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Remitting distal lower extremity swelling with pitting oedema in acute sarcoidosis
  1. FABRIZIO CANTINI,
  2. LAURA NICCOLI
  1. 2^ Divisione di Medicina, Ospedale di Prato
  2. Servizio di Reumatologia, Policlinico S Orsola-Malpighi, Bologna
  3. Servizio di Radiodiagnostica, Policlinico S Orsola-Malpighi, Bologna
  4. Istituto Pratese di Radiodiagnostica, Prato
  5. 2^ Divisione di Medicina, Arcispedale S Maria Nuova, Reggio Emilia, Italy
  1. Dr F Cantini, 2^ Divisione di Medicina, Ospedale di Prato, Piazza Ospedale, 1-50047, Prato, Italy.
  1. IGNAZIO OLIVIERI
  1. 2^ Divisione di Medicina, Ospedale di Prato
  2. Servizio di Reumatologia, Policlinico S Orsola-Malpighi, Bologna
  3. Servizio di Radiodiagnostica, Policlinico S Orsola-Malpighi, Bologna
  4. Istituto Pratese di Radiodiagnostica, Prato
  5. 2^ Divisione di Medicina, Arcispedale S Maria Nuova, Reggio Emilia, Italy
  1. Dr F Cantini, 2^ Divisione di Medicina, Ospedale di Prato, Piazza Ospedale, 1-50047, Prato, Italy.
  1. LIBERO BAROZZI,
  2. PIETRO PAVLICA
  1. 2^ Divisione di Medicina, Ospedale di Prato
  2. Servizio di Reumatologia, Policlinico S Orsola-Malpighi, Bologna
  3. Servizio di Radiodiagnostica, Policlinico S Orsola-Malpighi, Bologna
  4. Istituto Pratese di Radiodiagnostica, Prato
  5. 2^ Divisione di Medicina, Arcispedale S Maria Nuova, Reggio Emilia, Italy
  1. Dr F Cantini, 2^ Divisione di Medicina, Ospedale di Prato, Piazza Ospedale, 1-50047, Prato, Italy.
  1. ALESSANDRO BOZZA
  1. 2^ Divisione di Medicina, Ospedale di Prato
  2. Servizio di Reumatologia, Policlinico S Orsola-Malpighi, Bologna
  3. Servizio di Radiodiagnostica, Policlinico S Orsola-Malpighi, Bologna
  4. Istituto Pratese di Radiodiagnostica, Prato
  5. 2^ Divisione di Medicina, Arcispedale S Maria Nuova, Reggio Emilia, Italy
  1. Dr F Cantini, 2^ Divisione di Medicina, Ospedale di Prato, Piazza Ospedale, 1-50047, Prato, Italy.
  1. PIER LUIGI MACCHIONI,
  2. ANGELA PADULA,
  3. CARLO SALVARANI
  1. 2^ Divisione di Medicina, Ospedale di Prato
  2. Servizio di Reumatologia, Policlinico S Orsola-Malpighi, Bologna
  3. Servizio di Radiodiagnostica, Policlinico S Orsola-Malpighi, Bologna
  4. Istituto Pratese di Radiodiagnostica, Prato
  5. 2^ Divisione di Medicina, Arcispedale S Maria Nuova, Reggio Emilia, Italy
  1. Dr F Cantini, 2^ Divisione di Medicina, Ospedale di Prato, Piazza Ospedale, 1-50047, Prato, Italy.

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Remitting distal extremity swelling with pitting oedema is a clinical manifestation that may be observed in different rheumatic conditions such as RS3PE syndrome,1 polymyalgia rheumatica,2 late onset undifferentiated spondyloarthropathy,3 ankylosing spondylitis,4 psoriatic arthritis,5 and more rarely in other rheumatic disease.5 In RS3PE the swelling with pitting oedema predominantly involves the upper extremities symmetrically.1 This clinical finding may occur unilaterally in polymyalgia rheumatica and upper and lower limbs may be equally affected,2 whereas in seronegative spondyloarthropathies it has been described more frequently in lower extremities with asymmetric involvement.3 4

The oedema is characterised by a rapid response to small doses of corticosteroids.

