Migrainous Limb Pain. A Historical Note.
R.J. Guiloff1 and M. Fruns2
From the Department of Neurology, Westminster Hospital, Charing Cross and Westminster Medical School, London, UK. Reprint requests to: Dr. R.J. Guiloff, Westminster Hospital, 17 Page Street, London SW1P 2AP, UK. Current address: Institute of Neurology, Queen Square, London WC1, UK. Accepted for Publication: December 30, 1989. SYNOPSIS
Upper and lower limb pain associated to attacks of migraine or cluster headache has been mentioned by many authors since the early descriptions of Liveing, Gowers and Jeliffe. The symptom was also described by Sluder as part of the syndrome of "sphenopalatine ganglion neuralgia." Several authors in the 1920's and 1930's including Cushing and Harris reported cases currently classifiable as migraine or cluster headache with limb pain, but did not accept the mechanisms for pain proposed by Sluder. The scarcity of more recent reports suggests that many patients with migrainous limb pain may be assumed to have other causes for this pain. (Headache 30:138-141, 1990) INTRODUCTION
The existence of limb pain as a migrainous accompaniment has not been widely recognised1 although several authors have mentioned it in the past. Limb pain associated with migraine or cluster headache was recently reported in 22 patients and was seen with a frequency similar to hemiplegic migraine in a London district.2 The clinical features of the symptom suggested that it may be of central origin, like paraesthesiae or numbness, and weakness, two accepted focal migrainous manifestations. We review critically early clinical descriptions and controversies surrounding the mechanisms of migrainous limb pain. EARLY CLINICAL DESCRIPTIONS
In 1873 Liveing referred to limb pain in association with paraesthesiae in migraine.3 He quoted M. Piorry who stated that in severe migraine "one side of the tongue or face, the inferior members, and still more the superior, experience a painful sense of thrilling (frémissement douloureaux)." M. Piorry also described a slow march for the pain: "Commencing...in the tip of the tongue, at one part of the face, at the ends of the fingers or toes, it mounts little by little towards the cerebrospinal axis, successively disappearing about those parts where it was first developed...When this pain has reached the neighbourhood of the nervous centres, it ceases to be felt.''3 Gowers stated that, in migraine, pain may radiate from the side of the head to the neck and arm and described a patient with migraine preceded or followed by pain in the leg.4 Jeliffe described a patient with right sided migraine and ipsilateral pain and numbness in the arm and numbness and weakness in the leg.5 SLUDER'S SPHENOPALATINE GANGLION NEURALGIA OR LOWER HALF HEADACHE
Sluder developed the concept of sphenopalatine ganglion neuralgia or "lower half" headache between 1908 and 1915. He reported on ten patients with pain behind the eyes, upper jaw, hard or soft palate, nose, teeth, temple, occiput and neck.6 The pain "behaved like migraine." He added later pain in the upper part of the head above the line of the zygoma, teeth, throat and ear, mastoid, shoulder blade, shoulder, axilla and, when severe, also arm, forearm, hand and fingers.7 His cases 1 and 3 would now be classified as cluster headache and his case 15 as either chronic cluster headache or migraine. He reported a case with an attack of pain in the upper limb extending into the leg and feet and another with an episode culminating in severe pain extending to the entire left upper limb and leg.6 The breast as a site for pain, and upper limb stiffness and weakness, fortification scotomata and itching of the skin of the limb as associated phenomena, were added in 1913.9 The neuralgia may appear "as constant pain or it may stop and reappear cyclically as a migraine or it may stop and reappear with stabbing sharpness as a tic." He also referred to a case with violent neuralgia in "other parts of the body." Sluder used the term "nasal ganglion neuralgia" for pain in the head and upper face and "vidian neuralgia" to denote pain in the ear, mastoid, occiput, neck and upper limb.10 He believed that in the latter, pain impulses originated in the vidian nerve. The "sympathetic syndrome," including conjunctival and nasal congestion, was also reported that year but only five of the 12 cases described in detail had "vidian neuralgia" and three had "nasal ganglion neuralgia." In one
of the latter it was "cyclical" with scotomata scintilans, aphasia and amblyopia.11 Cushing described three cases of "Sluder's neuralgia" with pain, and one of them also weakness, in the ipsilateral upper limb.12 Harris believed that many of Sluder's cases were examples of migrainous, supraorbital and ciliary neuralgia and of chronic neuralgia of the upper jaw.13 He included in his classification of hemicrania a "chronic persistent neuralgia of the jaws, temple and side of the head and neck" and recognized that the pain may be constant, or throbbing and paroxysmal, and that it may extend to the arm.14,15 Vail's case16 probably had cluster headache and pain in the neck and shoulder. Only one case with upper limb pain was fully described by him in a report on 31 cases.17 Many of the 200 cases of "atypical facial neuralgia" reported by Glaser18,19 had radiation of pain from lower and upper jaw to the neck, shoulder and arm. One had also body paraesthesiae. The only fully described case with radiation of pain to the arm probably had chronic cluster headache. The body was mentioned as a rare site of pain. Brunelli reported a patient with left sided migraine and ipsilateral pain in the limbs and body at an international neurological meeting on pain in France20 and thought the pain had an autonomic origin. Several reasons may underlie subsequent controversies about "lower half headache." First, Sluder was convinced that pathology of the sphenopalatine ganglion was the central element linking a wide variety of symptoms. Some of his patients did not even have headache10,11 and he included cases with eye disorders, asthma and suppurative sinus disease. He stated that the symptoms responded to cocainization of, or alcohol injection into, the sphenopalatine ganglion6,7 but later wrote that a small number got worse and that the procedure may produce "benefit so transitory as to arouse the enquiry: "Was it worthwhile?.9 Second, rather than studying in detail the clinical features of patients with headache and limb pain, it was assumed that they were a group different from migraine. Thus Sluder and other early authors directed their efforts instead towards the mechanisms and pathways mediating limb pain and headache. Third, migrainous neuralgia or cluster headache was recognised as a particular variety of headache only later.13-15,21,22 EARLY THEORIES ON THE MECHANISMS OF LIMB PAIN IN SLUDER'S NEURALGIA
The origin of the pain impulses was sited by Sluder in the sphenopalatine ganglion or in the vidian nerve10 and by Vail in the vidian nerve.23 Both believed that these impulses would eventually reach the cervical sympathetic ganglia either via the carotid plexus (Sluder) or the deep petrosal, glossopharyngeal or vagus nerves (Vail) and then reach the cervical nerves or brachial plexus giving rise to pain in the arm and shoulder. Sluder claimed that faradic stimulation of the vidian nerve evoked a painful reaction along the "lines of the neuralgic distribution" in a patient described by him in 1910 who had pain extending to upper and lower limbs.11 Fay reported a case of "Sluder's syndrome" who did not improve after removal of the sphenopalatine ganglion and section of the posterior root of the fifth nerve but improved after superior cervical ganglionectomy.24 He stated that pressure over the sympathetic trunk below the operation site produced pain sometimes referred to the arm and shoulder and postulated that the pain fibers entered the cervical cord below the superior cervical ganglion, whether they came from the sphenopalatine ganglion above or the thoracic trunk below. Later, Fay proposed a vascular pain mechanism; vagal branches associated with large cranial vessels would travel with the sympathetic branches to their fields of distribution and fibers in the carotid sheath with connections to the vagus would enter the lower cervical and upper thoracic cord.25 Braeucker described a patient with pain in the left face, head, neck, shoulder, chest and arm who did not improve with removal of the gasserian and sphenopalatine ganglions but did so with division of the left rami communicantes C1-C4 and of branches of the superior sympathetic ganglion and hypoglossal nerve to the external carotid artery.26 Adson described pain in the shoulder by compression of the coeliac ganglion.27 Harris believed that sympathetic and psychogenic elements played a role and reported severe pain in the right shoulder, breast, arm and foot in one patient with right hemicrania, with stimulation of "the branches of the right stellate ganglion.''13 Others have later produced limb pain by sympathetic stimulation. Walker and Nulson described tingling burning or prickling sensation in the arm and hand in 3 out of 10 patients by electrical stimulation of the sympathetic chain between T2 and T3.28 Echlin reported pain in the lower limb, including phantom limb pain, in one case by electrical or mechanical stimulation of the sympathetic trunk in the lumbar segment.29 Kuntz described pain in the lower extremity by stimulation of the symapthetic trunk between the second and third ganglia but not when the connections of the sympathetic trunk with the second lumbar nerve were interrupted.30 Major clinical evidence against Sluder's theory was produced by Cushing who removed the sphenopalatine ganglion in one of Sluder's patients with no improvement.20 Other authors had equally negative results with this operation24-26,31 and with cocainization of the ganglion.18,19 No improvement with cervical sympathectomy and stripping of the periarterial carotid plexus was reported by several authors.18,19,32,33 Stimulation of the nasal and paranasal structures34 and of the greater, lesser and external superficial petrosal nerves and middle meningeal artery33 did not result in pain referred to the arm.
