Rheumatol Int (2012) 32:2557–2559

DOI 10.1007/s00296-011-2047-z

SHORT COMMUNICATION

Polyarthritis and posterior interosseous nerve palsy associated with gastric carcinoma

Kensuke Ochi · Yukio Horiuchi · Miyoka Seki · Kazuo Nishi · Hiroyuki Nozaki · Hiroki Yabe

Received: 1 March 2011 / Accepted: 10 July 2011 / Published online: 24 July 2011© Springer-Verlag 2011

Abstract Cancer polyarthritis is a very rare condition.Here, we present a case of cancer polyarthritis, who alsohad mononeuritis simplex as a manifestation of paraneo-plastic neuralgic syndrome. A 71-year-old man, who ini-tially presented symmetrical polyarthritis and unilateralposterior interosseous nerve palsy, was subsequently diag-nosed to have gastric cancer. Total gastrectomy was per-formed, and his polyarthritis and the palsy simultaneouslydisappeared within 2 weeks after the resection. His gastriccancer was found to be metastasized to his liver 16 monthsafter the total gastrectomy; however, the polyarthritis andthe palsy did not recurrent throughout his course. The poly-arthritis in this case was diagnosed as a cancer polyarthritisfrom its features. On the other hand, the isolated posteriorinterosseous nerve palsy in this case met the diagnostic cri-teria for paraneoplastic neurological syndrome. This case

was also unique in that the manifestations of paraneoplasticsyndromes did not recur even after the metastasis of the pri-mary cancer, suggesting that some speciWc clones in thecancer were responsive to the manifestations of paraneo-plastic syndromes. Our case suggested that relapse of themanifestations of paraneoplastic syndromes may notalways herald the recurrence of primary or metastatictumour, and other tumour markers and signs should be peri-odically followed to search for the recurrence of thetumours.

Keywords Cancer polyarthritis · Mononeuritis simplex · Paraneoplastic neurological syndrome · Paraneoplastic rheumatic disorder · Posterior interosseous nerve syndrome · Posterior interosseous nerve palsy

AbbreviationsPINP Posterior interosseous nerve palsyPNS Paraneoplastic neurological syndrome

Introduction

Malignant neoplasms or its metastases are known to aVectvarious organs remote from the site of malignant cancer[1, 2]. This condition is referred as ‘paraneoplastic syn-dromes’ and believed to be because the tumour secretessubstances that mimic normal hormones or that interfereswith circulating proteins, and also immune-mediated [1–6].Paraneoplastic rheumatic disorders have been described ina number of solid tumours and haematological malignan-cies. Clinical features of some of the paraneoplastic rheu-matic disorder patients are very similar to those ofrheumatoid arthritis and have been described and repre-sented as cancer polyarthritis [1], carcinoma polyarthritis or

K. Ochi (&) · Y. HoriuchiDepartment of Orthopaedic Surgery, Kawasaki Municipal Kawasaki Hospital, 12-1 Shinkawa-dori, Kawasaki-ku, Kawasaki City, Kanagawa 210-0013, Japane-mail: ochi@1998.jukuin.keio.ac.jp

M. SekiSakura Seikeigeka Orthopaedic Clinic, Saitama, Japan

K. NishiDepartment of Rheumatology, Kawasaki Municipal Kawasaki Hospital, Kanagawa, Japan

H. NozakiDepartment of Neurology, Kawasaki Municipal Kawasaki Hospital, Kanagawa, Japan

H. YabeDivision of Rheumatology, Jichi Medical University Saitama Medical Center, Saitama, Japan

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rheumatoid arthritis-like paraneoplastic syndrome [1–3].However, this condition is very rare (<1/1,000,000 in apopulation [7]) and sometimes diYcult to diagnosis if aphysician is not aware of cancer polyarthritis since it isoften asymptomatic and sometimes occult [1–3, 7, 8]. Thus,awareness of the possibility of a paraneoplastic process isnecessary for the timely treatment of the underlying malig-nancy and may be life-saving in some cases.

Paraneoplastic neurological syndromes (PNSs) are alsorare syndromes (<1/10,000 patients with cancer) and diY-cult to diagnose without awareness of this condition.Although it is believed that PNSs can aVect any part of thenerve system [4–6], there is no report of mononeuritis sim-plex associated with malignant neoplasms to the best of ourknowledge.

We here report a patient who initially presented symmet-rical inXammatory polyarthritis and unilateral posteriorinterosseous nerve palsy (PINP) and was subsequentlydiagnosed to have gastric cancer. Both his polyarthritis andpalsy completely remitted 3 months after the total gastrec-tomy, suggesting that these manifestations were of paraneo-plastic syndromes. None of the manifestations ofparaneoplastic syndrome recurred even after metastasis ofprimary cancer. This case suggested that tumour recurrencemay not always have been heralded by the return of mani-festations of paraneoplastic syndrome and that some spe-ciWc clones in the primary cancer were responsive to themanifestations of paraneoplastic processes.

Case presentation

A 71-year-old Japanese man with good previous health Wrstpresented to our clinic because of abrupt onset of symmetri-cal polyarthritis involving the shoulders, knees and wristswith marked morning stiVness of the hands. His arthritisstarted on his right knee in mid-July 2008 and eventuallyprogressed to symmetrical multiple joint pains. He had nopersonal or family history of rheumatic diseases, and therewas no vaccination or viral symptoms prior to this presenta-tion. Physical examination showed swelling and tendernessof the involved joints without pitting oedema. There wereno subcutaneous nodules, clubbing or palmar fasciitis. Hehad weight loss of 6 kg within the last 2 months. A com-plete blood count showed elevated white blood cell count(11,240/�l) and platelet count (40.6 £ 104/�l). Laboratoryinvestigations were negative for the rheumatoid factor, anti-nuclear antibody and the anti-cyclic citrullinated peptide(CCP) antibody. His erythrocyte sedimentation rate (ESR)was elevated to 104 mm/1 h, and so was the C-reactive pro-tein (CRP) (15.6 mg/dl), matrix metal protease-3 (MMT-3)(533 ng/ml) and soluble interleukin-2 receptor (sIL-2R)(766 U/ml). CA 19–9 was 197.4 U/ml. Serum urate level

was normal. Radiological examination of the respectivejoints did not reveal any erosion. A chest radiograph wasnormal.

