Successful treatment of hepatitis B-associated polyarteritis nodosa with a combination of lamivudine and conventional immunosuppressive therapy: a case report
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(2) Letters to the Editor. [1], which may also possibly worsen the vasculitis by enhancing viral antigenaemia. Lamivudine has recently been shown to suppress the replication of HBV by >98% [2–5] and could thus allow the use of conventional immunosuppressive agents to control the vasculitis. We report the case of a patient treated successfully in this manner. The patient, a general surgeon, developed diffuse myalgias in August 1998. The symptoms initially resolved with high-dose prednisone (150 mg daily), but returned 4 weeks later following tapering of the dose. There was also anorexia, lethargy, night sweats and weight loss of 3 kg. A biopsy of the left temporal artery was normal. A biopsy of the left gastrocnemius showed slight lymphocytic perivascular interstitial infiltration. Four weeks later, in October 1998, admission to our unit was prompted by the development of mononeuritis multiplex. There was a 15-yr history of mild labile hypertension treated with a beta-blocker. No other risk factors for hepatitis B could be elicited apart from his profession. There was a current smoking history (30 pack-yr). General examination was unremarkable apart from a blood pressure of 140/90 mmHg in both arms. There was slight tenderness of the calves, but no evidence of synovitis. Sensorimotor defects were evident in the distribution of several peripheral nerves, most profoundly involving the left common peroneal nerve, but also involving the right common peroneal, both tibial and the left median nerves. The left ankle jerk was absent. The laboratory data showed: leucocytes 15.8 × 109/l, C-reactive protein (CRP) 280 mg/l, alanine aminotransferase (ALT ) 77 U/l, c-glutamyltranspeptidase (GGT ) 21 U/l, normal renal function tests and urinary microscopy, slight proteinuria (0.13 g/l ), positive hepatitis B surface antigen ( HBsAg), ‘e’ antigen (HBeAg) and antibodies to the core antigen (anti-HBc), HBV viral load 1 × 107 genome equivalents (geq)/ml, other viral serology negative, antinuclear antibodies (ANA) negative, p-ANCA borderline. The liver biopsy showed chronic minimally active hepatitis with slight intralobular lymphocytic infiltration, occasional cell necroses, slight periportal lymphocytic infiltration and scattered piecemeal necroses. The left sural nerve biopsy was, however, unrevealing. Hepatitis B-associated polyarteritis nodosa was diagnosed and treatment with lamivudine 150 mg daily was started. Three daily pulses of 500 mg methylprednisolone were administered i.v., followed by 80 mg daily in tapering dosage. On discharge from hospital 1 week later on oral prednisone, 40 mg daily, the viral load had reduced to 2 × 105 geq/ml. Over the next 4 weeks, the patient complained of increasing anorexia, post-prandial epigastric fullness and weight loss of a further 11 kg. The neurological symptoms were unchanged. The blood pressure varied between 140/105 and 150/110 mmHg. The patient was readmitted in November 1998 in a severely cachectic state. The CRP was 161 mg/l. The viral load had. 107. F. 1. Visceral digital subtraction arteriography of the superior mesenteric artery showing microaneurysms and irregularities of callibre consistent with polyarteritis nodosa.. rebounded to 4 × 107 geq/ml following cessation of the lamivudine treatment by the patient 4 days previously. Visceral angiography (Fig. 1) performed at this point showed multiple microaneurysmata and calibre irregularities in the arterial supply of the liver, spleen, pancreas, intestine and kidneys. Cyclophosphamide was commenced, initially 150 mg daily, reducing to 100 mg daily after 2 weeks. Three further daily i.v. pulses of methylprednisolone 500 mg were administered, followed by 80 mg daily i.v. in tapering dose, before changing to oral prednisone. Lamivudine was restarted at a dose of 50 mg twice daily. The hypertension was treated with enalapril, 10 mg daily. The patient improved rapidly and was discharged 3 weeks later. The cyclophosphamide was reduced to 50 mg daily due to profound lymphopenia. In April 1999, after 4 months of treatment, the cyclophosphamide was stopped and the lamivudine reduced to 50 mg daily due to an unclear asymptomatic increase in the liver enzymes (maximal ALT 754 U/l, GGT 295 U/l ), most probably due to drug toxicity however as the HBV viral load remained low and there was no evidence of active vasculitis. The prednisone had been tailed to 7.5 mg daily. The transaminases gradually returned to normal over 3 months. At the last review in June 1999, while on prednisone 5 mg daily and lamivudine 50 mg daily, the patient had regained his previous weight and the weakness in the distribution of the involved peripheral nerves had improved considerably, although there was still marked sensory loss on the soles of the feet and.
