[4] Wiesel BS, Ignatius P, Marvel JP, Rothman RH. Intradural neurofi-broma simulating lumbar disc disease. The Journal Bone and JointSurgery 1976;58A:1040–2.

[5] Mathew P, Todd NV. Intradural conus and cauda equina tumours: aretrospective review of presentation, diagnosis and early outcome.J Neurol Neurosurg Psychiatry 1993;56:69–74.

[6] Idir ABC, Doyon D, Husson B, Hurth M. Pathologie tumorale de lamoelle et de ses enveloppes chez l’adulte et chez l’enfant. Éditionstechniques—Encycl. Méd. Chir. (Paris, France); Radiodiagnostic—Neurologie-Appareil locomoteur 1991; 31673(A20):12.

[7] Komatsu Y, Narushima K, Kobayashi E, Ebihara R. Small caudaequina neurinoma detected by MR Imaging. AJNR 1988;9:1243.

[8] Narvaez J, Alegre-Sancho JJ, Clavaguera MT, Juanola X, Roig-Escofet D. Cauda equina tumour presenting as atypical sciatica.Br J Rheumatol 1997;36:605–6.

Latifa Harzallah *Medical Imaging Department,

Farhat Hached Hospital, Sousse, TunisiaE-mail address: latifa_harzalla@yahoo.fr (L. Harzallah).

Elyès BouajinaMehdi Ghanouchi

Rheumatology Department,Farhat Hached Hospital, Sousse, Tunisia

Habib AmaraLamia Ben Chérifa

Ch. KraiemMedical Imaging Department,

Farhat Hached Hospital, Sousse, Tunisia

Received 20 April 2004; accepted 23 September 2004

Available online 08 December 2004

* Corresponding author. Medical ImagingDepartment, Hôpital Farhat Hached, 4000 Sousse, Tunisia.Tel.: +216-73-22-14-11; fax: +216-73-22-67-02.

1297-319X/$ - see front matter © 2004 Elsevier SAS. All rights reserved.doi:10.1016/j.jbspin.2004.09.007

Chronic polyarthritis as the presenting manifestationof acute leukemia

Keywords: Acute leukemia; Polyarthritis

1. Introduction

Only 4% of adults with acute leukemia present with mus-culoskeletal manifestations [1,2]. We report a highly unusualcase of acute lymphoblastic leukemia in an adult with pol-yarthritis as the inaugural manifestation.

2. Observation

In this 22-year-old woman with an unremarkable medicalhistory, bilateral symmetric polyarthritis developed gradu-

ally, starting in November 2001. The wrists, metacarpopha-langeal joints, proximal interphalangeal joints, elbows, shoul-ders, ankles, and temporomandibular joints were affected.Nonsteroidal antiinflammatory drugs provided no relief. InJune 2002, swelling of the right parotid gland with a feverand a decline in general health developed, and she was admit-ted.

At admission, body temperature was normal and bodyweight was 50 kg with a height of 1 m 64 cm. Symmetricsynovitis was noted, involving the wrists, metacarpopha-langeal joints, proximal interphalangeal joints, elbows, andankles was noted. Pressure on the sternum elicited pain. Theright parotid gland, cervical lymph nodes, and spleen wereenlarged, whereas the liver was normal to palpation. Labora-tory tests showed marked inflammation (erythrocyte sedimen-tation rate, 85 mm/h; and C-reactive protein, 96 mg/l). Theperipheral leukocyte count was increased to 21,200/mm3 with82% of lymphocytes but no immature forms; the platelet countwas low (94,000) and the hemoglobin level was normal. Abone marrow smear disclosed nearly complete invasion bylymphoblasts. Findings were negative from tests for rheuma-toid factor (latex and Waaler–Rose) and antinuclear antibod-ies. No proteinuria was found in a 24-hour urine sample. Liverand renal function tests were normal, as were serum levels ofcalcium, phosphate, and uric acid. Plain radiographs dis-closed microgeodes in the carpal bones and metacarpal headswith heterogeneous demineralization of the distal radius andulna (Fig. 1). The chest radiograph was normal. An ultra-sound scans of the abdomen showed enlargement of the spleenwithout enlargement of the liver or deep lymph nodes.A diag-nosis of acute lymphoblastic leukemia was given, and shewas transferred to the hematology ward. The blast-cell phe-notype was CD3–, CD7–, CD10+ and CD20+, consistent withpre-B acute lymphoblastic leukemia. The karyotype showed

