Kas iskelet sistemi sendromları neoplastik hastalıklarla ilişkili olabilir. Bu yazıda, kliniğimizde sakroiliit tanısı alan ve takiplerde Hodgkin lenfoma tanısı koyduğumuz olguyu tanımladık. Sakroiliit tanısı manyetik rezonans görüntüleme ile de doğrulansa dahi sakroiliitle ilişkili olabilecek malignite tanısı gecikebilir ve bu durum klinik bir karışıklığa yol açabilir. Amacımız literatürü gözden geçirerek, malignite varlığında sakroiliak eklem tutulumun olası mekanizmasını tartışmaktır.Musculoskeletal syndromes may be associated with neoplastic Kas iskelet sistemi sendromları neoplastik hastalıklarla ilişkili olabilir. Bu diseases. We hereby report a case of sacroiliitis that was diagnosed as yazıda, kliniğimizde sakroiliit tanısı alan ve takiplerde Hodgkin lenfoma Hodgkin's lymphoma during the follow-up process. Though sacroiliitis was confirmed with magnetic resonance imaging (MRI), a diagnosis of malignancy associated with sacroiliitis may be delayed and cause a clinical challenge. We aimed to discuss the presentation of lymphoma with sacroiliac joint involvement and its probable mechanisms and review the literature

