ANKYLOSIS OF THE FINGER JOINTS IN RHEUMATOID · that ankylosis with stout bony fusion had occurred...

Ann. rheum. Dis. (1958), 17, 365.

ANKYLOSIS OF THE FINGER JOINTS INRHEUMATOID ARTHRITIS

BY

ROBERT M. STECHERFrom the Department of Medicine of Western Reserve Medical School, City Hospital, Cleveland, Ohio

The ordinary manifestations of rheumatoidarthritis are well known and readily recognized.They are subject to wide variations and, as theydeviate farther from the conventional picture,diagnosis becomes difficult, doubtful, or evenimpossible. The aetiology is unknown and proofof diagnosis is lacking. Even the proposed diagnosticcriteria for rheumatoid arthritis (Ropes, Bennett,Cobb, Jacox, and Jessar, 1957) have not completelyeliminated the difficulty, although they have madegreater uniformity of classification possible. Undersuch circumstances, doubtful cases conforming tosome diagnostic criteria but not to others are ofconsiderable interest and often worthy of detailedstudy. Bony ankylosis of joints is a characteristicresult of rheumatoid arthritis in a small proportionof cases, but it is non-specific for the disease. Severalcases in which ankylosis of the finger joints hasoccurred or has been an outstanding feature havebeen observed which seem worthy of further atten-tion and will be described here. In two such cases,ankylosis of the interphalangeal joints of the fingerswere observed in long-standing, generalized, andseverely crippling rheumatoid arthritis. These

cases are described briefly for comparison. Twoother cases, however, have been followed for severalyears, one through a period of soft tissue inflam-mation and bone destruction, progressing intoankylosis. The trouble with the fingers has beenthe outstanding complaint. No other joints havebeen involved, general health has not been impaired,and since the inflammation has subsided, immobilityof the fingers has been the only complaint.

Case ReportsCase 1, a woman aged 52, entered the City Hospital in

1933. Her hospital record has been lost so that theclinical story is not available in detail. She had then hadgeneralized rheumatoid arthritis for some years and wasbedridden and completely handicapped. According todiagnostic criteria she was classified as a case of definiterheumatoid arthritis. According to the therapeuticcriteria adopted by the New York Rheumatism Asso-ciation and the American Rheumatism Association(Steinbrocker, Traeger, and Batterman, 1949), she wasclassified as "Stage IV Class IV", because of osteo-porosis, cartilage and bone destruction, muscle atrophy,ulnar deviation, and bony ankylosis.The hands show little deformity (Fig. 1). The wrists

Fig. 1.-Case 1, hands,showing slight swelling ofwrists, ulnar deviation offingers of right hand, andflexion of right forefingerand left little finger. Lossof transverse creases of theskin of the backs of thefingers is noticeable. Thefinger nails are normal.

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ANNALS OF THE RHEUMATIC DISEASESseem to be slightly swollen, and there is obvious ulnardeviation of the fingers of the right hand, the fingers arestraight except for slight flexion of the right forefingerand the left little finger, and the skin of the fingers of theright hand is particularly smooth with loss of the normaltransverse creases over the proximal joints. Thesecreases have completely disappeared over the distaljoints and the fingers here seen to be constricted. Thenails are normal.

Radiographs of the hands (Fig. 2) show markeddemineralization of the bones, particularly around the

wrists and metacarpophalangeal and interphalangealjoints of both hands. The metacarpal bones of thewrists have lost their individual outlines and have beenfused into one bony mass. The joint spaces betweenthem and the carpal bones have completely disappearedand are diminished between them and the bones of thelower arm. All the metacarpophalangeal joints showsubluxation with destruction of the proximal phalanges.The fingers of the right hand show ulnar deviation. Allthe interphalangeal joints, save only the distal joint of theleft little finger, show complete bony ankylosis.

Fig. 2.-Case 1, postero-anterior radiographs, showing bony ankylosis of all finger joints and subluxation of the metacarpophalangealjoints. The bones of the wrist are fused and ankylosed to the radius and ulna and metacarpal bones in both hands. There is also

generalized demineralization.

