sign in sign up
<<
Home , Dermatomyositis, Hypercalcaemia

Case Communications IMAJ • VOL 17 • october 2015

Multiple Oleomas Presenting as

Jalaa Zarroug MD1, Graham R.V. Hughes MD FRCP2 and Christopher J. Edwards MD FRCP1

1NIHR Wellcome Trust Clinical Research Facility, University Hospital Southampton NHS Foundation Trust, Southampton, UK 2The London Centre, London Bridge Hospital, London, UK

especially in the deltoid, biceps normal CPK 89 IU/L (26–140). Renal func- Key words: oleoma, dermatomyositis, and triceps muscles. A subsequent biopsy tion, function and glucose were all , connective tissue taken from an area of deep tenderness and normal. Serum angiotensin-converting disease swelling in the region of the left deltoid (ACE) was slightly raised at 66 IMAJ 2015; 17: 644–645 muscle was described as showing mini- U/L (8–52), as was gammaglobulin, 41 g/L mal non-specific ; Congo (20–40). including ANA, red staining to seek amyloid was negative. extractable nuclear antigens (ENA) and he injection of silicone or oil-based Based on the patients’ history and pre- RF were all negative, anti-double stranded T substances into muscles for cosmetic sentation and these investigations, a diag- DNA antibodies were 7 IU/ml (< 50), IgG reasons can result in masses called oleo- nosis of dermatomyositis (DM) was made. anticardiolipin antibodies 4.6 U/ml (0–17) mas. We present the case of a 38 year old Treatment was started with and IgM anticardiolipin antibodies 1.5 U/ man who presented with muscle pain and and (AZA). The dose of pred- ml (0–15), but lupus anticoagulant screen- swelling associated with a high C-reactive nisolone decreased gradually from 60 to 40 ing was positive. The complement factor-3 protein (CRP). This was initially thought mg per day and AZA increased gradually (C3) and complement factor 4 (C4) were to be due to an idiopathic inflammatory from 50 to 200 mg per day (approximately both raised: 2.36 g/L (0.9–1.8) and 0.59 g/L muscle disease. However, detailed ques- 2.5 mg/kg/day). Despite this there was no (0.1–0.4) respectively. A Quantiferon test tioning and subsequent biopsy revealed apparent improvement after 5 months and for (TB) was negative. Urine other possibilities. the patient stopped treatment with no sig- analysis was normal and urine culture nificant deterioration. showed no growth. A chest X-ray was nor- At his latest presentation in our facility mal. MRI of the musculature of the upper PATIENT DESCRIPTION the patient complained mainly of symmet- body revealed evidence of inflammatory A 38 year old man originating from Qatar ric pain and swelling in a number of areas changes in discrete patches within biceps, presented to our unit with a history of including the triceps, biceps and deltoid triceps and deltoid bilaterally with hyper- dermatomyositis that had been resistant muscle groups. On examination, these intense signal on T1-weighted images and to treatment despite the use of predniso- areas appeared tender and swollen with heterogeneous intensity on T2 [Figure 1A]. lone and azathioprine. Two years earlier a palpable increase in temperature. The A plastic surgeon performed a deep he experienced symmetric muscle pain involved sites appeared as discrete hard biopsy of a lesion within the right triceps and swelling in the upper limbs. There swellings but the associated inflammatory muscle [Figure 1B], which on histological was also a history of some skin peeling tissue did not appear to extend through all examination showed areas of homogenous around the dorsal surface of the hands and the muscles involved. Muscle strength was material consistent with a foreign body, as a result he had undergone a number of normal in all muscle groups. Otherwise, such as an oil, surrounded by a granulo- investigations. These had shown an acute- the patient appeared well and further matous reaction [Figure 1C]. These find- phase response and a mild normochromic examination for features of myositis and ings led to further direct questioning of the normocytic anemia (hemoglobin 12 g/dl), connective tissue disease was unremark- patient who reported having received sev- but creatinine and liver function tests were able. In addition, from the history and eral injections of an oily substance into his normal. In particular, creatinine phospho- examination there was no suspicion of muscles for cosmetic reasons in an attempt kinase (CPK) was also normal. In addition, underlying . to enhance their definition. He did not (RF) and antinuclear Further investigations showed hemo- experience any problems for about 4 years antibodies (ANA) were negative; blood globin 147 g/L (130–17 g/L), white blood after receiving the last injection. However, cultures, urine culture and chest X-ray cells (WBC) 9.2 x109/L (4.0–10.0), slightly pain and swelling had developed and con- were normal. A magnetic resonance imag- raised eosinophils 0.6 x109/L (0.02–0.5), tinued for 2 years prior to review in our ing scan (MRI) with contrast of both upper high erythrocyte sedimentation rate (ESR) unit. The pain and swelling had become extremities showed evidence of active 56 mm/hour, raised CRP 129 mg/L, and more acute in the past 3 months.