Fig. 1. Radiograph of the hands showing patchy demineralization of the dis-tal radius and ulna, as well as microgeodes in the carpal bones and heads ofthe metacarpals and phalanges.

189Letters to the Editor / Joint Bone Spine 72 (2005) 187–191

t(1,19) and t(4,11) translocation with no t(9,22) transloca-tion. Combination chemotherapy was given according to theLALA protocol (vincristine, prednisone, daunorubicine, andcyclophosphamide). A remission of the leukemia wasachieved. Concomitantly, the clinical manifestations of pol-yarthritis resolved almost completely. The radiographic abnor-malities remained unchanged, although nine months later, sheexperienced a serious infection responsible for fatal sepsis.

3. Discussion

Musculoskeletal manifestations of acute leukemia areuncommon in adults. Only 4% of adults with acute leukemiapresent with musculoskeletal symptoms, as compared to 14%of children [1,3]. Musculoskeletal symptoms may reveal arelapse or conversion of chronic to acute leukemia [4]. In chil-dren, males are more likely than females to exhibit muscu-loskeletal symptoms [5]. Bone pain is present in only 5% ofadults at diagnosis but occurs in 50% of cases during thecourse of the disease. The pain is usually diffuse but is morecommon in the axial than in the peripheral skeleton [3,5].Fixed pain with an inflammatory time pattern may occur atfoci of histological bone marrow necrosis [1]. Joint manifes-tations include migratory fleeting arthralgia and acute oligoar-thritis or polyarthritis predominantly affecting the large limbjoints (e.g., the knees and ankles) but also involving the smalljoints in the hands and feet [1,2,6–8]. The joint manifesta-tions may run a migratory course or remain fixed, withinvolvement of additional joints over time [9]. Joint involve-ment is symmetric in 60% of cases [3,5,10]. Our patient hadchronic polyarthritis with predominant involvement of thehands, a pattern very rarely described in the literature. Thediagnosis of leukemic synovitis rests on the identification ofblast cells in joint fluid or synovial biopsy specimens [1,3,6–9,11]. Few studies have reported joint fluid findings; inflam-matory properties with a predominance of neutrophils arecommon. Cytology usually fails to identify blast cells in jointfluid, whereas immunohistochemistry has a better yield[3,9,11]. In most patients, plain radiographs are normal orshow osteoporosis or osteolysis predisposing to pathologicalfractures. A sclerotic reaction to the leukemic infiltration isexceedingly rare. Also uncommon is glucocorticoid-inducedepiphyseal necrosis, which may induce joint incongruence[1,5]. The radiographs obtained in our patient disclosed micro-geodes in the carpal bones and metacarpophalangeal joints.This pattern is atypical, and most previously reported casespredominantly affected the large limb joints. Plain radio-graphs must be obtained in patients with local symptoms, mostnotably bone pain [5]. Computed tomography (CT) is rarelyuseful; the main indication is investigation of solitarychloroma-like bone lesions, which raise diagnostic and thera-peutic problems. The CT provides an accurate evaluation ofthe bone and soft tissue lesions prior to local radiation therapy,and is also useful for guiding diagnostic biopsies. Magneticresonance imaging (MRI) is the most sensitive imaging

method, particularly in adults. Infiltration of the bone mar-row by blast cells is seen as low signal on T1-weighted images.The MRI is also extremely valuable for diagnosing spinal orcerebral complications [5], as well as iatrogenic complica-tions such as osteonecrosis and osteomyelitis. Many patho-physiogenic mechanisms have been considered to explain theoccurrence of joint disease in patients with acute leukemia[1,9,11–13]. Leukemic arthritis strictly speaking is infiltra-tion of the synovial membrane by blast cells. Nonspecificsynovitis may develop in response to blast-cell infiltration ofthe juxtaarticular bone. Subcapsular or intraarticular bleed-ing may occur, as well as microcrystalline or septic arthritiswith immune complex deposition. Chemotherapy is the cor-nerstone of the management of osteoarticular involvement inpatients with acute leukemia. The prognosis of the joint lesionsis governed by the prognosis of the malignancy [2].