The pathogenesis of swelling with pitting oedema has not been completely defined, but the distribution along the tenosynovial membranes suggests that it probably results from tenosynovitis.5

Sarcoidosis is a systemic granulomatous disease that is associated with rheumatic manifestations in 6% to 39% of patients.6 The most common form of arthritis occurs acutely, involves preferentially the ankles, and it is often the initial feature of sarcoidosis.6 Frequently, erythema nodosum and bilateral hilar adenopathy are associated with arthritis (Lofgren’s syndrome). Erythema nodosum may also be absent.7

Periarticular oedema and tenosynovitis accompanying arthritis have been emphasised in some reports.6 8-10 However, remitting distal lower extremity swelling with pitting oedema of the dorsa of the feet in patients with acute sarcoidosis has not been mentioned in previous reports.

We describe five patients with acute sarcoidosis who presented as first manifestation of the disease an impressive distal lower extremity swelling with pitting oedema of the dorsum of both feet.

We made a retrospective review of all patients with acute sarcoidosis seen in two Italian Divisions of Internal Medicine ( Prato and Reggio Emilia Hospitals) over a two year period. The diagnosis and presenting clinical features were assessed by clinical examination and review of the patient’s chart.

Remitting distal extremity swelling with pitting oedema was the presenting manifestation in five (29%) of 17 consecutive patients with acute sarcoidosis seen between January 1995 and December 1996.

Table 1 summarises the demographic and clinical characteristics of the five patients. All patients presented with pain and swelling of both ankles and feet.

Table 1

Demographic, clinical, and magnetic resonance imaging features of the five patients with acute sarcoidosis

The swelling and pitting oedema were most prominent over the dorsum of both feet and ankles, malleolar and perimalleolar areas were also involved (fig 1). In perimalleolar areas the oedema followed the distribution of tibialis and peroneal tendons. It is possible that pitting oedema in our series may have been present in some other cases, but less pronounced and not considered by the examiner.

Figure 1

Patient 2. Soft tissue swelling of malleolar and perimalleolar areas with pitting oedema of the dorsum of the right foot. The distribution of the oedema follows the course of peroneal, tibialis, and extensor tendons of the foot.

Swelling and oedema completely resolved after a few days of corticosteroid treatment.

To investigate the structures responsible for the swelling and oedema we examined three patients by magnetic resonance imaging.

As reported by Olivieri et al 5 in other rheumatic conditions associated with distal extremity swelling with pitting oedema, magnetic resonance imaging confirmed in three patients the clinical impression that a severe tenosynovitis of peroneal, tibialis, and extensor tendons was the main lesion responsible for the oedema in the subcutaneous and peritendineous ankle and foot soft tissues. A slight joint synovitis with scanty intrarticular fluid was present (fig 2).

Figure 2

Patient 4. Right ankle axial magnetic resonance imaging (SE T2weighted): pronounced tenosynovitis of posterior tibial tendon (arrow). Subcutaneous oedema and mild joint synovitis with scanty intrarticular fluid may be observed.

Similar findings were reported by Kellner et al 9 by using ultrasound to study ankle swelling in acute sarcoidosis. Sonographic features of tenosynovitis were detected in eight of 24 patients, only in two cases a concomitant arthritis was found.

In conclusion our cases confirm that a tenosynovitis is part of the spectrum of articular manifestations of sarcoidosis. Moreover, our clinical series suggest that distal extremity swelling with pitting oedema over the dorsum of feet may be the presenting feature of acute sarcoidosis. Consequently the disease may be included among the rheumatic disorders associated with remitting distal extremity swelling and pitting oedema.

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