(3) Letters to the Editor. 108. slight paraesthesiae in the median nerve distribution of the left hand. The CRP was <5 mg/l and the liver enzymes were within the normal range. The viral load was now only 100 geq/ml, but the patient was still HBsAg and HBeAg positive. This case illustrates successful treatment of hepatitis B-associated polyarteritis nodosa with a combination of lamivudine to prevent increased viral replication and conventional immunosuppression to treat lifethreatening vasculitis. We had initially hoped that the reduction of viraemia would be sufficient in combination with high-dose corticosteroids to control the vasculitis, but improvement only occurred when cyclophosphamide was added to the regimen. There was, however, no relapse following the cessation of cyclophosphamide treatment after 4 months, suggesting that the vasculitis was now in remission following almost complete suppression of viral replication. D. M, M. B1, A. L. J2, A. T University Department of Rheumatology, Felix Platter Spital, Burgfelder Strasse 101, Basel, 1University Medical Outpatient Department and 2University Institute of Diagnostic Radiology, Kantonsspital Basel, Petersgraben 4, Basel, Switzerland Accepted 13 July 1999 Correspondence to: D. Maclachlan, University Department of Rheumatology, Felix Platter Spital, Burgfelder Strasse 101, CH-4055 Basel, Switzerland. 1. Lam KC, Lai CL, Tre´po C, Wu PC. Deleterious effect of prednisolone in HBsAg-positive chronic hepatitis. N Engl J Med 1981; 304:380–6. 2. Lai CL, Ching CK, Tung AKM et al. Lamivudine is effective in suppressing hepatitis B virus DNA in Chinese hepatitis B surface antigen carriers: a placebo controlled trial. Hepatology 1997; 25:241–4. 3. Dienstag JL, Perillo RP, Schiff ER, Bartholemew M, Vicary C, Rubin R. A preliminary trial of lamivudine for chronic hepatitis B infection. N Engl J Med 1995;333:1657–61. 4. Nevens F, Main J, Honkoop P et al. Lamivudine therapy for chronic hepatitis B: a six month randomised dose-ranging study. Gastroenterology 1997;113:1258–63. 5. Lai CL, Chien RN, Leung NWY et al. A one-year trial of lamivudine for chronic hepatitis B. N Engl J Med 1998;339:61–8.. Antiphospholipid syndrome with proliferative vasculopathy and bowel infarction S, Since antiphospholipid syndrome (APS) was first described in 1983 [1], many complications have been reported which result from vascular occlusion as a result of thrombosis [2]. More recently, a few cases have been described where a proliferative vasculopathy appears to have been the major mechanism of vascular occlusion rather than thrombosis or vasculitis [3–5]. Relatively few cases have been described with intestinal infarction as a result of vascular occlusion from this ‘proliferative vasculopathy’ of APS [6–9]. We describe one such patient who had massive intestinal infarction as a result of a proliferative vasculopathy involving the coeliac,. superior mesenteric (SMA), and inferior mesenteric arteries (IMA). A 43-yr-old female was first referred to us in January 1994 when she was diagnosed as having systemic lupus erythematosus with APS. The clinical features up to that point (developing over a 10 yr period) included polyarthralgia, immune thrombocytopenia necessitating a splenectomy, livedo reticularis, Raynaud’s phenomenon, photosensitive skin disease, myocardial dysfunction with evidence of a left ventricular thrombus, and neurological events including initial transient facial weakness followed years later by a right sided middle cerebral artery stroke. She recovered from the neurological deficits except for a dysarthria for which she used an electronic communicator. Over the years the patient also developed hypertension with small smooth kidneys but normal renal function and no proteinuria. Interestingly, she had no history of fetal wastage (three sons and no miscarriages). Blood results included a positive antinuclear antibody (1/2560, homogeneous pattern) and DNA binding (237 IU/ml, normal <100 IU/ml ). IgG anticardiolipin antibodies were mildly elevated at 10–12 GPL and IgM always negative (on repeated measurement). Antibodies to b2-GP1 were also negative. Protein C, protein S and complement levels were normal. Of interest, there was a mild prolongation of the PTT (partial thromboplastin time) at the time of the stroke, suggesting the presence of a lupus anti-coagulant. The platelet count always remained stable but depressed at around 120 × 109/l. The patient remained well for a year on therapy with lisinopril, nifedipine, hydroxychloroquine, erythromycin and warfarin at optimal doses (INR always maintained at 3–4). However, in April 1996 she developed symptoms ascribed to an irritable bowel syndrome. Within 2 months she lost 6 kg in weight and was having symptoms of mesenteric angina. Endoscopy revealed multiple haemorrhagic areas in the small and large bowel, biopsies of which showed no vasculitis or thrombosis. However, histological features were in keeping with mucosal ischaemia. In the interim the patient developed an ‘acute abdomen’ and required urgent laparotomy. She was found to have extensive infarction involving the jejunum, ileum and large bowel extending to the sigmoid region, all of which required resection. Although inspection of the intestinal vessels appeared normal at surgery, intra-operative angiography revealed total occlusion of coeliac, SMA and IMA at their origins (with the right renal artery resembling radiological features of fibromuscular hyperplasia at its origin; Fig. 1). There was very extensive collateral vessel formation from the iliac and internal thoracic arteries indicating chronicity of the vascular disease. Despite intensive surgical and medical efforts including bowel resection, SMA revascularization by venous grafting, parenteral feeding, intravenous antibiotics, and total anticoagulation, the patient continued to deteriorate with ongoing features of ischaemia in the residual bowel. It was thus decided to embark on immunosuppression with pulse cyclophosphamide and steroids, but after a.
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