He also had complete palsy in his right thumb and Wngerextensors at his Wrst presentation. He had a severe pain inhis right arm, 2 weeks before the onset of the palsy. Elect-rodiagnostic evaluations were made, and isolated posteriorinterosseous nerve palsy (PINP) was conWrmed [9]. Mag-netic resonance imaging of his right elbow showed no evi-dence of space-occupying lesion (tumour or severesynovitis).

Although this case seemed to be rheumatoid arthritiscoincided with isolated PINP, we also searched for anoccult malignancy since polyarthritis in this case had somefeatures for carcinoma polyarthritis: late age of onset,explosive onset, absence of rheumatoid factor and no fam-ily history of rheumatoid disease [1–3]. Upper gastrointesti-nal endoscopic evaluation demonstrated an advancedtumour of the stomach. The patient underwent total gastrec-tomy, and histological evaluation revealed it as moderatelydiVerentiated tubular adenocarcinoma. Within 2 weeksafter the total gastrectomy, both polyarthritis and PINPstarted to improve. After 3 months of gastrectomy, bothpolyarthritis and his palsy remitted completely.

Liver metastasis was found 16 months after the total gas-trectomy. Partial resection of liver was performed, and his-tological evaluation revealed it as adenocarcinomacompatible with metastasis of primary gastric cancer. How-ever, none of the manifestations of neither arthritis nor neu-ritis recurred despite the metastasis of primary carcinoma.Serum sIL-2R and MMP-3 levels also did not increase afterthe appearance of the metastasis.

Discussion

Paraneoplastic rheumatic disorders are very rare conditions[1–8]. The pathophysiology of these conditions is yetunknown; however, tumour-related immune mechanismssuch as cytokines, hormones, antigens, antibodies and cyto-toxic lymphocytes are believed to be involved [1–5].

This case was diagnosed as cancer polyarthritis since ithad most of the clinical features of cancer polyarthritis [1–3, 7, 8]. The cancer found in our case was adenocarcinoma,which is known to be the most frequent histological typefor cancer polyarthritis [7]. Serum sIL-2R level, which isone of the markers for immune activation [10], alsodecreased as the manifestations in our case disappeared.This also seemed to support an involvement of neoplasm-related immunological mechanisms in the pathogenesis ofthe manifestations in our case.

Not only the polyarthritis but also the PINP disap-peared soon after the removal of primary gastric carcinoma,

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suggesting that this palsy was the manifestation of paraneo-plastic syndrome. The clinical features of this palsy metthe diagnostic criteria for PNSs [4], also suggesting thatthis was the Wrst case of mononeuritis simplex as a manifes-tation of PNS. Another sign supporting the possibility ofthis PINP as PNS may be the severe pain seen just beforethe onset of palsy. This severe pain may be because of someimmune-mediated mechanisms reacting directly on thenerve itself. The immune responses ampliWed by cancerpolyarthritis may be one of the possible mechanisms ofthis unusual condition.

It has been reported that tumour recurrence could be her-alded by the return or initial appearance of a paraneoplasticrheumatic disorder [1, 2]. However, none of the manifesta-tions of paraneoplastic syndrome relapsed in our case. Oneof the reasons may be because the polyarthritis and PINP inour case were due to some speciWc clones in the primarygastric cancer. In this condition, polyarthritis and PINPmay completely remit if such speciWc clones were com-pletely removed by surgery. Other possibility may begrowth rate of such speciWc clones was much slower thanrest of the clones, or the speciWc clones might have trans-formed to lose its potential to induce mononeuritis and pol-yarthritis. Our case suggested that relapse of themanifestations of paraneoplastic syndromes may notalways herald the recurrence of primary or metastatictumour, and other tumour markers and signs should be peri-odically followed to search for the recurrence of thetumours.

Palmar fasciitis and polyarthritis, which is another para-neoplastic rheumatic disorder [11], may be a diVerentialdiagnostic disease in our case. Clinical features of palmarfasciitis and polyarthritis are rapidly progressive Xexioncontractures of both hands and polyarthritis. Flexion con-tractures seen in this disorder are believed to be because ofnodular thickening of fascia of the palms. However, theWndings in the present case diVered from the above disorderin that the PINP was conWrmed electrophysiologically andthickening of palmar fascia and Xexion contracture werenot observed in our case.

Acknowledgments We would like to thank Dr. Y. Oosone, Dr.T. Suzuki and Dr. H. Taguchi at the Department of Rheumatology, Ka-wasaki Municipal Kawasaki Hospital, and Prof. C. Terai at the Divi-sion of Rheumatology, Jichi Medical University Saitama MedicalCenter, for helpful discussions. We also would like to thank Dr.M. Kihara, Dr. N. Nakamichi, Dr. K. Morita, Dr. M. Yagi, Dr.K. Takeda, Dr. K. Ninomiya, Dr. T. Hasegawa and Dr. E. Okada in theDepartment of Orthopaedic Surgery, Kawasaki Municipal KawasakiHospital, and also members in the hand surgery group, Keio UniversitySchool of Medicine, for helpful suggestions.

ConXict of interest The authors declare that they have no conXict ofinterest.

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