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Congenital Horner's syndrome is an infrequent illness caused by a lesion of the cervical sympathetic nerve fiber. It's clinical features are facial anhidrosis, ptosis, miosis, and hypochromia iridis of the affected side. The subject of this report, a full-term male newborn, had had a smooth birth process but was found on the second day of life to have narrowing of the palpebral fissure and absence of facial flushing on the right side when he cried. Ophthalmologic examination revealed a smaller right pupil. The above abnormalities proved to result from a post-ganglionic lesion, after pharmacologic test. Roentgenograms of the skull, chest and cervical spine were normal, and a computed tomography scan of the cervical spine showed no abnormalities. The diagnosis was of congenital Horner's syndrome. Since no congenital Horner's syndrome to the newborn period could be found in previous literature, this report is presented.
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PMID:Congenital Horner's syndrome: report of one case. 151 4

A 5-year-old girl presented with headache, vomiting, flushing, ptosis, and paroxysmal tachycardia. The neurological findings were partial motor and sensory left trigeminal palsy, left conductive hearing defect, and left cerebellar deficit. The radiological and neuropathological findings were typical for trigeminal schwannoma, which is rare in childhood. The present patient is the youngest yet recorded.
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PMID:Trigeminal schwannoma in a child. 647 86

Cluster headache is a strictly unilateral headache that occurs in association with cranial autonomic features. We report a patient with a trigeminal nerve section who continued to have attacks. A 59-year-old man described a 14-year history of left-sided episodes of excruciating pain centred on the retro-orbital and orbital regions. These episodes lasted 1-4 h, recurring 2-3 times daily. The attacks were associated with ipsilateral ptosis, conjunctival injection, lacrimation, rhinorrhoea and facial flushing. From 1986 to 1988, he had trials of medications without any benefit. In February 1988, he had complete surgical section of the left trigeminal sensory root that shortened the attacks in length for 1 month without change in their frequency or character. In April 1988, he had further surgical exploration and the root was found to be completely excised; post-operatively, there was no change in the symptoms. From 1988 to 1999, he had a number of medications, including verapamil and indomethacin, all of which were ineffective. Prednisolone 30 mg orally daily rendered the patient completely pain free. Sumatriptan 100 mg orally and 6 mg subcutaneously aborted the attack after approximately 45 and 15 min, respectively. He was completely anaesthetic over the entire left trigeminal distribution. Left corneal reflex was absent. Motor function of the left trigeminal nerve was preserved. Neurological and physical examination was otherwise normal. MRI scan showed a marked reduction in the calibre of the left trigeminal nerve from the nerve root exit zone in the pons to Meckel's cave. An ECG-gated three-dimensional multislab MRI inflow angiogram was performed. No dilatation was observed in the left internal carotid artery during the cluster attack. Blink reflexes were elicited with a standard electrode and stimulus. Stimulation of the left supraorbital nerve produced neither ipsilateral nor contralateral blink reflex response. Stimulation of the right supraorbital nerve produced an ipsilateral response with a mean R2 onset latency of 36 ms and a contralateral response with a mean R2 onset latency of 32 ms. Lack of ipsilateral vessel dilatation makes the role of vascular factors in the initiation of cluster attacks questionable. With complete section of the left trigeminal sensory root the brain would perceive neither vasodilatation nor a peripheral neural inflammatory process; however, the patient continued to have an excellent response to sumatriptan. The case illustrates that cluster headache may be generated primarily from within the brain, and that triptans may have anti-headache effects through an entirely central mechanism.
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PMID:Persistence of attacks of cluster headache after trigeminal nerve root section. 1196 Aug 88