Frazier reported local pain in all but one case, who referred pain to the face, when he stimulated the cervical sympathetic ganglia or the periarterial carotid plexus.32 Definitive conclusions cannot be drawn from the early clinical data summarised above. Many of the cases are not described in enough detail. Different operative techniques may have been used by various workers. Follow-up is often insufficient or not given. The precise details of the stimulation techniques are often not available and conduction through the assumed pathways was not proven. Recent anatomical, physiological and clinical data relevant to the mechanisms of migrainous limb pain are discussed elsewhere.2 LIMB PAIN, MIGRAINE AND THE "THORACIC OUTLET SYNDROME"
Although the existence of limb pain associated with migraine or cluster headache is supported by the description of many patients by early workers, as reviewed above, it has only been mentioned sporadically in the more recent literature.2 Possible reasons include rejection of the diagnosis of migraine or cluster headache because of the presence of limb pain and the assumption that this symptom is psychogenic or is due to some other cause, such as cervical or lumbar radiculopathy. In addition, some patients diagnosed as having the thoracic outlet syndrome might have limb pain associated with migraine or cluster headache. Headache was present in between 23% and 78% of patients in two large surgical series.35,36 Clover et al reported a patient with a simultaneous recurrence of "symptoms and migraine" 13 months post-operatively.37 A presentation with unilateral headache, with the usual associations of migraine, before symptoms attributed to the thoracic outlet syndrome, was reported in 21 out of 30 patients; eleven of their patients still required vasoactive drugs after transaxillary first rib resection.38 In no case was improvement in headache not associated with improvement in the other symptoms. White and Sweet reported a woman with left hemicranial migraine who had anterior scalenectomy, resection of left cervical rib, procaine block of C2-C3-C4 and carotid bifurcation, and infiltration of the scalenectomy scar. She also had division of the cervical sympathetic trunk but "right sided pain and attacks of incapacitating migraine ten days per month continued.33 Further, in the absence of neurological signs, the clinical criteria for the diagnosis of the "thoracic outlet syndrome" are controversial.39,40 The location of the pain and its aggravation by certain postures or exertion38 can also be seen in other conditions. Obliteration of the pulse by certain manoeuvres may occur in healthy subjects.40,41 Swelling of the face15,42,43 Or limbs5,44 have been reported in cluster headache and hemiplegic migraine respectively. The presence of circulatory changes in the skin of patients with a diagnosis of "thoracic outlet syndrome" may not be a specific finding either Raynaud phenomena and migraine had both a similarly significantly higher incidence in patients with variant angina leading the authors to propose the existence of a generalised vasospastic disorder in their patients.45 In 2 of 3 patients with migraine and unilateral minor swelling and/or bluish skin patches in one upper limb in one series, limb pain alternated sides.2 CONCLUSION
The existence of limb pain as a migrainous accompaniment, associated with both migraine and cluster headache, is supported by many clinical descriptions since Liveing referred to it in 1873. The symptom was identified as part of sphenopalatine ganglion neuralgia or lower half headache, a syndrome claimed by Sluder to be different from migraine, but which included a heterogenous group of patients; attention was then focused on the mechanisms of pain rather than on the clinical features of limb pain and headache. Although some of the patients described by Sluder were subsequently acccepted as cases of cluster headache, limb pain as a migrainous symptom has not been widely recognised and references to it in the recent literature are sporadic. It is possible that many patients with migrainous limb pain are assumed to have other causes for this pain, including psycho-genic pain, cervical and/or lumbar radiculopathy or the thoracic outlet syndrome. REFERENCES
1.
