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Published online 2012 Oct 15. doi: 10.1258/shorts.2012.011135
PMID: 23162681

We report a case of Horner's syndrome, which developed following a therapeutic blood patch for post-dural puncture headache.

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Introduction

A young patient with post-dural puncture headache (PDPH) following inadvertent dural puncture with a Tuohy needle developed Horner's syndrome following a therapeutic blood patch at the thoracic level. Other causes for Horner's syndrome such as carotid artery dissection and brainstem lesion were excluded with magnetic resonance imaging.

Case presentation

A 30-year-old woman presented for an elective nephrectomy and consented to epidural analgesia for postoperative pain relief. She had a past medical history of duplex kidney and previous partial nephrectomy following recurrent urinary tract injections from pelviureteric junction obstruction. Epidural insertion at the mid-thoracic level prior to surgery resulted in inadvertent dural puncture with a Tuohy needle.

On the second postoperative day, she developed bilateral frontal headaches that worsened when sitting forward, tinnitus, nausea and vomiting. A diagnosis of PDPH was made and treatment initiated with caffeine, oral analgesia and increased fluid intake. Although the patient subsequently failed to respond to conservative management, she developed wound sepsis, which the microbiology team felt would preclude an epidural blood patch due to the risk of neuraxial infectious complications. Computer tomography imaging revealed no space-occupying lesion in the brain, and a diagnosis of meningitis was excluded on the basis of a normal lumbar puncture. An autoimmune screen was negative.

The pain management team carried out a therapeutic epidural blood patch on the eighth postoperative day, after complete resolution of the sepsis. The patient was placed in the left lateral position and the epidural space located using a 16G Tuohy needle and loss of resistance to air technique. Correct placement of the needle in the epidural space was visually confirmed using C-arm fluoroscopy (Figure 1), prior to injection of twenty millilitres of sterile autologous blood.

Fluoroscopic image showing radio-opaque contrast spread into the epidural space

The following morning the patient reported no further headaches, but stated that her right pupil was smaller than the left, and that her right eyelid was starting to droop. A neurologist reviewed her and made a diagnosis of Horner's syndrome (miosis, ptosis and enophthalmos). Magnetic resonance imaging of her neck and brain revealed no brainstem lesion or carotid artery dissection.

Over the next two days her right pupil was starting to resume normal size and the patient was discharged home. During a telephone follow-up six weeks later, she reported that she had returned to work full-time and the headaches had not recurred.

Discussion

The diagnosis of PDPH is a clinical one, based on a typical history of a postural headache following dural puncture. The Headache Classification Committee of the International Headache Society defines PDPH as bilateral headaches that develop within seven days of after a lumbar puncture and disappears within fourteen days. The headache worsens within fifteen minutes of resuming the upright position, and disappears or improves within thirty minutes of resuming the recumbent position. The exact pathophysiology of PDPH remains unclear, but is thought to be due to a persistent leak of cerebrospinal fluid (CSF) from the subarachnoid space as a result of the dural puncture, resulting in a fall in both CSF volume and pressure. The reduced CSF volume may cause gravitational traction on pain-sensitive intracranial structures, resulting in the postural headaches, and may cause direct activation of adenosine receptions, which further stretch these structures in addition to causing cerebral vasodilatation. Approximately 1–2% of epidural blocks result in unintentional dural puncture, with headaches occurring in 30–70% of these cases.

Following the inadvertent dural puncture, the patient was monitored for characteristic features of PDPH, and when this was diagnosed a number of treatment modalities were offered, including an epidural blood patch. A recent Cochrane review concluded that there were too few trials to support or refute the use of prophylactic blood patching, whereas a therapeutic epidural blood patch showed benefit over conservative treatment. One study examining the timing of blood patching concluded that the success rate was lower if performed within the first twenty-four hours of the dural puncture, possibly due to a higher rate of CSF leakage interfering with blood clotting. As this patient had developed postoperative sepsis, the blood patch was postponed; an advisory committee has recommended that alternatives to neuraxial techniques should be considered in all patients at high risk of infectious complications.

There are currently very few systematic reviews or meta-analyses to assess the evidence for the management of PDPH. Conservative, non-invasive management options include bed rest, hydration, prone positioning, the use of abdominal binders, and analgesics.8 No analgesic agent has been shown to be superior over the other in treating PDPH, although they do provide symptomatic relief. One randomized controlled trial has shown the benefits of oral caffeine versus placebo.

