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Mesenteric Panniculitis in a Patient with New Onset Dermatomyositis Pankaj Bansal,1 Emily L Gilbert ‍ ‍ ,2 Rodrigo Otavio Lami Pereira,3 Andrew R Virata4

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Mesenteric Panniculitis in a Patient with New Onset Dermatomyositis Pankaj Bansal,1 Emily L Gilbert ‍ ‍ ,2 Rodrigo Otavio Lami Pereira,3 Andrew R Virata4 BMJ Case Rep: first published as 10.1136/bcr-2019-232183 on 2 February 2020. Downloaded from Unusual association of diseases/symptoms Case report Mesenteric panniculitis in a patient with new onset dermatomyositis Pankaj Bansal,1 Emily L Gilbert ,2 Rodrigo Otavio Lami Pereira,3 Andrew R Virata4 1Rheumatology, Mayo Clinic SUMMARY osteoarthritis of knees and hips, Bell’s palsy, gener- Health System, Eau Claire, Mesenteric panniculitis (MP), part of the spectrum alised anxiety disorder, irritable bowel syndrome Wisconsin, USA of sclerosing mesenteritis, is an often asymptomatic and osteoporosis with T12 compression fracture. 2Internal Medicine, Mayo disorder that is characterised by chronic inflammation Four months prior to rheumatologic evaluation, she Clinic Hospital Jacksonville, of abdominal mesentery. We present a case of an Jacksonville, Florida, USA noted a pruritic, erythematous rash on her face and 3Anesthesiology and 83- year- old woman who presented with proximal upper torso in a photodistribution (figure 1). She Perioperative Medicine - FLA, muscle weakness and erythematous, photosensitive was evaluated by a dermatologist and underwent Mayo Clinic, Jacksonville, rash of the face and upper torso and was subsequently skin biopsy showing vacuolar interface dermatitis Florida, USA diagnosed with dermatomyositis based on skin with increased dermal mucin, for which, she was 4 Pathology, Mayo Clinic Health biopsy, electromyography and muscle biopsy. She had prescribed topical corticosteroids (figures 2–4). System, Eau Claire, Wisconsin, radiographic evidence of panniculitis on CT scan of the A few weeks later, she started noticing proximal USA abdomen and pelvis for malignancy surveillance, which muscle weakness in both the upper and lower improved on serial CT scan 3 months after beginning extremities. Specifically, she had difficulty getting Correspondence to treatment for her underlying dermatomyositis with Dr Pankaj Bansal; up from a chair and used her hands to support prednisone and mycophenolate mofetil. Our case bansal. pankaj@ mayo. edu herself and complete the movement. Review of highlights that MP can be associated with underlying systems was specifically negative for shortness of Accepted 14 January 2020 autoimmune disease. Connective tissue disease could be considered in the differential of MP when other breath, cough, chest pain, dysphagia, odynophagia, etiologies such as surgery, trauma and malignancy are Raynaud’s, joint pain or swelling, fevers, night by copyright. ruled out. sweats, weight loss, abdominal pain, vomiting, or any new gastrointestinal complaints. Medical history was negative for any malignancies. Physical examination was remarkable for violaceous, edem- BACKGROUND http://casereports.bmj.com/ atous periorbital erythema with minimal overlying Mesenteric panniculitis (MP) is a rare disorder scaling. On her chest, a telangiectatic eruption was first recognised nearly a century ago involving seen in a photodistribution. Joint examination chronic fibrosing inflammation of the abdominal demonstrated Heberden's and Bouchard nodes mesentery.1 2 MP is part of a spectrum of conditions based on histological findings including mesenteric in the hands without tenderness or synovitis and lipodystrophy and retractile mesenteritis; collec- no synovitis in any other joint. Muscle exam- tively these are grouped under the umbrella term ination showed symmetric weakness (4/5) in the sclerosing mesenteritis (SM).3–7 Though many hip and thigh flexor and extensor muscles. Distal patients are asymptomatic, chronic inflammation of lower extremity and upper extremity strength was the mesentery can lead to non- specific symptoms preserved. She had difficulty getting out of a chair on March 30, 2020 at Weill Cornell Medical Library. Protected such as nausea/vomiting, abdominal pain, fever without support. and weight loss. While the aetiology of mesen- teric panniculitis is yet to be elucidated, it has been associated with surgery, trauma, cancer and less frequently with autoimmune diseases including Sjögren’s syndrome, systemic lupus erythematosus, ankylosing spondylitis, rheumatoid arthritis and IgG4 disease.1 4 6 8–13 However, MP has not been associated with inflammatory myopathies to the best of our knowledge. We report a case of MP incidentally discovered in a patient with new onset © BMJ Publishing Group dermatomyositis (DM). Limited 2020. No commercial re-use . See rights and permissions. Published by BMJ. CASE PRESENtatION An- 83- year old woman was referred to the rheuma- To cite: Bansal P, Gilbert EL, Pereira ROL, et al. BMJ Case tology department for a constellation of symptoms Rep 2020;13:e232183. including skin rash and muscle weakness. Medical Figure 1 83- year- old Caucasian woman with a doi:10.1136/bcr-2019- history was significant for obesity, hyperlipidemia, pruritic erythematous rash on her upper torso in a 232183 hypertension, asthma, obstructive sleep apnea, photodistribution. Bansal P, et al. BMJ Case Rep 2020;13:e232183. doi:10.1136/bcr-2019-232183 1 BMJ Case Rep: first published as 10.1136/bcr-2019-232183 on 2 February 2020. Downloaded from Unusual association of diseases/symptoms Figure 2 4 mm punch biopsy of the chest. H&E stain demonstrates superficial dermal lymphohistiocytic inflammation. Figure 4 4 mm punch biopsy of the chest. Low power (4×) colloidal INVESTIGatIONS iron stain demonstrates a prominent increase in dermal mucin. Initial investigations revealed elevation in creatine kinase at 998 U/L along with aldolase at 16 U/L. Serological work- up was positive for ANA 4.4 U, anti- Ds- DNA 81.7 IU/mL, and weakly scan of the chest, abdomen and pelvis. Both the CT chest scan positive MI-2. Rest of the myomarker panel, and serological and pelvic ultrasound were unremarkable. A suspicious lesion work- up including extractable nuclear antibodies panel was on mammography was biopsied and was negative for breast negative. C3 and C4 complement levels were normal, as were cancer. Colonoscopy showed multiple non- malignant polyps the basic chemistries, thyroid and parathyroid hormone levels scattered throughout the large bowel. Leukaemia and lymphoma and serum protein electrophoresis. Electromyography and nerve work- up were negative. CT scan of the abdomen/pelvis revealed by copyright. conduction study (EMG/NCS) showed low amplitude motor significant mesenteric stranding and thickening suggestive of responses and mixed motor unit potentials on needle examina- mesenteric panniculitis (figure 8). No evidence of underlying tion, suggestive of inflammatory myopathy. Right deltoid muscle malignancy was found. biopsy showed active inflammatory myopathy with necrotic and http://casereports.bmj.com/ regenerating fibres in addition to perivascular inflammation and OUTCOME AND FOLLOW-UP invasion of vessel wall by mononuclear inflammatory cells indi- The patient was initiated on prednisone 40 mg daily and myco- cating a vasculitic component (figures 5–7). Overall, the patient’s phenolate mofetil 3 g daily with goal to taper the prednisone clinical presentation including physical examination, laboratory off to maintain steroid free treatment regimen for long-term findings, EMG/NCS and skin and muscle biopsies confirmed the management of the dermatomyositis. She started showing clin- diagnosis of dermatomyositis. Cancer screening was pursued ical improvement within 1 month, and prednisone taper was including pelvic ultrasound, colonoscopy, mammogram, serum initiated. After 3 months, she had improved dramatically with protein electrophoresis, leukaemia/lymphoma panel, and CT on March 30, 2020 at Weill Cornell Medical Library. Protected Figure 5 Right deltoid muscle biopsy demonstrating active Figure 3 4 mm punch biopsy of the chest. Intermediate power inflammatory myopathy with a asculiticv component. Low power (2×) (10×) H&E stain demonstrates vacuolar interface inflammation with H&E stain demonstrates perivascular inflammatory exudate in the keratinocyte necrosis and thickening of the basement membrane. perimysium. 2 Bansal P, et al. BMJ Case Rep 2020;13:e232183. doi:10.1136/bcr-2019-232183 BMJ Case Rep: first published as 10.1136/bcr-2019-232183 on 2 February 2020. Downloaded from Unusual association of diseases/symptoms Figure 6 Right deltoid muscle biopsy demonstrating active inflammatory myopathy with a asculiticv component. ATPase stain demonstrates type 2 fibre atrophy. Figure 8 Axial CT image of the abdomen with oral and intravenous contrast demonstrates moderate fat stranding within the abdomen at the level of (A) mid abdomen, (B) aortic bifurcation and (C) lower normal muscle examination and normal muscle enzymes. She was abdomen. continued on mycophenolate mofetil 3 g daily, and we continued tapering prednisone from 10 mg daily dose. Repeat CT scan of as the ‘misty mesentery’ is extensive, and other etiologies must the abdomen and pelvis was performed which showed signifi- 16 cant improvement in the mid abdominal mesenteric fat stranding be excluded before MP is considered by the radiologist. Non- and panniculitis (figure 9). Nodularity and adenopathy of the Hodgkin’s lymphoma is one mimic; others include mesenteric mesenteric fat had improved significantly as well. She had close oedema, focal inflammation (appendicitis or diverticulitis for follow- up in the rheumatology clinic and internal medicine clinic example), other neoplasms and haemorrhage. It is worthy to by copyright. during this time, and no clinical, laboratory or imaging evidence note that many of these diagnoses present acutely and
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