Bruyn GW: Migraine equivalents. In: Handbook of Clinical Neurology, Volume 4(48). Edited by PJ Vinken, GW Bruyn, HL Klawans, FC Rose. Amsterdam: Elsevier Science Publishers, 1986, pp 155-171.
2.
Guiloff RJ, Fruns M: Limb pain in migraine and cluster headache. J Neurol Neurosurg & Psychiat 51:1022-1031, 1988.
3.
Liveing E: On Megrim, Sick-headache and AIlied Disorders: A Contribution to the Pathology of Nerve-Storms. London: J and A Churchill, 1873, p 481.
4.
Cowers WR: A Manual of Diseases of the Nervous System. Philadelphia: P Blakiston, Son and Co, 1988, pp 1176-1177.
5.
Jeliffe SE: Aphasia, hemiparesis and hemianaesthaesia in migraine. New York Mad J 83:33-36, 1906.
6.
Sluder G: The role of sphenopalatine (or Meckle's) ganglion in nasal headaches. New York Mad J 87:989-990, 1908.
7.
Sluder G: The anatomical and clinical relations of the sphenopalatine (Meckel's) ganglion to the nose and its accessory sinuses. New York Mad J 88:293-298, 1909.
8.
Sluder G: The syndrome of sphenopalatine-ganglion neurosis. American J of Mad Sci 140:868-878, 1910.
9.
Sluder G: Etiology, diagnosis, prognosis and treatment of sphenopalatine ganglion neuralgia. J of the American Mad Assoc 61:1201-1206, 1913.
10.
Sluder G: Hyperplastic Sphenoiditis: and its Clinical Relations to the Second, Third, Fourth, Fifth, Sixth and Vidian Nerves and Nasal Ganglion. Transactions of the American Laryngological Association 37:215-242, 1915a.
11.
Sluder G: The Sympathetic Syndrome of Sphenopalatine or Nasal Ganglion Neurosis: together with a Consideration
of the Neuralgic Syndrome: and their Treatment. Transactions of the American Laryngological Association 37:243-262, 1915b. 12.
Cushing H: The major trigeminal neuralgias and their surgical treatment based on experiences with 332 Gasserian operations. American J of Mad Sci 160:157-184, 1920.
13.
Harris W: Neuritis and Neuralgia. Oxford: Oxford University Press, 1926.
14.
Harris W: Ciliary (migrainous) neuralgia and its treatment. British Medical Journal 1:457-460, 1936a.
15.
Harris W: The role of the sympathetic in sensory conduction and certain neuralgias. British Medical Journal 2:112-115, 1936b.
16.
Vail HH: Vidian neuralgia from disease of the sphenoidal sinus. Report of a case. Archives of Surgery (Chicago) 18:1247-1255, 1929.
17.
Vail HH: Vidian neuralgia. Annals of Otology, Rhinology and Laryngology 41:837-856, 1932.
18.
Glaser MA: Atypical neuralgia, so called: A critical analysis of one hundred and forty-three cases. Arch Neurol & Psychiat (Chicago), 20:537-558, 1928.
19.
Glaser MA, Beerman HM: Atypical facial neuralgia: an analysis of 200 cases. Arch Int Med 161:172-173, 1938.
20.
Brunelli MA: Migraine et sympathalgia homolaterale de I'hemicorp. Revue Neurologique 68:167-169, 1937.
21.
Horton BT, Maclean AR, Craig W McK: A New Syndrome of Vascular Headache: Results of Treatment with Histamine: Preliminary Report. Proc Staff Meetings Mayo Clinic 14:257-260, 1939.
22.
Kunkle EC, Pfeiffer JB, Wilhoit WM et al: Recurrent brief headache in Cluster pattern. Transactions of the American Neurological Association 77:240-243, 1952.
23.
Vail HH: Pathways of Reflex Pain in Vidian Neuralgia. Arch Otolaryngol 21:277-284, 1935.
24.