Horner's syndrome, or oculosympathetic paresis, is a lesion along the sympathetic pathway supplying the head and neck; signs include a unilateral pupillary constriction, slight relative ptosis and enophthalmos, injected conjunctival vessels, and ipsilateral loss of sweating if the lesion is proximal to the superior cervical ganglion. It may be congenital or iatrogenic, or the result of pathology in the head, neck or surrounding structures. A MEDLINE literature search has revealed a number of published case reports of Horner's syndrome following epidural analgesia, with a 0.4–2.5% incidence in the labour population. More cases have been reported following thoracic than lumbar epidural analgesia, and with high sensory blockade. There may be a higher incidence following unintended subdural or paravertebral block. Horner's syndrome has also been reported following internal jugular vein cannulation, possibly due to excessive rotation of the head, direct blunt trauma to the cervical plexus, or local haematoma formation causing disruption of the sympathetic chain.

In this case report, fluoroscopic images prior to injection of blood into the epidural space demonstrated that two millilitres of radio-opaque contrast had spread to several spinal segments both superiorly and inferior. As twenty millilitres of autologous blood was subsequently injected, it is possible that there may have been inadvertent subdural or dural spread of blood through the previous puncture site, or high cephalad epidural spread with mass effect. The patient did not have signs of anhidrosis but presented with miosis, ptosis and enophthalmos; this could be caused by disruption of the oculosympathetic pathways where the second order neurons exit the spinal cord (C8/T1) on their way to the superior cervical ganglia, distal to the superficial cervical ganglion (Figure 2). It has been demonstrated in some patients that the preganglionic sympathetic outflow tract receives fibres from as low as T9.

The oculosympathetic pathway, showing the proposed site of the lesion causing Horner's syndrome

Horner's syndrome may be diagnosed pharmacologically with cocaine, followed by hydroxyamphetamine to localize the lesion. If the oculosympathetic pathway is intact, topical cocaine into the eye will cause mydriasis via noradrenaline reuptake inhibition; this will not occur if a lesion is present. Topical hydroxyamphetamine causes release of endogenous noradrenaline from presynaptic vesicles, resulting in mydriasis of the lesion is preganglionic (first or second order neuron), and no dilatation if the lesion is postganglionic (third order neuron).

There is no known treatment for Horner's syndrome caused by epidural blood patching. However, the signs and symptoms in the patient did begin to resolve within days, presumably as the blood in the epidural space was absorbed. Pharmacological testing was not required in this instance.

The case report was discussed at the local morbidity and mortality meeting. It was felt that a smaller gauge Tuohy needle should be used to reduce the risk of PDPH, and that the blood patch could have been attempted at a lower site (lumbar instead or thoracic) with a smaller volume of blood to reduce the risk of developing Horner's syndrome.

DECLARATIONS

Funding

The article processing fee will be met by: Vivek Mehta, Pain and Anaesthesia Research Centre St Bartholomew's Hospital West Smithfield London EC1A 7BE

Ethical approval

Written informed consent was obtained from the patient or next of kin

Contributorship

Both authors contributed equally to the preparation and approval of the final manuscript

Reviewer

Geoffrey Knox

References

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Published online 2014 Sep 29. doi: 10.3892/etm.2014.1993
PMID: 25371756
This article has been cited by other articles in PMC.

Abstract

Pneumothorax in patients with progressive systemic sclerosis (PSS) often presents as a difficult-to-treat disease. Autologous blood-patch pleurodesis has previously been used for the treatment of pneumothorax. Blood outside its own environment is an irritant; therefore, chest physicians must watch closely for an allergic reaction. The injection is simple, painless, causes no side effects, is an inexpensive treatment for pneumothorax and is available not only in patients with persistent air leak but also in those with residual air space. A case is reported here of blood-patch pleurodesis for pneumothorax in lung fibrosis due to PSS. As an alternative therapy for difficult-to-treat pneumothorax in patients with PSS with persistent air leak and residual air space, autologous blood-patch pleurodesis would be one of the treatment options.

Keywords: blood-patch pleurodesis, pneumothorax, lung fibrosis, progressive systemic sclerosis

Introduction

Pneumothorax is not a rare complication of lung fibrosis due to progressive systemic sclerosis (PSS); however, a number of patients with PSS who have extensive pulmonary fibrosis with enlarged sub-pleural blebs and honeycomb lungs have been shown to develop pneumothorax. Various treatment options have been described (). Surgery is a treatment option for recurrent and refractory pneumothorax. Chemical pleurodesis with talc or minocycline is also another treatment option, but there were complication with deterioration of pulmonary fibrosis especially in patients with pulmonary fibrosis. However, the success rate of the of PSS patients with pneumothorax appeared to be unsatisfactory. A case is reported here following successful treatment with a blood patch introduced into the chest via an intercostal chest drain. The purpose of this case report is to report the usefulness of this successful treatment.