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ANKYLOSIS OF THE FINGER JOINTS IN RHEUMATOID ARTHRITISThere was also demineralization and severe loss of

joint space of the right knee and condensation of bone andloss ol joint spaces in both hips.

This woman had had severe generalized rheumatoidarthritis for many years resulting in complete disabilityand invalidism. One outstanding feature of the diseasein her case was ankylosis of all finger joints.

Case 2, a woman aged 42, was an in-patient from Aprilto December, 1936, because of severe generalizedrheumatoid arthritis of long duration. The disease hadbegun 8 years before with arthritis of the feet, and thishad spread to involve the hands, shoulders, knees, andelbows. The pain at times was severe and stiffnessdeveloped. She became so disabled that she could notwalk and had been confined to a wheel chair for the last7 years. On admission there was marked limitation ofmotion of nearly all of her joints and complete stiffness

of all of the proximal joints. She was classified as a caseof definite rheumatoid arthritis (Stage IV, Class IV).Attempts to correct her flexion deformities were un-successful and she was discharged.The hands showed some enlargement of the wrists.

There was little abnormality in the hands except that theskin over the fingers was smooth, all of the normaltransverse creases over all the finger joints having beenlost. The finger nails were normal.

Radiographs of both hands (Fig. 3) 8 years after theonset show demineralization of bones particularlymarked in the phalanges. There is bone absorption ofthe carpal bones, worse in the right wrist, with fusionto each other, the metacarpal bones, and the lower armbones. Subluxation is seen in most of the metacarpo-phalangeal joints. All the interphalangeal joints, exceptthe joints of the right index and middle fingers, are theseat of bony ankylosis.

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Fig. 3.-Case 2, postero-anterior radiographs of both hands 8 years after onset, showing demineralization of bones which is par-ticularly marked in the phalanges. Absorption of the carpal bones is seen, which is more marked in the right wrist with fusion to

each other, to the metacarpal bones, and to the radius and ulna. Most of the interphalangeal joints show bony ankylosis.

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38ANNALS OF THE RHEUMATIC DISEASESThis woman had had severe generalized rheumatoid

arthritis for 8 years, resulting in complete disability andinvalidism. One outstanding feature of the disease inher case was ankylosis of nearly all finger joints.

Case 3, a woman aged 50, was first seen in March,1947. Her disease had started 11 years before, at theage of 39, with swelling and pain in her hands. Limita-tion of motion in the fingers had been noted shortlyafterwards and this had progressed so that in 1945 theyhad become stiffened, but after this the pain and swellinghad disappeared. Pains in the feet had begun in 1940,7 years before her first visit to hospital. They hadpersisted and increased in severity and had been asso-ciated at times with tenderness and swelling. For severalyears she had had pain in the right wrist, shoulders,hips, and knees. Her general health was fairly good butshe slept poorly. Despite a poor appetite she had gained15 lb. in 3 years. She kept her house warm, felt worsein rainy and damp weather, and had frequent chilly andnervous spells. She had be-n treated with innumerablehip shots, short-wave diathermy treatments, high colonicirrigations, and aspirin, and repeatedly told that she had

arthritis. Menses became irregular in 1945, at the ageof 47.

Physical examination was negative except for thefingers. They were slightly flexed in the right hand andthe joints were obviously ankylosed in the proximaljoints. In the left hand motion was present but restricted.All the other joints seemed normal.

She was next seen in January, 1948, with dermatitismedicamentosis, which cleared up after 4 months oftreatment. At this time the erythrocyte sedimentationrate was 28 mm. Hg/hr. She was next seen at her ownhome in June, 1952, when she said that the pain hadgradually become worse so that she had finally becomebedridden. She went on a complete fast for 21 days,then ate salads and fruits for several weeks and againfasted for 27 days. She lost 40 lb. in weight during thistime, and the joint swelling and pain disappeared and shefelt much better. The condition of her fingers was thesame, however, except that the entire left little fingerhad become completely stiffened.