Fay T: Atypical Neuralgia. Arch Neurol & Psychiat (Chicago) 18:309-313, 1927.
25.
Fay T: Atypical facial neuralgia, a syndrome of vascular pain. Annals of Otology, Rhinology and Laryngology 41:1030-1062, 1932.
26.
Braeucker W: Die Anatomie und Chirurgie des Vegetativen Nervensystems. Deutsche Zeitschrift fur Nervenheilkunde 106:163-171, 1928.
27.
Adson AW: Splachnic pain. Proc Staff Meetings Mayo Clinic 10:623-624, 1935.
28.
Walker AE, Nulson F: Electrical stimulation of the upper thoracic portion of the sympathetic chain in man. Arch of Neurol & Psychiat (Chicago) 59:559-560, 1948.
29.
Echlin F: Pain response to stimulation of the lumbar sympathetic chain under local anaesthesia. J Neurosurgery 6:530-533, 1949.
30.
Kuntz A: Afferent innervation of peripheral blood vessels through sympathetic trunks. Its clinical implications. South Mad J 44:673-678, 1951.
31.
Frazier CH, Russell EC: Neuralgia of the face. An analysis of 754 cases with relation to pain and other sensory phenomena before and after operation. Arch Neurol & Psychiat (Chicago) 11:557-563, 1924.
32.
Frazier CH: Atypical Neuralgia. Unsuccessful attempts to relieve patients by operations on the cervical sympathetic system. Arch Neurol & Psychiat (Chicago) 19:650-659, 1928.
33.
White JC, Sweet WH: Cephalic Pain-Transmission pathways uncertain. In: Pain. Its Mechanisms and Neurosurgical Control. Edited by JC White, WH Sweet. Charles C Thomas. Springfield, Illinois, 1955, pp 494-542.
34.
McAuliffe GW, Gooddell H, Wolff HG: Experimental studies on headache: Pain from the Nasal and Paranasal Structures. Res Publ Assoc Res Nerv & Mental Dis 23:185-207, 1943.
35.
McGough EC, Pearse MB, Byrne JP: Management of thoracic outlet syndrome. J Thoracic & Cardiovasc Surg 77:169-179, 1979.
36.
Sanders RJ, Monsour JW, Gerber WF et al: Scanelectomy versus first rib resection for treatment of the thoracic outlet syndrome. Surgery 85:109-119, 1979.
37.
GIover JL, Worth RM, Bendick PJ et al: Evoked responses in the diagnosis of thoracic outlet syndrome. Surgery 89:86-92, 1981.
38.
Raskin NH, Howard MW, Ehrenfeld WK: Headache as the leading symptom of the thoracic outlet syndrome. Headache 25:208-210, 1985.
39.
Eaton LM: Neurologic causes of pain in the upper extremities. Surg Clin North America 26:810-833, 1946.
40.
Gilliatt RW: Thoracic outlet syndromes. In: Peripheral Neuropathy. Edited by PJ Dyck, PK Thomas, EH Lambert, R Bunge, Philadephia: WB Saunders 1984, pp 1409-1419.
41.
Wright IC: The neurovascular syndrome produced by hyperabduction of the arms; immediate changes produced in 150 normal controls, and effects in some persons of prolonged hyperabduction of arms as in sleeping, and in certain occupations. Am Heart J 29:1-19, 1945.
42.
Lance JW, Anthony M: Migrainous neuralgia or cluster headache. J Neurol Sci 13:401-404, 1971.
43.
Sutherland JM, Eadie MJ: Cluster Headache. In: Research and Clinical Studies in Headache. An International Review, Volume 3. Edited by AP Friedman. Basel: Karger, 1972 pp 92-125.
44.
Montgomery BM, King WW: Hemiplegic Migraine. A case with Paroxysmal Shoulder-Hand Syndrome. Ann Int Mad 57:450-455, 1962.
45.
Miller DM, Waters DD, Warnica W et al: Is variant angina the coronary manifestation of a generalised vasospastic disorder? New Eng J Med 394:763-766, 1981.