Case report

A 69-year-old female was admitted to the Mito Medical Center (Mito, Japan) due to dyspnea on exertion and right chest pain lasting all day. Three years previously she was diagnosed as having PSS with lung fibrosis (Fig. 1). Since then, the patient had experienced a pneumothorax on the right side several times and had subsequently received a video-assisted thoracoscopic surgery for persistent pneumothorax.

So faraway pinc inc rar download. Chest computed tomography scan showing lung fibrosis at the time of diagnosis of progressive systemic sclerosis.

Physical examination on admission revealed decreased respiratory sound in the right lung and diffuse fine crackles in both lungs. Skin thickening and tightness were observed on the fingers of both hands. Laboratory data on admission were as follows: White blood cell, 4,900/μl; C-reactive protein, 3.77 mg/dl; anti-nuclear antibody, 1:640; anti-centromere antibody, 1:640; rheumatoid factor, 4 U/ml. Tests for anti-neutrophil cytoplasmic antibodies to proteinase 3 and myeloperoxidase and anti-ribonucleoprotein and -topoisomerase 1 antibodies were negative. A chest radiograph revealed pneumothorax in the right lung, and reticulonodular opacities predominant in the bilateral lower lungs (Fig. 2). A 20 French intercostal chest tube (Argyle thoracic catheter, Covidien, Tokyo, Japan) was inserted into the pleural cavity, but the air leak continued and no improvement of the pneumothorax was obtained. The patient declined surgery; therefore, considering the deterioration of her respiratory condition following pleurodesis by other chemical agents, pleurodesis through the instillation of an autologous blood-patch was selected. A total of 50 ml autologous blood was injected via the chest tube. The tube was clamped for 3 h and connected to suction. The procedure was repeated twice over the next two days. Discontinuation of the air leak was achieved in three days and the chest tube was removed. Eight months later the patient was still well and attending the outpatient clinic without any recurrent pneumothorax (Fig. 3). This therapy was approved by the National Health Insurance of Japan as a postoperative persistent air leak therapy and by the Ethics Committee of the Mito Medical Center, University of Tsukuba-Mito Kyodo General Hospital (Mito, Japan). Informed consent was obtained from the patient.

Chest radiograph reveals right lung pneumothorax, and reticulonodular opacities predominant in the bilateral lower lungs.

Chest radiograph 8 months after the autologous blood-patch pleurodesis shows no recurrence of pneumothorax.

Cs 1.6 Blood Patch Plugin

Discussion

PSS is a systemic disease that sometimes affects the lungs, resulting in lung fibrosis (). Diffuse interstitial fibrosis is the most common pulmonary manifestation and pneumothorax is usually associated with the pulmonary complication of interstitial fibrosis (). The underlying mechanism of pneumothorax is believed to result from the rupture of acquired subpleural cystic spaces associated with the diffuse interstitial fibrosis (). In patients with PSS with interstitial fibrosis, the distinctive rigidity of lung parenchyma may prevent pre-expansion of the ruptured lung. Additionally, immunosuppressants including corticosteroids, which are frequently used for PSS, may aggravate persistent air leak; therefore, pneumothorax in patients with PSS often presents as a difficult-to-treat disease and prognosis is predicted to be poor. Chemical pleurodesis with tetracycline or talc has been successfully used (–), but pleurodesis is more usually performed once the air leak has resolved. Chemical pleurodesis using such agents can contribute to the onset of acute exacerbation of the interstitial fibrosis (). Furthermore, if pleurodesis can be successfully achieved, the development of constrictive respiratory impairment may occur due to pleural thickening as a result of the chemical pleurodesis. If there is no improvement despite conservative treatment, a more invasive approach may be necessary. Video-assisted thoracic surgery is the next option for patients with recurrent pneumothorax and those for whom conservative treatment was not successful (,). Due to the severity of their underlying disease itself and their respiratory condition, these patients are often not suitable candidates for surgical treatment. As a consequence, their optimal management may be eventful. As an alternative therapy, autologous blood-patch pleurodesis has been used for the treatment of pneumothorax (–). Blood outside its own environment is an irritant; therefore, chest physicians must watch closely for an allergic reaction. The injection is simple, painless, causes no side effects and is an inexpensive treatment for pneumothorax available not only for patients with persistent air leak but also those with residual air space (). In the case reported here, recurrent pneumothorax developed shortly after surgical therapy for the disease and there was persistent air leak as well as residual air space. Due to the high risk of deterioration of the respiratory condition of the patient by tight chemical pleurodesis, we selected to seal the pleural space with the injection of autologous blood as a successful pleurodesis agent in the treatment of recurrent pneumothorax.

In conclusion, as an alternative therapy for difficult-to-treat pneumothorax in patients with PSS with persistent air leak and residual air space, autologous blood-patch pleurodesis would be one of the treatment options.

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