravenous fluids, and discontinuing all medications designed to reduce ICP[7,8]. In addition, by clamping all CSF drainage tubes or performing CSF leakage repair to solve the underlying cause of CSF loss, clinical improvement is usually expected within a few hours[4].Emergency cranioplasty is also a treatment option[6].

CONCLUSION

After large bone craniectomy and decompression, long-term treatment with large amounts of mannitol may cause a rare paradoxical herniation. Paradoxical herniation should be considered if flaps are severely sunken, the brain beating under the skin flap is normal, and head CT excludes secondary intracranial hypertension. Placing the patient in the supine or Trendelenburg position, along with rehydration therapy, may quickly save lives without the need for another operation.

FOOTNOTES

Du C and Tang HJ designed this work, analyzed the data, and wrote the manuscript; Du C collected the patient's clinical data; Fan SM and Du C performed the surgery; Fan SM designed and reviewed this paper; all authors have read and approved the final version of this manuscript.

the Natural Science Project of Chengdu University Clinical School, No. 2020YYZ18.

Informed written consent was obtained from the patient for publication of this report and any accompanying images.

So the girl seated herself on the horse, and rode for a long, long way, and at last she came to the mountain, where an aged woman was sitting outside with a gold carding-comb.52 The girl asked her if she knew the way to the castle which lay east of the sun and west of the moon; but she said what the first old woman had said: I know nothing about it, but that it is east of the sun and west of the moon, and that you will be a long time in getting to it, if ever you get there at all; but you shall have the loan of my horse to an old woman who lives the nearest to me: perhaps she may know where the castle is, and when you have got to her you may just strike the horse beneath the left ear and bid it go home again. Then she gave her the gold carding-comb, for it might, perhaps, be of use to her, she said.

On the 7th d after the operation, the patient underwent a blood cell analysis and examinations of blood electrolyte, liver function, and renal function. The test results were all normal, except for a slight decrease in the blood sodium concentration (130.3 mmol/L) and chlorine concentration (94.3 mmol/L).On the 8th d after the operation, examination of blood electrolyte showed sodium concentration (132 mmol/L) and chlorine concentration (95.6 mmol/L).

The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).

An urgent head CT revealed increasing midline shift, transtentorial herniation, and brainstem compression (Figure 2).

China

Chuan Du 0000-0001-6995-4417; Hua-Juan Tang 0000-0002-3504-4623; Shuang-Ming Fan 0000-0001-5378-5487.

Now the farm was a royal farm, and, one holiday, when ‘Donkey Skin’ (as they had nicknamed the princess) had locked the door of her room and clothed herself in her dress of sunshine, the king’s son rode through the gate, and asked if he might come and rest himself a little after hunting. Some food and milk were set before him in the garden, and when he felt rested he got up, and began to explore the house, which was famous throughout the whole kingdom for its age and beauty. He opened one door after the other, admiring the old rooms, when he came to a handle that would not turn. He stooped and peeped through the keyhole16 to see what was inside, and was greatly astonished at beholding30 a beautiful girl, clad in a dress so dazzling that he could hardly look at it.

Ma YJ

Simon, what brings you here; are you on the look out for a bargain? I ve come to get some provisions, he answered, because some friends are coming to dine with me today, and it would give me much pleasure if you were to honour me with your company also

Filipodia

A creative publicist was walking by the blind man and stopped to observe that the man only had a few coins in his hat. He put a few of his own coins in the hat, and without stopping to ask for permission（） , took the sign, turned it around, and wrote a new message. He then placed the sign by the feet of the blind man, and left.

Ma YJ

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