We report on a case demonstrating unilateral Horner's syndrome (HS) after lumbar epidural obstetric anesthesia. A healthy, 32-year-old woman with a breech presentation was scheduled for an elective Cesarean section. The patient had normal vital signs throughout the surgical procedure. The operation lasted for 50 min. In the recovery room, she complained of left nasal stuffiness, left cheek numbness, and heaviness in her left eye. Meanwhile, left nipple sensory loss was noted during baby suckling training. On physical examination, her left eyelid was droopy along with left-side ptosis and facial flushing. Reduced sensation over the left hemifacial region and upper arm was also noted, which resolved completely over the next 110 min. A diagnosis of unilateral HS was then made. Although typically a benign side effect which often spontaneously resolves, HS is likely to cause anxiety in both the patient and the doctor. Prompt recognition of this syndrome and determination of its cause from lumbar epidural anesthesia can prevent unnecessary and potentially dangerous diagnostic workup and can reassure both patients and clinicians. The patient was discharged from the hospital 5 days after onset with a good outcome.
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PMID:Postpartum unilateral Horner's syndrome following lumbar epidural anesthesia after a Cesarean delivery. 1555 11

We report a Japanese infant with Horner syndrome whose clinical examination and testing suggested the location of the causative lesion. A 4-year-old Japanese girl had an acquired right ocular ptosis and unequal pupils presenting shortly after birth. She also exhibited left hemifacial flushing and loss of sweating on the contralateral side (harlequin sign). Physical examination demonstrated 2.0 mm of ptosis of the right upper lid with normal elevator function. The diameters of the pupils were 4 mm on the left and 2.5 mm on the right. No sweating was induced in the right frontal region at 40 degrees C for 15 minutes of sweat challenge test. Otherwise, no abnormalities were found by the neurophysiologic examinations or magnetic resonance imaging of the brain. Based on the clinical examination, we speculated that the responsible lesion might be in the preganglionic areas. Harlequin sign was informative for making the diagnosis of Horner syndrome.
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PMID:Harlequin sign (hemifacial flushing and contralateral hypohidrosis) in a 4-year-old girl with Horner syndrome. 1691 33

We present a seven-month-old baby with miosis of the left pupil, left hypochromia, mild ipsilateral ptosis, left hemifacial anhidrosis and asymmetrical facial flushing. A diagnosis of Horner's syndrome (HS) was presumed and was confirmed by instillation of apraclonidine eye drops. Miosis was reversed upon apraclonidine instillation. Magnetic resonance imaging of the head, neck and thorax and ultrasonography of the neck and abdomen did not reveal any pathological conditions. Although delivery-related brachial plexus injury is known as the most common cause of congenital HS, it should be investigated and should include neuroimaging of the sympathetic pathway, to exclude a serious underlying disease. As in our case, a specific etiology may not always be elicited. Pharmacological testing with apraclonidine may be a practical alternative to cocaine in the diagnosis of HS.
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PMID:Congenital Horner's syndrome and the usefulness of the apraclonidine test in its diagnosis. 1692 Dec 19

Harlequin syndrome, the presentation of hemifacial flushing and sweating, is a well recognized, though rarely reported, phenomenon associated with cervical sympathetic trauma. It is thought to result from disruption to sudomotor and vasomotor neurons present in the cervical sympathetic chain. The more common Horner's syndrome classically comprises the triad of unilateral miosis, ptosis and ipsilateral facial anhydrosis, and may also present as a sequela of cervical sympathetic denervation. We report a 26-month-old child with concomitant Horner's and harlequin syndromes, following neck dissection to address a large cervical lymphatic malformation. To our knowledge this is the first reported case of both syndromes resulting from surgery, and illustrates the particular challenge of lymphatic malformations in neck surgery due to their non-adherence to anatomical planes.
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PMID:Coexisting harlequin and Horner syndromes after paediatric neck dissection: a case report and a review of the literature. 1908 11