R.J. Guiloff1 and M. Fruns2
From the Department of Neurology, Westminster Hospital, Charing Cross and Westminster Medical School, London, UK. Reprint requests to: Dr. R.J. Guiloff, Westminster Hospital, 17 Page Street, London SW1P 2AP, UK. Current address: Institute of Neurology, Queen Square, London WC1, UK. Accepted for Publication: December 30, 1989. SYNOPSIS
Upper and lower limb pain associated to attacks of migraine or cluster headache has been mentioned by many authors since the early descriptions of Liveing, Gowers and Jeliffe. The symptom was also described by Sluder as part of the syndrome of "sphenopalatine ganglion neuralgia." Several authors in the 1920's and 1930's including Cushing and Harris reported cases currently classifiable as migraine or cluster headache with limb pain, but did not accept the mechanisms for pain proposed by Sluder. The scarcity of more recent reports suggests that many patients with migrainous limb pain may be assumed to have other causes for this pain. (Headache 30:138-141, 1990) INTRODUCTION
The existence of limb pain as a migrainous accompaniment has not been widely recognised1 although several authors have mentioned it in the past. Limb pain associated with migraine or cluster headache was recently reported in 22 patients and was seen with a frequency similar to hemiplegic migraine in a London district.2 The clinical features of the symptom suggested that it may be of central origin, like paraesthesiae or numbness, and weakness, two accepted focal migrainous manifestations. We review critically early clinical descriptions and controversies surrounding the mechanisms of migrainous limb pain. EARLY CLINICAL DESCRIPTIONS
In 1873 Liveing referred to limb pain in association with paraesthesiae in migraine.3 He quoted M. Piorry who stated that in severe migraine "one side of the tongue or face, the inferior members, and still more the superior, experience a painful sense of thrilling (frémissement douloureaux)." M. Piorry also described a slow march for the pain: "Commencing...in the tip of the tongue, at one part of the face, at the ends of the fingers or toes, it mounts little by little towards the cerebrospinal axis, successively disappearing about those parts where it was first developed...When this pain has reached the neighbourhood of the nervous centres, it ceases to be felt.''3 Gowers stated that, in migraine, pain may radiate from the side of the head to the neck and arm and described a patient with migraine preceded or followed by pain in the leg.4 Jeliffe described a patient with right sided migraine and ipsilateral pain and numbness in the arm and numbness and weakness in the leg.5 SLUDER'S SPHENOPALATINE GANGLION NEURALGIA OR LOWER HALF HEADACHE
Sluder developed the concept of sphenopalatine ganglion neuralgia or "lower half" headache between 1908 and 1915. He reported on ten patients with pain behind the eyes, upper jaw, hard or soft palate, nose, teeth, temple, occiput and neck.6 The pain "behaved like migraine." He added later pain in the upper part of the head above the line of the zygoma, teeth, throat and ear, mastoid, shoulder blade, shoulder, axilla and, when severe, also arm, forearm, hand and fingers.7 His cases 1 and 3 would now be classified as cluster headache and his case 15 as either chronic cluster headache or migraine. He reported a case with an attack of pain in the upper limb extending into the leg and feet and another with an episode culminating in severe pain extending to the entire left upper limb and leg.6 The breast as a site for pain, and upper limb stiffness and weakness, fortification scotomata and itching of the skin of the limb as associated phenomena, were added in 1913.9 The neuralgia may appear "as constant pain or it may stop and reappear cyclically as a migraine or it may stop and reappear with stabbing sharpness as a tic." He also referred to a case with violent neuralgia in "other parts of the body." Sluder used the term "nasal ganglion neuralgia" for pain in the head and upper face and "vidian neuralgia" to denote pain in the ear, mastoid, occiput, neck and upper limb.10 He believed that in the latter, pain impulses originated in the vidian nerve. The "sympathetic syndrome," including conjunctival and nasal congestion, was also reported that year but only five of the 12 cases described in detail had "vidian neuralgia" and three had "nasal ganglion neuralgia." In one