She was free of all complaints and her physical exami-nation was negative except for her joints when she wasseen in 1957 and 1958. In 1958 when she clenched her

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Fig. 4.-Case 3, radiographs of both hands in 1947, Il years after onset, showing complete ankylosis of all proximal and fifthfinger distal joints of the right hand. In the left hand the distal joint of the little finger is ankylosed, the proximal joints show bone

destruction and alteration of joint surfaces, which is most marked in the fifth finger. Other hones and joints are normal.

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ANKYLOSIS OF THE FINGER JOINTS IN RHEUMATOID ARTHRITIS

fists, the metacarpophalangeal joints flexed at right-angles, but the proximal interphalangeal joints remainedextended in the right hand and showed very little flexionin the left. Radiographs of the right shoulder, elbow,knee, ankle, and feet, and of the pelvis including the hipswere normal.

Clinical laboratory investigations were made in 1947,1948, 1953, 1957, and 1958. The red blood count was3,360,000 cells per cu. mm. in 1957, but was otherwisenormal, as was the haemoglobin content, white bloodcell count, and serum uric acid. The erythrocyte sedi-mentation rate varied from 28 to 47 mm. Hg/hr by amodified Westergren method. Serological tests inSeptember, 1957, showed latex-fixation test positive in1/320 dilution, Heller F II test positive 1/7,000 dilu-tion, and Waaler-Rose fixation test positive 1/1,024dilution. Similar results were obtained in January, 1958.

This patient was considered to have rheumatoidarthritis Class IV, Stage I.

Fig. 4 shows an antero-posterior radiograph of bothhands taken in 1947. Both wrists and all the metacarpo-

phalangeal joints are normal. In the right hand, theproximal interphalangeal joints of the fingers and thedistal joint of the little finger are ankylosed. The distaljoints of the other three fingers and the thumb are normal.In the left hand the distal joint of the little finger isankylosed, and the proximal joint is abnormal in that thejoint surfaces are irregular and saw-toothed because ofirregular bone destruction. The other joints have aslightly similar but not nearly so advanced appearance.Radiographs in 1953 and 1957 showed no change exceptthat ankylosis with stout bony fusion had occurred by1953 in the proximal joint of the left little finger.

Fig. 5 shows lateral radiographs of each finger takenin 1953. Complete fusion is seen of all joints in bothlittle fingers and of the proximal joints of the fingers ofthe right hand. All signs of joint lines or joint spaceshave been obliterated. The proximal joints of the threefingers of the left hand showed a marked antero-posteriorenlargement of the proximal ends of the middle phalanges,giving a cup-shaped appearance of the joint surface withrounded spurs. The distal joints of the first three

Left RightFig. 5.-Case 3, lateral radiographs of the fingers 17 years after onset showing complete fusion of all the joints in both little fingersand in the proximal joints of the other three fingers of the right hand. The proximal joints of the first three fingers of the left handshow marked antero-posterior enlargement of the proximal ends of the middle phalanges. Spurs are seen on the posterior surface

of the distal joints of the first three fingers of each hand similar to those seen in Heberden's nodes.

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370~~ANNALSOF THE RHEUMATIC DISEASESfingers all show marked antero-posterior enlargementsarising from the dorsal aspect of the proximal end ofeach of the distal phalanges which resemble those seenin Heberden's nodes (Stecher and Hauser, 1948, 1954).Both thumbs are normal.

Radiographs taken in September, 1957, showed noalteration. Radiographs of both feet, right ankle,right knee, both hips and pelvis, right shoulder, rightelbow, both wrists, and all metacarpophalangeal jointsshowed no sign of arthritic disease or other abnormality.Radiographs of the chest show normal heart and lungs.Although the distribution of the joint disease is

unusual and the lack of constitutional symptoms issurprising, this patient is considered to be a case of rheu-matoid arthritis, because of the ankylosis, the consis-tently elevated sedimentation rate, and the positiveagglutination tests. Her disease is now in completeclinical remission.

