Ann. rheum. Dis. (1962), 21, 199.

A LATE EFFECT ON THE CERVICAL SPINE INANKYLOSING SPONDYLITIS

BY

WILLIAM MARTEL, JOHN F. HOLT, AND WILLIAM D. ROBINSONFrom the Departments of Radiology and Internal Medicine (Rackham Arthritis Research Unit),

University of Michigan, Ann Arbor, Mich.

Adults with advanced ankylosing spondylitis willoccasionally exhibit a dramatic resorption of bonefrom the anterior surface of the cervical spine. Thismay be confused with the vertebral growth dis-turbances of juvenile rheumatoid arthritis and anerroneous inference may be drawn that in thesecases ankylosing spondylitis must necessarily havebegun in childhood (Wilkinson and Bywaters, 1958).It seems pertinent to call attention to this finding toemphasize that it may be acquired in adulthood andis not a "growth disturbance".

Case ReportsCase 1, a 43-year-old male, first noted stiffness and

pain in the back with radiation down both thighs in 1939.These symptoms gradually progressed and he developedarthritis of both hips for which he was admitted to theUniversity Hospital in 1944. Physical examinationshowed moderate rigidity and kyphosis of the dorso-lumbar spine and limitation of motion in both hips.Chest expansion was reduced. Neck motion was

normal. There were no rheumatoid nodules. Radio-logical examination disclosed ankylosis of both sacro-iliac joints and dorsolumbar spondylitis with "squaring"of the vertebral bodies, paraspinal ossifications, andfusion of the apophyseal joints. The hips were involved,but the cervical spine was normal (Fig. la). A diagnosisof ankylosing spondylitis was made and radiation therapygave some relief of symptoms.He was seen again in 1949 for recurrent episodes of

faintness which were not relieved by food and wereunrelated to posture. Physical examination now showedextreme kyphosis and spinal rigidity. Neck motion waspartially limited. Neurological examination was nega-tive. Cervical spine films (Fig. lb) showed a dramaticresorption of bone from the anterior surfaces of C4,5, and 6, so that the antero-posterior diameters of thesevertebral bodies were reduced. The anterior margin ofthe cervical spine was continuous and smooth, as thoughit had been "planed down". The apophyseal joints seenin the lateral projection were fused, the intervertebraldisks were reduced in height, and many were partiallycalcified, and the bones were demineralized. The causeof these symptoms was uncertain. The results of an oral

(ci) (b) (C)Fig. .--Case 1. (a) 1944 at age 27. Normal cervical spine. (b) 1949. (c) 1954. Extensive, progressive resorption of bone from theanterior surface of C4, 5, and 6. The vertical dimensions of these vertebral bodies are preserved. Note apophyseal ankylosis and atrophy of'

intervertebral disks.

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ANNALS OF THE RHEUMATIC DISEASESglucose tolerance test suggested functional hypo-glycaemia, but the symptoms continued despite a highcarbohydrate diet. He was again seen in 1954 becauseof an exacerbation of the arthritis. Cervical spine films(Fig. ic) at this time showed further resorption of bonefrom the anterior aspect of the cervical spine. Filmswere again obtained in 1960 and these showed no signifi-cant change, though flexion and extension views demon-strated fair motion in the atlanto-occipital and atlanto-axial joints.

Case 2, a 44-year-old male, first developed progressivepain and stiffness of the lumbar spine at the age of 20years. Arthritis of the hips and shoulders began 5 yearslater. When the patient was first seen at the UniversityHospital in 1944, he had marked limitation of motionof the entire spine, including the cervical region. Therewas marked kyphosis of the dorsolumbar spine, and chestexpansion was diminished. There was arthritis of thehips, shoulders, knees, and several joints of the righthand and left foot. Neurological examination wasnegative. Radiological examination showed typical,far-advanced changes of ankylosing spondylitis withextensive paraspinal ossifications, "squaring", andankylosis of the sacro-iliac, apophyseal, and costo-vertebral joints. Cervical spine films were not obtained.

In 1954 x-ray films showed progression of these changesand severe kyphosis of the dorsolumbar spine. Thecervical apophyseal joints were completely ankylosedand there was resorption of bone from the anteriorsurface of C6 and C7 and TI (Fig. 2). Extensiveligamentous ossification was present. The intervertebraldisks were atrophic and partially calcified and thevertebrae were demineralized.

Fig. 2.-Case 2. 1954 at age 38. Resorption of bone similar to thatshown in Fig. 1 has taken place at C6 and 7 and TI. Advanced

ankylosing spondylitis of the entire spine was present.

CommentSuperficial erosions of the anterior surfaces of the

vertebrae, so-called "anterior spondylitis", are wellknown in ankylosing spondylitis and account forthe characteristic "squaring" of the vertebral bodies(Romanus and Yden, 1955). These erosions arefrequent in the dorsolumbar region, but may alsobe observed in the cervical spine. The vertebralchanges which are illustrated are remarkable in thatthe loss of bone substance was so extreme andoccurred predominantly in the cervical region. Wehave noted this now in several patients with advancedankylosing spondylitis. It seems to be a late mani-festation associated with complete ankylosis andextreme kyphosis of the dorsolumbar spine, suchthat the cervicodorsal segments tend to be horizontalwith the patient in the upright position. Continualhyperextension of the neck becomes necessary topermit the patient to look forward rather than down.One may postulate that, as a result of this continualstress, a physiological resorption of bone occurs atthe anterior, convex surface of the cervical arch byWolff's law of transformation (Weinmann andSicher, 1955). It is likely that this effect is super-imposed on superficial erosions in this region, andthereby accounts for this degree of bone loss. Theparticular segments which undergo this transforma-tion vary and this probably depends on the precedingpattern of ankylosis.