of the latter it was "cyclical" with scotomata scintilans, aphasia and amblyopia.11 Cushing described three cases of "Sluder's neuralgia" with pain, and one of them also weakness, in the ipsilateral upper limb.12 Harris believed that many of Sluder's cases were examples of migrainous, supraorbital and ciliary neuralgia and of chronic neuralgia of the upper jaw.13 He included in his classification of hemicrania a "chronic persistent neuralgia of the jaws, temple and side of the head and neck" and recognized that the pain may be constant, or throbbing and paroxysmal, and that it may extend to the arm.14,15 Vail's case16 probably had cluster headache and pain in the neck and shoulder. Only one case with upper limb pain was fully described by him in a report on 31 cases.17 Many of the 200 cases of "atypical facial neuralgia" reported by Glaser18,19 had radiation of pain from lower and upper jaw to the neck, shoulder and arm. One had also body paraesthesiae. The only fully described case with radiation of pain to the arm probably had chronic cluster headache. The body was mentioned as a rare site of pain. Brunelli reported a patient with left sided migraine and ipsilateral pain in the limbs and body at an international neurological meeting on pain in France20 and thought the pain had an autonomic origin. Several reasons may underlie subsequent controversies about "lower half headache." First, Sluder was convinced that pathology of the sphenopalatine ganglion was the central element linking a wide variety of symptoms. Some of his patients did not even have headache10,11 and he included cases with eye disorders, asthma and suppurative sinus disease. He stated that the symptoms responded to cocainization of, or alcohol injection into, the sphenopalatine ganglion6,7 but later wrote that a small number got worse and that the procedure may produce "benefit so transitory as to arouse the enquiry: "Was it worthwhile?.9 Second, rather than studying in detail the clinical features of patients with headache and limb pain, it was assumed that they were a group different from migraine. Thus Sluder and other early authors directed their efforts instead towards the mechanisms and pathways mediating limb pain and headache. Third, migrainous neuralgia or cluster headache was recognised as a particular variety of headache only later.13-15,21,22 EARLY THEORIES ON THE MECHANISMS OF LIMB PAIN IN SLUDER'S NEURALGIA
The origin of the pain impulses was sited by Sluder in the sphenopalatine ganglion or in the vidian nerve10 and by Vail in the vidian nerve.23 Both believed that these impulses would eventually reach the cervical sympathetic ganglia either via the carotid plexus (Sluder) or the deep petrosal, glossopharyngeal or vagus nerves (Vail) and then reach the cervical nerves or brachial plexus giving rise to pain in the arm and shoulder. Sluder claimed that faradic stimulation of the vidian nerve evoked a painful reaction along the "lines of the neuralgic distribution" in a patient described by him in 1910 who had pain extending to upper and lower limbs.11 Fay reported a case of "Sluder's syndrome" who did not improve after removal of the sphenopalatine ganglion and section of the posterior root of the fifth nerve but improved after superior cervical ganglionectomy.24 He stated that pressure over the sympathetic trunk below the operation site produced pain sometimes referred to the arm and shoulder and postulated that the pain fibers entered the cervical cord below the superior cervical ganglion, whether they came from the sphenopalatine ganglion above or the thoracic trunk below. Later, Fay proposed a vascular pain mechanism; vagal branches associated with large cranial vessels would travel with the sympathetic branches to their fields of distribution and fibers in the carotid sheath with connections to the vagus would enter the lower cervical and upper thoracic cord.25 Braeucker described a patient with pain in the left face, head, neck, shoulder, chest and arm who did not improve with removal of the gasserian and sphenopalatine ganglions but did so with division of the left rami communicantes C1-C4 and of branches of the superior sympathetic ganglion and hypoglossal nerve to the external carotid artery.26 Adson described pain in the shoulder by compression of the coeliac ganglion.27 Harris believed that sympathetic and psychogenic elements played a role and reported severe pain in the right shoulder, breast, arm and foot in one patient with right hemicrania, with stimulation of "the branches of the right stellate ganglion.''13 Others have later produced limb pain by sympathetic stimulation. Walker and Nulson described tingling burning or prickling sensation in the arm and hand in 3 out of 10 patients by electrical stimulation of the sympathetic chain between T2 and T3.28 Echlin reported pain in the lower limb, including phantom limb pain, in one case by electrical or mechanical stimulation of the sympathetic trunk in the lumbar segment.29 Kuntz described pain in the lower extremity by stimulation of the symapthetic trunk between the second and third ganglia but not when the connections of the sympathetic trunk with the second lumbar nerve were interrupted.30 Major clinical evidence against Sluder's theory was produced by Cushing who removed the sphenopalatine ganglion in one of Sluder's patients with no improvement.20 Other authors had equally negative results with this operation24-26,31 and with cocainization of the ganglion.18,19 No improvement with cervical sympathectomy and stripping of the periarterial carotid plexus was reported by several authors.18,19,32,33 Stimulation of the nasal and paranasal structures34 and of the greater, lesser and external superficial petrosal nerves and middle meningeal artery33 did not result in pain referred to the arm.