Case 4, a woman aged 56, was seen in 1949 because ofarthritis. She had been completely well until 3 yearsbefore when she had noted swelling and soreness of thefirst two fingers of the right hand. Radiographs taken2 years later had shown fusiform soft tissue swelling ofthe right index and middle fingers, loss of joint space,and irregularity with destruction of the bone ends. Slightchanges were apparent in the ring finger. All the otherjoints in the fingers and wrists of both hands were normal.The process had progressed slowly involving otherfingers, until all the proximal interphalangeal joints hadbecome enlarged, tender, and partially stiffened. Thepatient had at different times complained of sore hands,neck, elbows, shoulders, and a toe, but no enlargement,deformity, or dysfunction had developed. Her generalhealth was good. She had lost no weight. She had had

much conventional therapy, besides lamp treatments,bee-sting therapy, and hot baths at Hot Springs,Arkansas, and had spent 3 months in Florida withoutrelief. Her family history was negative, in that herparents, five brothers, and sisters had had no jointdisease of any kind.

Physical examination was negative except for thefingers, which showed fusiform enlargement of all of theinterphalangeal joints including the thumbs. The skinwas smooth and shiny, most of the normal wrinklingbeing decreased or absent over the backs of the distaljoints and the proximal joints of the left little and ringfingers and the right index and little fingers and boththumbs. The appearance of the other joints and the skinover the metacarpophalangeal joints and the wrists wascompletely normal.The diagnosis at this time was doubtful. Despite the

typical appearance of rheumatoid arthritis of the fingers,the diagnosis of osteo-arthritis of the fingers was madebecause of absence of other joint disease 3 years afteronset and because of her normal health and normallaboratory findings.The patient was next seen in December, 1951, when

she was still complaining of stiff and painful wrists, hands,and fingers. Since her previous visit, she had haddiathermy treatments and paraffin baths for one yearwithout relief. This was followed by cortisone for20 days, which gave her relief from pain in the wristsand neck, improved her appetite and digestion, and gaveher a feeling of well-being, but oedema developed.Acetysalicylic acid and codeine had upset her stomachand had to be discontinued. Gold therapy was sug-gested but not used. At this visit she was emotionallydisturbed, easily upset, wept readily, and complained offrequent headaches. These symptoms were attributed

Fig. 6.-Case 4, 3 years afteronset; the hands appear

A normal except for fusiformenlargement of the proximaljonsof the fingers and lossof transverse creases of the

skin the terminal joints

ofthe little fingers andthumbs.

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largely to the illness and disability of her husband.Physical examination at this time was negative exceptfor complete ankylosis of all proximal interphalangealjoints.

Little change in her condition was noted on visitsin 1952, 1955, and 1957. Cortisone had been startedagain but this was stopped in 1953 because she haddeveloped a moon face and buffalo hump. She thenhad 36 gold shots without effect, and since 1953 she hashad little or no therapy. Her spirits, her appetite, andher general outlook on life have improved, and despiteher bedridden husband whom she cares for at home,she seems happy, contented, and well, with no com-plaints except a stiffness of the fingers which does notbother her.

Physical examination in 1955 and again in 1958revealed no abnormality whatsoever in the spine,shoulders, elbows, hips, knees, ankles, or feet. Motionof both wrists were limited to about half the normalrange; the interphalangeal joints were fixed, of course,but the other joints of the hand functioned normally.

Laboratory investigations carried out in 1949, 1951,1952, 1955, 1957, and 1958, showed normal red bloodcell count, white blood cell count, haemoglobin level,and haematocrit. The red blood cell sedimentation ratewas 21, 22, and 26 mm. Hg/hr (corrected) until 1952.In 1955 and 1958 the rate was 9 and 3 mm. respectively.

The latex-fixation test, Heller F II test, and Waaler-Rose test were negative on May 21, 1957, and January 20,1958.

If the diagnosis of rheumatoid arthritis is accepted inthis case, it can be considered as Class IV, Stage I. Thediagnosis is very doubtful, however, because the erythro-cyte sedimentation rate has been normal or only slightlyelevated, the serological agglutination tests have beennegative, and no joint changes have been recognizedexcept those of the hands and the wrists. Except forthe ankylosis the joints have not been typical of rheuma-toid arthritis. The patient's subjective symptoms can beaccounted for by her personal problems.The progress of the disease in this case can best be

followed by examination of photographs and radio-graphs. Photographs of the hands taken on the firstvisit in 1949, 3 years after onset (Fig. 6), show fusi-form enlargements of the fingers as described above.Radiographs of both hands (first taken in 1948, 2 yearsafter onset) showed, besides demineralization, softtissue swelling about the proximal interphalangeal joints,decrease in joint space, and destruction of the jointsurfaces of the index and middle fingers of the right hand.Radiographs of both hands repeated in May, 1949,(Fig. 7) show extension of the process. The proximalends of the middle interphalangeal joints are broadened,roughened, and eroded, indicating loss of bone substance.

