Extranodal Rosai-Dorfman disease presenting as an isolated glenoid lesion in a high school athlete Andrew R. Hsu, MD a, *, Sanjeev Bhatia, MD a , Richard W. Kang, MD a , Leonidas Arvanitis, MD b , Greg P. Nicholson, MD a , Walter W. Virkus, MD a a Department of Orthopaedic Surgery, Rush University Medical Center, Chicago, IL, USA b Department of Pathology, Rush University Medical Center, Chicago, IL, USA Rosai-Dorfman disease (RDD), also known as sinus his- tiocytosis with massive lymphadenopathy (SHML), was first described as a clinical entity by Rosai and Dorfman in 1969. 15 The first description of the disease reported a rare non- neoplastic disorder involving the proliferation of histiocytes predominantly involving the lymph nodes. ‘‘Sinus histiocy- tosis’’ refers to histiocytosis that occurs in the distended sinuses of lymph nodes, but since its initial description, RDD has been reported to involve a wide variety of organ systems and has been found in numerous locations throughout the body. 6 RDD most commonly presents in young patients in the second or third decade of life, with an increased prevalence in men. 6 RDD generally presents as bilateral, nontender, painless adenopathy in the neck, which may be accompanied by fever, elevated erythrocyte sedimentation rate, weight loss, and immunologic abnormalities such as leukocytosis and Q1 polyclonal hypergammaglobulinemia. Less frequently involved nodal sites are mediastinal, hilar, retroperitoneal, axillary, and inguinal. 3,4,12,18 Extranodal RDD occurs in 42% of patients, with 23% having isolated extranodal disease. 6 Of the nearly 1000 patients with documented RDD, 3,4,18 less than 3% have presented with isolated osseous involve- ment. 6 In the 423 RDD cases presented by Foucar et al, 6 7.8% had osseous involvement, 2% had osseous involvement without lymphadenopathy, and 0.5% had isolated osseous involvement. The skull is the most commonly cited location of solitary bone lesions and often presents with pain and headaches. 1 In another series by Sundaram et al, 19 15 patients had isolated bone lesions and 3 had multiple bone lesions as the sole presentation of RDD. Pain at the involved site was the most commonly cited symptom among patients. In this case report we present a 16-year-old male patient with an extranodal lesion within the glenoid found incidentally and misdiagnosed as osteomyelitis and later Langerhans cell his- tiocytosis before finally being diagnosed as RDD through immunohistochemical staining. A primary isolated extranodal intraosseous lesion of the glenoid is an extremely rare presen- tation of RDD and this is the only report in the literature to date of this presentation. We obtained informed written consent from the patient’s guardian for publication of this report. Case report A 16-year-old African American high school football player presented to his primary care provider 1 week after sustaining a mild impact injury to the anterior portion of his right shoulder during practice. The patient reported pain with overhead activities and associated weakness. There was no numbness or tingling or any radiation of pain down the affected arm. His medical history was positive only for attention deficit hyperactivity disorder treated with methylphenidate. On examination, there was no overlying skin erythema, warmth, or swelling. Range of motion testing showed forward flexion was 180  with a painful arc of motion during the last 40  , abduction to 160  with pain, external rotation with the elbow at the side to 35  with pain, and internal rotation to L1 with anterior shoulder pain. Investigational Review Board approval is not required for a case report. *Reprint requests: Andrew R. Hsu, MD, Department of Orthopaedic Surgery, Rush University Medical Center, 1611 W Harrison St, Ste 200, Chicago, IL 60612, USA. E-mail address: andyhsu1@gmail.com (A.R. Hsu). J Shoulder Elbow Surg (2011) -, e1-e6 www.elsevier.com/locate/ymse SCO 5.1.0 DTD  YMSE2036_proof  11 July 2011  6:26 pm  ce Ln 1058-2746/$ - see front matter Ó 2011 Journal of Shoulder and Elbow Surgery Board of Trustees. doi:10.1016/j.jse.2011.05.017 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56 57 58 59 60 61 62 63 64 65 66 67 68 69 70 71 72 73 74 75 76 77 78 79 80 81 82 83 84 85 86 87 88 89 90 91 92 93 94 95 96 97 98 99 100 101 102 103 104 105 106 107 108 109 110