Apophyseal ankylosis is a common sequel to thespondylitis of juvenile rheumatoid arthritis and,characteristically, the adjacent vertebrae remainsmall in all diameters in this disease and the inter-vertebral disks atrophy and may calcify (Martel,Holt, and Cassidy, 1961). These changes arepresumably due to growth disturbance and toatrophy secondary to non-use. This appearancemay be distinguished from that noted above inwhich the antero-posterior diameters of the vertebralbodies are reduced. While ankylosing spondylitismay occasionally begin in childhood, it is probablyquite uncommon, particularly before puberty.

SmryExtensive resorption of bone from the anterior

surface of the cervical spine is not uncommon inankylosing spondylitis and is not to be confusedwith the growth disturbances seen in juvenilerheumatoid arthritis. Two examples of this defor-mity are illustrated. The radiological findings inone case clearly show that the onset occurred inadulthood, after the spine had reached full maturity.Furthermore, one may expect a generalized reduc-tion in the size of the cervical vertebral bodies in

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LATE EFFECT ON THE CERVICAL SPINE IN ANKYLOSING SPONDYLITIS 201

juvenile rheumatoid arthritis, whereas in these adultcases the antero-posterior diameters are primarilyaffected.

REFERENCESMartel, W., Holt, J. F., and Cassidy, J. T. (1961).

"Radiologic Manifestations of Juvenile Rheuma-toid Arthritis." Presented at the 62nd annualmeeting of the American Roentgen Ray Societyon September 27, 1961.

Romanus, R., and Yden, S. (1955). "Pelvo-spondylitisOssificans." Munksgaard, Copenhagen.

Weinmann, J. P., and Sicher, H. (1955). "Bone andBones. Fundamentals of Bone Biology," 2nd ed.,p. 134. Mosby, St. Louis.

Wilkinson, M., and Bywaters, E. G. L. (1958). Ann.rheum. Dis., 17, 209.

Effets eloignes sur la colonne cervicale dans laspondylarthrite ankylosante

RisuMfUne resorption osseuse prononcee de la face anterieure

de la colonne cervicale n'est pas rare dans la spondyl-

arthrite ankylosante, et on ne doit pas la confondre avecdes desordres de croissance observes dans l'arthriterhumatismale juvenile. On decrit deux exemples decette deformation. Les images radiologiques dans uncas montrent clairement que la lesion avait debut6 al'age adulte, apres que la colonne ait atteint sa maturity.Chez ces adultes, c'est le diametre antero-posterieur quiest surtout atteint, alors que, s'il s'etait agi d'une anciennearthrite rhumatismale juvenile, on aurait observe unediminution totale de volume des corps vertebraux.

Efectos remotos sobre la columna cervical en laespondilartritis anquilosante

SUMARIOMarcada reabsorci6n de hueso en la superficie anterior

de la columna cervical no es infrecuente en la espondil-artritis anquilosante, y no se debe confundir con distur-bios de crecimiento vistos en la artritis reumatoidejuvenil. Se describen dos ejemplos de esta deformidad.Los hallazgos radiol6gicos en un caso muestran clara-mente que las alteraciones empezaron en la edad adulta,despues que la columna habia Ilegado a su pleno desar-rollo. En lugar de la anticipada reduccion generalizadaen el tamano de los cuerpos vertebrates en la artritisreumatoide juvenil, en estos casos adultos los diametrosantero-posteriores se ven primeramente afectados.

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ANNALS OF THE RHEUMATIC DISEASESProphylaxis of Occupational Rheumatic Lesions with

Exercise Apparatus. (Zur Prophylaxe rheumatischerArbeitsschaden am Bewegungsapparat.) SCHOGER,G. A. (1962). Z. Rheumaforsch., 21, 55.

Experimental Rheumatism induced with Vinidicol andHydralazine. (Artritis experimentales por el Vincidoly la Hidralazina.) PEREZ GIL, L. (1961). Rev. esp.Reum., 9, 156. 22 refs.

Two Cases of Sjogren's Syndrome in Rome and theirTherapy by means of Resochin. (Zwei Falle vonSjogrensyndrom und deren therapeutische Beeinflus-sung durch Resochin.) STOYANOV, P. K. (1961).Med. Kin., 56, 957. 7 refs.

Rehabilitation of the Rheumatic Patient. (A Reabilitagaodo reumAtico.) KLEIN, B., and HOULI, J. (1961).Brasil-mid., 75, 314.

CORRIGENDUM

It is regretted that in the paper entitled "A Late Effect on the Cervical Spine in Ankylosing Spondylitis", by W.Martel, J. F. Holt, and W. D. Robinson, which appeared in the June, 1962, issue of the Annals, the three blocks forFig. 1 were printed upside down. The correct version is shown below:

Fig. 1.-Case 1. (a) 1944 at age 27. Normal cervical spine. (b) 1949. (c) 1954. Extensive, progressive resorption of bone from theanterior surface of C4, 5, and 6. The vertical dimensions of these vertebral bodies are preserved. Note apophyseal ankylosis and atrophy of

intervertebral disks.

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