IFig. 7. Case 4. radiogtaphs tboth hands veafter onset. showing that the 'hape of the bone', andthe appearance of the joint. .are completely' normal.except the proxritmal interphalangeal joint- Lind thedistal joint of' the elet little finger. Thee Showsdecrease in joint space and irregular ity of joint sur-tlace faith broadening. rouLghening. and crro)ion of the

proximal ends o f the middle phalanges.

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ANNALS OF THE RHEUMATIC DISEASES1il tI| rt ICC 'O ti zL L Itiiit JI IIlkI l

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difficulties in making the photographic prints.Thus, despite a destructive joint disease resulting in

a complete bony ankylosis studied for 10 years, the diag-nosis of rheumatoid arthritis is doubtful because of thesharp localization of the disease, the absence of con-stitutional symptoms, the only slightly elevated erythro-cyte sedimentation rate, and the negative serologicaltests.

Discussion

Case histories, clinical records, and iconographicdescriptions are given of four women, each of whomsuffered from ankylosis of the interphalangealjoints of the fingers. The first two are cases ofsevere generalized rheumatoid arthritis. Thesepatients had disease of long standing, with wide-

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spread joint damage throughout the body, andcomplete disablement. The patients were placedin Class IV, Stage IV, according to the classificationadopted by the American Rheumatism Association.The other two cases differ from the first two in thatpermanent joint damage has been limited to thehands and constitutional symptoms, which werenever severe, have completely disappeared. Thepatients seem to be in excellent health and onlyslightly handicapped by the changes in the hands.The third case has been classified as Class IV,Stage I, solely because some joints show completeankylosis. Radiographic studies of the fingersshow progression of the disease from a stage offusiform enlargement and bone destruction aboutthe proximal interphalangeal joints to the presentcondition when the bones are completely ankylosed

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Fig. 9.-Case 4, lateral views of the fingers 7 years after onset, showing bony ankylosis of all the proximal joints and ofthe distal joints of the right little finger and thumb. Spurs are seen in the dorsal aspects of the proximal ends of the

distal phalanges similar to those seen in Heberden's nodes.

and the fingers have regained their normal size.No other joint changes in the body are recognizableby clinical examination or by extensive radiographicsurvey. The diagnosis in this case is substantiatedby positive serological tests on two occasions andan elevated erythrocyte sedimentation rate. Adefinite diagnosis is justified according to the pro-posed diagnostic criteria for rheumatoid arthritis,since there was at one time pain and tenderness,swelling, symmetrical involvement, radiographicchanges, and a positive latex agglutination test.Despite the ankylosis, clinical activity is nowminimal; the unusual aspects of the case are thevery restricted distribution of joint involvement,the temporary and mild constitutional symptoms,and the complete restoration of health and activityfor several years. If this patient were seen now andnot carefully studied the diagnosis might easily beoverlooked.

The fourth case is similar to the previous one inthat the disease has been limited to the hands, andthat constitutional symptoms have been mild andhave now completely disappeared. In this patientthe erythrocyte sedimentation rate has neverexceeded 26 mm. Hg/hr, and for the last 3 years hasbeen below 10 mm. The agglutination tests havebeen completely negative twice in the last year.A definite diagnosis is not so clearly justifiedaccording to the proposed diagnostic criteria.Symmetrical involvement, radiographic changes,and stiffness in symmetrical joints have been noted,but because she was not observed, during the earlystages, details of her symptoms at that time are notknown. The patient has been classified as Class IVbecause of bony ankylosis, but has been placed inthe Stage I category because of her complete abilityto carry on all her usual duties without handicap.She must be considered as at least a possible case