Frazier reported local pain in all but one case, who referred pain to the face, when he stimulated the cervical sympathetic ganglia or the periarterial carotid plexus.32 Definitive conclusions cannot be drawn from the early clinical data summarised above. Many of the cases are not described in enough detail. Different operative techniques may have been used by various workers. Follow-up is often insufficient or not given. The precise details of the stimulation techniques are often not available and conduction through the assumed pathways was not proven. Recent anatomical, physiological and clinical data relevant to the mechanisms of migrainous limb pain are discussed elsewhere.2 LIMB PAIN, MIGRAINE AND THE "THORACIC OUTLET SYNDROME"
Although the existence of limb pain associated with migraine or cluster headache is supported by the description of many patients by early workers, as reviewed above, it has only been mentioned sporadically in the more recent literature.2 Possible reasons include rejection of the diagnosis of migraine or cluster headache because of the presence of limb pain and the assumption that this symptom is psychogenic or is due to some other cause, such as cervical or lumbar radiculopathy. In addition, some patients diagnosed as having the thoracic outlet syndrome might have limb pain associated with migraine or cluster headache. Headache was present in between 23% and 78% of patients in two large surgical series.35,36 Clover et al reported a patient with a simultaneous recurrence of "symptoms and migraine" 13 months post-operatively.37 A presentation with unilateral headache, with the usual associations of migraine, before symptoms attributed to the thoracic outlet syndrome, was reported in 21 out of 30 patients; eleven of their patients still required vasoactive drugs after transaxillary first rib resection.38 In no case was improvement in headache not associated with improvement in the other symptoms. White and Sweet reported a woman with left hemicranial migraine who had anterior scalenectomy, resection of left cervical rib, procaine block of C2-C3-C4 and carotid bifurcation, and infiltration of the scalenectomy scar. She also had division of the cervical sympathetic trunk but "right sided pain and attacks of incapacitating migraine ten days per month continued.33 Further, in the absence of neurological signs, the clinical criteria for the diagnosis of the "thoracic outlet syndrome" are controversial.39,40 The location of the pain and its aggravation by certain postures or exertion38 can also be seen in other conditions. Obliteration of the pulse by certain manoeuvres may occur in healthy subjects.40,41 Swelling of the face15,42,43 Or limbs5,44 have been reported in cluster headache and hemiplegic migraine respectively. The presence of circulatory changes in the skin of patients with a diagnosis of "thoracic outlet syndrome" may not be a specific finding either Raynaud phenomena and migraine had both a similarly significantly higher incidence in patients with variant angina leading the authors to propose the existence of a generalised vasospastic disorder in their patients.45 In 2 of 3 patients with migraine and unilateral minor swelling and/or bluish skin patches in one upper limb in one series, limb pain alternated sides.2 CONCLUSION
The existence of limb pain as a migrainous accompaniment, associated with both migraine and cluster headache, is supported by many clinical descriptions since Liveing referred to it in 1873. The symptom was identified as part of sphenopalatine ganglion neuralgia or lower half headache, a syndrome claimed by Sluder to be different from migraine, but which included a heterogenous group of patients; attention was then focused on the mechanisms of pain rather than on the clinical features of limb pain and headache. Although some of the patients described by Sluder were subsequently acccepted as cases of cluster headache, limb pain as a migrainous symptom has not been widely recognised and references to it in the recent literature are sporadic. It is possible that many patients with migrainous limb pain are assumed to have other causes for this pain, including psycho-genic pain, cervical and/or lumbar radiculopathy or the thoracic outlet syndrome. REFERENCES
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