Paroxysmal hemicrania is a rare syndrome characterized by repeated attacks of strictly unilateral, severe, short-lasting pain occurring with cranial autonomic features. The hallmarks of this syndrome are the relatively short attacks and the exquisite response to indometacin. We describe the phenotype of this condition in a series of 31 patients. The mean duration of attack was 17 min. The mean attack frequency was 11. The distribution of the pain was orbital and temporal in 77% of the patients, retro-orbital in 61%, frontal in 55%, occipital in 42%; although pain was also reported in the vertex, second division of trigeminal nerve, neck, nose, jaw, parietal region, ear, teeth, eyebrow, shoulder (ipsilateral and bilateral), arm and third division of trigeminal nerve. Of the cohort, 87% had lacrimation, 68% had conjunctival injection, 58% rhinorrhoea, 54% nasal congestion, ptosis and facial flushing. Other cranial autonomic features include eyelid oedema, forehead/facial sweating, sense of aural fullness and periaural swelling, miosis, mydriasis and swelling of the cheek. The majority of the patients (80%) were agitated or restless, or both, with the pain and 26% were aggressive. All patients had positive placebo control indometacin test (100-200 mg intramuscularly), or a positive oral indometacin trial or both. We suggest the International Headache Society criteria be revised to remove specification of attack site, and to include the full range of cranial autonomic features. Currently, the sine qua non for paroxysmal hemicrania is a response to indometacin. Since there is no reliable clinical marker of that response we recommend an indometacin test, either orally or by injection for any patient with lateralized discrete attacks of head pain with associated cranial autonomic symptoms.
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PMID:Paroxysmal hemicrania: a prospective clinical study of 31 cases. 1825 75

Horner's syndrome is characterized by a classic triad of ipsilateral pupillary miosis, partial eyelid ptosis, and facial anhydrosis. This case study reports a 7-year-old boy with right miosis, mild blepharoptosis, and iris hypopigmentation detected in a routine pediatric follow-up without ipsilateral facial anhydrosis, flushing, or pain. There was no history of birth trauma and test with cocaine provoked no response of the right pupil, suggesting right Horner's syndrome. Mediastinal tumor was ruled out and brain magnetic resonance imaging incidentally showed absence of flow in the right internal carotid artery. Subsequent magnetic resonance angiography demonstrated agenesis of the right internal carotid artery without other vascular-associated malformations. The final diagnosis was right, congenital Horner's syndrome due to ipsilateral internal carotid agenesis. We describe in detail the radiological findings and pathophysiological mechanisms of this unusual association.
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PMID:Agenesis of internal carotid artery in a child with ipsilateral Horner's syndrome. 1916 24

Hemicrania continua is an uncommon primary headache disorder, characterized by continuous unilateral pain, where pain exacerbations are associated with cranial autonomic features. The hallmark of this condition is the absolute response to indometacin. We describe the phenotype of this condition in a large series of patients. Thirty-six (92%) patients had side-locked pain and 3 (8%) had side-alternating pain. The majority (82%) of the patients had the chronic (unremitting) form and the severity range of background pain was 1-10 out of 10 on verbal rating scale, with a mean of 6.5. Thirty-eight (97%) of the patients rated the painful exacerbations between 6.5 and 10 with a mean of 9 and 28 (71%) described their severe pain as excruciating. Of the cohort, 97% had at least one cranial autonomic feature during exacerbations: 73% had lacrimation, 51% nasal congestion, 46% conjunctival injection and 40% ptosis and facial flushing. Other cranial autonomic features included rhinorrhoea, forehead/facial sweating, itching eye, eyelid oedema, sense of aural fullness and periaural swelling, miosis, mydriasis and swelling of the cheek and face. Thirty-one (79%) had phonophobia, which was unilateral in 14 (48%); 29 (74%) had photophobia, which was unilateral in 14 (48%); and 27 (69%) had motion sensitivity. In addition, about two-thirds were agitated or restless, or both, and about one-quarter were aggressive, mainly verbally, with severe pain. All patients had a positive placebo-controlled indometacin test (100-200 mg intramuscularly) or a positive oral indometacin trial, or both. We suggest the International Headache Society criteria be revised to remove the absence of side-shift pain as a criterion. Furthermore, revised criteria should encompass a more extensive range of cranial autonomic features and consider pain as fluctuating with moderate, severe and very severe intensity. Currently the sine qua non for hemicrania continua is a response to indometacin. Since there is no reliable clinical marker of that response, we recommend an indometacin test, either orally or by injection, for any patient with unilateral pain, with or without cranial autonomic symptoms.
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PMID:Hemicrania continua: a clinical study of 39 patients with diagnostic implications. 2055 16


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