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ANNALS OF THE RHEUMATIC DISEASES

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Fig. 10.-Case 4, radiograph 12 years after onset showing complete bony ankylosis of the proximal interphalangeal joints of bothhands and of the distal joints of the right little finger and thumb. The metacarpophalangeal joint of the left little finger shows a

cup-shaped deformity of the phalanx. Wrists show decrease in size of the proximal carpal bones.

of rheumatoid arthritis if not a probable case, butthe diagnosis cannot be considered as proven.Although discussion has been limited to a con-

sideration of rheumatoid arthritis in these cases,

there are certain additional features suggestive ofdegenerative joint disease. Lateral views of thefingers (Figs 5 and 9) show large spurs projectingdorsally from the proximal ends of the distalphalanges. These spurs are thick and have roundedends suggestive of traumatic Heberden's nodes.It is unusual, however, to see six instances of trau-matic Heberden's nodes in one individual as inFig. 5. Idiopathic Heberden's nodes are usuallyassociated with loss of joint space, greater irregu-larity of joint surface, and pointed spurs. Heber-den's nodes with spurs as large as those shown hereare always associated with considerable enlargementof the fingers, but these fingers were not enlargedin the region of the terminal joints. The carpalbones in Fig. 10 show changes in outline with loss

of substance particularly of the semi-lunar bones.The joint spaces are adequately preserved and thejoint surfaces are sharp and show condensation ofbone. The rest of the carpal bones are not altered.These changes are difficult to classify because theyseem to differ sharply from those usually seen in thearthritic diseases. Bone seems to have been ab-sorbed, but there is no inflammation and the causa-tive process seems to be completely quiescent andhealed. Even if the changes in the terminal fingerjoints and the wrists are accepted as manifestationsof osteo-arthritis, the limited distribution of thesechanges do not justify the characterization of thecondition as generalized osteo-arthritis as describedby Kellgren and Moore (1952).

Summary

Ankylosis of the phalanges without deformity isuncommon even in severe cases of rheumatoid

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arthritis. Four female patients who suffered fromankylosis of the interphalangeal joints of thefingers are described in detail.Two had severe generalized rheumatoid arthritis

leading to complete disablement, but in the othertwo permanent joint damage was limited to thehands and the mild systemic symptoms disappeared,leaving little if any disability.

REFERENCESKeligren, J. H., and Moore, R. (1952). Brit. med. J., 1, 181.Ropes, M. W., Bennett, G. A., Cobb, S., Jacox, R., and Jessar, R. A.

(1957). Ann. rheum. Dis., 16, 118.Stecher, R. M., and Hauser, H. (1948). Amer. J. Roentgenol.,

59, 326.

- (1954). Ibid.. 72, 452.Steinbrocker, O., Traeger, C. H., and Batterman, R. C. (1949).

J. Amer. med. Ass., 140, 659.

Ankylose des articulation digitales dans l'arthriterhumatismale

RESUME

L'ankylose des phalanges sans deformation es peucommune meme dans des cas severes d'arthrite rhuma-

tismale. On decrit en detail quatre cas de femmesatteintes d'ankylose des articulations interphalangiennesdes doigts.Deux d'entre elles souffraient d'une arthrite rhuma-

tismale generalisee et sev&re menant A l'incapacite totale,mais chez les deux autres le dommage se limitait auxmains, les sympt6mes generaux ayant disparu avec peuou pas d'incapacite residuelle.

Anquilosis de las articulaciones digitales en la artritisreumatoide

SUMARIOLa anquilosis de las falanges sin deformidad no es

comun hasta en los casos graves de artritis reumatoide.Se describen detalladamente cuatro casos de mujeres conanquilosis de las articulaciones interfalangeas de losdedos.Dos de estas sufrieron de una artritis reumatoide

generalizada y grave conduciendo a la incapacidad total,pero en las demas el dano se limit a las manos, lossintomas generates habiendo desaparecido con incapa-cidad residual poca o ninguna.

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ANKYLOSIS OF THE FINGER JOINTS IN RHEUMATOID · that ankylosis with stout bony fusion had occurred...

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