4. Conclusion

Bone involvement by blast cells can be the presentingmanifestation of acute leukemia in adults. Leukemic involve-ment of the synovial membrane is less often seen, and jointmanifestations are often due to iatrogenic complications.

References

[1] Bachmeyer C, Grateau G, Sauvage C. Douleurs ostéo-articulaires etostéocondensation révélatrices d’une leucémie aiguë chez l’adulte.Rev Rhum 1994;61:474–5.

[2] Gur H, Koren V, Ehrenfeld M, Ben-bassat I. Rheumatic manifesta-tions preceding adult acute leukemia: characteristics and implicationin course and prognosis. Acta Haematol 1999;101:1–6.

[3] Pertuiset E. Manifestations ostéo-articulaires des hémopathiesmalignes. EMC appareil locomoteur 1994;14(027-A-20):8.

[4] Taillan B, Fuzibet JG, Vinti H. Arthrite leucémique du genou au coursde la transformation blastique d’une leucémie myélomonocytairechronique. Rev Rhum Mal Ostéoart 1988;55:1038–9.

[5] Feger C, Frija J. Hémopathies: aspects radiologiques des atteintesostéo-articulaires des leucémies aiguës. In: Laredo JD, Morvan G,Wybier M, editors. Imagerie Ostéo-articulaire. Flammarion; 1998. p.110–7.

[6] Rennie JAN, Auchterlonie IA. Rheumatological manifestations ofleukemias and graft versus host disease. Baillieres Clin Rheumatol1991;5:231–51.

[7] Meyer R, Niederberger P, Chaix JM, Kuntz JL, Asch L. Arthritesrévélatrices de leucoses aiguës chez l’adulte. A propos de 4 observa-tions personnelles. Rev Rhum Mal Osteoartic 1985;52:255–8.

[8] Holdrinet R, Cotens F. Leukemic synovitis. Am J Med 1989;86:123–6.

[9] Taillan B, Leyge JF, Fuzibet JG. Knee arthritis revealing acute leuke-mia in a patient with rheumatoid arthritis. Clin Rheumatol 1991;10:76–7.

[10] Luzar MJ, Sharma HM. Leukemia and arthritis: including reports andlight, immunofluorescent and electron microscopy of the synovium. JRheumatol 1983;10:132–5.

[11] Weinberger A, Schumacher R, Schimmer B. Arthritis in acute leuke-mia. Clinical and histological observations. Arch Intern Med 1981;141:1183–7.

[12] Spilberg I, Meyezr GJ. The arthritis of leukemia. Arthritis Rheum1972;15:630–5.

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[13] Bedwell GA, Danson AM. Chronic leukemia in a child presenting asacute polyarthritis. Arch Dis Child 1954;29:78–9.

Bouchra Amine *Karima Benbouazza

Fadoua AllaliAfaf Faik

Houda MaaroufiSalma El Hassani

Najia Hajjaj-HassouniRheumatology Department B, El Ayachi Hospital,

Ibn Sina Teaching Hospital, Rabat- Salé, Morocco

E-mail address: amine_bouchra@yahoo.fr (B. Amine).

Received 26 March 2003; accepted 29 September 2004

Available online 15 December 2004

* Corresponding author. Tel.: +00+212+37+78+17+14;fax: +00+212+37+88+33+27.

1297-319X/$ - see front matter © 2004 Published by Elsevier SAS.doi:10.1016/j.jbspin.2004.09.009

191Letters to the Editor / Joint Bone Spine 72 (2005) 187–191