644 IMAJ • VOL 17 • october 2015 Case Communications

A B C

Figure 1. [A] MRI T2-weighted image of upper extremity showing symmetric hyper-intense STIR signal suggestive [C] Histopathology of muscle specimen (hematoxylin of inflammation within the deltoid, [B] Muscle specimen from excision and eosin stain) with a foreign body-type reaction biceps and triceps muscles biopsy of the right triceps muscle consistent with a granulomatous reaction to lipid

with a persistently raised CRP of more elevated CRP but normal CPK. The serum COMMENT than 100 mg/L secondary amyloidosis ACE was also raised, presumably due to the The injection of silicone or oil-based might develop. With this in mind the fol- granulomas evident on the biopsy. However, substances including paraffin, sesame oil, lowing treatment regimen was begun. We the patient only developed the reaction 4 walnut oil and purified long-and medium- surgically removed as much inflamed and years after the injections, suggesting a loss chain emulsions has been described a num- foreign tissue as possible while preserving of tolerance. We questioned him about ber of times although most reports appear muscle function. Six areas were targeted in other factors that may have led to a loss of in the non-English language literature [1]. a staged procedure to remove inflamma- tolerance, such as immunization, infection Masses associated with this practice are tory masses from the right and left deltoid, or other surgical procedures, but there were commonly called oleomas, paraffinomas biceps and triceps muscles. The rationale none. We hope that debulking the foreign or lipogranulomas [2]. The most common behind this was to attempt to “debulk” as bodies and modest immunosuppression presentation is painful muscle fibrosis. In much foreign material as possible with will preclude further pain, muscle damage the long term, side effects may include the hope that it would reduce the inflam- and the negative consequences of a long- disfiguring scarring, loss of function, and matory response to the foreign material. term uncontrolled inflammatory response. deformity. Also reported are the side effects The followed protocol was then started: Correspondence associated with such injections, such as prednisolone 7 mg daily, colchicine 500 µg Dr. C.J. Edwards infections, ulcers, pruritus, oil embolism, twice a day, 10 mg weekly (to NIHR Wellcome Trust Clinical Research Facility, University Hospital Southampton NHS Foundation , pulmonary and be titrated up), and folic acid 5 mg weekly. Trust, Mailpoint 218, Southampton General neurological complications, with other We also planned an amyloid (SAP) scan to Hospital, Southampton, SO16 6YD, UK rarely described complications including look for amyloid deposits. Phone: (44-0-23) 8120-8723 Fax: (44-0-23) 8120 6711 severe hypercalcemia and This case highlights the dangers of email: [email protected] [3]. has been documented unregulated cosmetic procedures and the References following injection of anabolic androgenic powerful effect of long-term exposure to a 1. Benedetto G, Pierangeli M, Scalise A, et al. Paraffin in bodybuilders [4]. There are foreign antigen. In addition, it illustrates the oil injection in the body: an obsolete and destructive very few reports on how to treat these possibility that foreign body injection into procedure. Ann Plast Surg 2002; 49: 391-6. lesions, although the use of a number of muscle groups to enhance definition may 2. Georgieva J, Assaf C, Steinhoff M, et al. Bodybuilder oleoma. Br J Dermatol 2003; 149: 1289-90. including antibiotics have lead to the mistaken diagnosis of inflam- 3. Gyldenløve M, Røving S, Skov L, Hansen DN. Severe been described (long-term oral tetracycline, matory myositis. The key to the diagnosis , nephrocalcinosis, and multiple , colchicine, compression was of course the history of muscle injec- paraffinomas caused by paraffin oil injections in a bandaging, surgical drainage and plastic tion, but it is perhaps understandable that young bodybuilder. Lancet 2014; 383: 9934, s. 2098. 4. Farkash U, Shabshin N, Pritsch Perry M. Rhab- surgery) [5]. the patient was reluctant to divulge this domyolysis of the deltoid muscle in a bodybuilder For our patient we had a number of information. The patient’s reaction to the using anabolic-androgenic steroids: a case report. concerns. These included his ongoing pain, injected substance was also interesting. J Athl Train 2009; 44: 98-100. 5. Bjerno T, Basse PN, Siemssen PA, et al. Injection of deformity and reduced muscle function. When we reviewed the situation there was high viscosity liquids. Acute or delayed excision? We also considered the possibility that an intense foreign body response with an Ugeskr Laeger 1993; 155: 1876-8.

645