Akçay Yalbuzdağ, Şeniz

Erol, Asiye Mukaddes

Karabulut, Banu

Sarıfakıo?lu, Ayşe Banu

Çelik, Canan

Türkiye Fiziksel Tıp ve Rehabilitasyon Dergisi

Namik Kemal University Institutional Repository

Case Report / Olgu SunumuDOI: 10.5152/tftrd.2014.56254Turk J Phys Med Rehab 2014;60:353-5Türk Fiz T›p Rehab Derg 2014;60:353-5Hodgkin’s Lymphoma: Case Report of Mimicking SacroiliitisSakroiliiti Taklit Eden Hodgkin Lenfoma: Olgu SunumuŞeniz AKÇAY YALBUZDAĞ1, Asiye Mukaddes EROL1, Canan ÇELİK1, Ayşe Banu SARIFAKIOĞLU2, Banu KARABULUT31Department of Physical Medicine and Rehabilitation, Bursa Şevket Yılmaz Education and Research Hospital, Bursa, Turkey2Department of Physical Medicine and Rehabilitation, Namık Kemal University Faculty of Medicine, Tekirdağ, Turkey3Burtom Radiology Screening Center, Bursa, TurkeyAbstractMusculoskeletal syndromes may be associated with neoplastic diseases. We hereby report a case of sacroiliitis that was diagnosed as Hodgkin’s lymphoma during the follow-up process. Though sacroiliitis was confirmed with magnetic resonance imaging (MRI), a diagnosis of malignancy associated with sacroiliitis may be delayed and cause a clinical challenge. We aimed to discuss the presentation of lymphoma with sacroiliac joint involvement and its probable mechanisms and review the literature. Key Words: Sacroiliitis, Hodgkin’s lymphoma, low back painÖzetKas iskelet sistemi sendromları neoplastik hastalıklarla ilişkili olabilir. Bu yazıda, kliniğimizde sakroiliit tanısı alan ve takiplerde Hodgkin lenfoma tanısı koyduğumuz olguyu tanımladık. Sakroiliit tanısı manyetik rezonans görüntüleme ile de doğrulansa dahi sakroiliitle ilişkili olabilecek malignite tanısı gecikebilir ve bu durum klinik bir karışıklığa yol açabilir.  Amacımız literatürü gözden geçirerek, malignite varlığında sakroiliak eklem tutulumun olası mekanizmasını tartışmaktır.  Anahtar Kelimeler: Sakroiliit, Hodgkin lenfoma, bel ağrısıAddress for Correspondence / Yazışma Adresi: Şeniz Akçay Yalbuzdağ, MD, Department of Physical Medicine and Rehabilitation, Bursa Şevket Yılmaz Education and Research Hospital, Bursa, Turkey. Phone: +90 242 595 50 00  E-mail: senizakcay@hotmail.comReceived/Geliş Tarihi: August/Ağustos 2013  Accepted/Kabul Tarihi: December/Aralık 2013©Telif Hakkı 2014 Türkiye Fiziksel Tıp ve Rehabilitasyon Derneği - Makale metnine www.ftrdergisi.com web sayfasından ulaşılabilir. ©Copyright 2014 by Turkish Society of Physical Medicine and Rehabilitation - Available online at www.ftrdergisi.com 353IntroductionMusculoskeletal syndromes, such as hypertrophic osteoarthrop-athy, carcinoma polyarthritis, dermatomyositis/polymyositis, and paraneoplastic vasculitis, fasciitis, panniculitis, erythema nodosum, Raynaud’s syndrome, digital gangrene, erythrome-lalgia, and lupus-like syndromes, may be associated with neo-plastic diseases (1). Although the association of malignancy and rheumatologic disorders is well described in the literature, only few cases have reported sacroiliitis as an initial presentation of lymphoproliferative disorder.In this report, we describe a patient with sacroiliitis and diag-nosed with Hodgkin’s lymphoma 6 weeks after admission to our clinic.Case ReportA 31-year-old man presented with right hip and buttock pain and left hip pain of 3 weeks in duration. The pain began acutely, initially involving the right side and involving the right buttock. He described marked morning stiffness lasting for 2 hours. In the course of this period, he was admitted to the emergency service of our hospital several times, and nonsteroidal anti- inflammatory and myorelaxant drugs were suggested, and he was referred to the physical medicine and rehabilitation out-patient clinic. He denied experiencing any symptoms of fever, weight loss, peripheral joint pain, skin lesions, urethritis, con-junctivitis, and venereal or gastrointestinal disease. The family history was negative for rheumatic diseases. He had no smoking history. In the physical examination, right sacroiliac compression and Gaenslen tests were positive. Sacroiliac joint was exquisitely painful to direct pressure. The cervical, thoracic, and lumbar spine and peripheral joint examination was unremarkable. In the laboratory examination, serum C-reactive protein (CRP) was 109.9 mg/L (normal range 0-5 mg/L), hemoglobin was 10.7 g/dL (normal range 12-15.5 g/dL), erythrocyte sedimenta-tion rate (ESR) was 105 mm/h (normal range 0-20 mm/h), and white blood cell count was 10,600/mm3 (normal range 3500-10,500/mm3). The patient was HLA-B27-negative. A plain radio-graph of the sacroiliac joint revealed minimal sclerotic changes in the iliac region of the right sacroiliac joint.Magnetic resonance imaging of the sacroiliac joint showed bone marrow edema of the iliac bone and sacroiliac joint and gadolinium enhancement with hyperintense areas that may be associated with sacroiliitis (Figure 1). The patient initially received sulfasalazine 1000 mg twice a day and indomethacin perorally 25 mg 3 times a day. At the first follow-up after 2 weeks, the patient’s clinical response to ther-apy was excellent. There was no pain or morning stiffness. ESR was still 97 mm/h and CRP was 44.3 mg/L at this time. After 3 weeks, the patient experienced fatigue and detected multi-ple 1-2 cm non-tender cervical and axillar lymph nodes in the physical examination. The liver and spleen were not enlarged. Abdominal ultrasonography revealed multiple hypoechoic, reg-ular-shaped solid lesions in the liver and spleen. An abdominal MRI confirmed hyperintense heterogeneity with contrast mate-rial in the liver and spleen that may be associated with lym-phoproliferative disorder or granulomatous disease. Excisional biopsy of the cervical lymph node showed Hodgkin’s lymphoma nodular sclerosis subtype. He was referred to the hematology clinic to receive a chemotherapy program. Positron emission to-mography (PET), performed in the hematology clinic, revealed hypermetabolic areas in the right iliac bone, which may be as-sociated with bone marrow involvement. Also, multiple hyper-metabolic areas in the cervical, supraclavicular, mediastinal, and infradiaphragmatic lymph nodes and a hypermetabolic area in right fourth costa that may be associated with metastatic bone lesions were reported. DiscussionHereby, we reported a patient with low back pain and diag-nosed as sacroiliitis, and the cervical and axillar lymph nodes directed with to perform advanced research with a suspicion of malignancy. We also aimed to emphasize the importance of a malignancy mimicking rheumatic conditions and reviewed the literature. Although musculoskeletal disorders may be associated with neo-plastic disease, an unusual presentation of a malignancy may be a cause of the delayed diagnosis of disease. Primary synovial neoplasm or metastasis to the bone or synovium may be the reason of musculoskeletal complaints. Periarticular metastases are not infrequent and may present as acute arthritis. These are neoplastic lesions of the bone without inclusion of the joint or deterioration of the joint because of metastases and non-neo-plastic reactions in the adjacent joint and neoplastic invasion of the joint (2). In addition to the paraneoplastic phenomenon, the unusual presentation of malignancies mimicking rheumatic disorders has been a clinical challenge and may be the cause of the delayed diagnosis of the main disease. In the literature, there are 4 cases of sacroiliitis associated with hematologic malignancy. One of them was acute lymphoblastic leukemia, in which a complete blood count revealed pancyto-penia (3). Our patient’s complete blood count was unremark-able, and the clinical response to medication was excellent. He had no complaint at the first follow-up, but he presented with constitutional symptoms, like fatigue and night sweats, and the physical examination determined lymphadenomegaly in the cervical chain at the next follow-up. Although there was an im-provement in low back pain of our patient’s, serum ESR and CRP levels were still high. These findings directed us to perform fur-ther diagnostic techniques. Two cases have been reported with sacroiliitis who were diagnosed with Hodgkin’s disease 12 and 5 months after sacroiliitis, respectively. The interval between our patient’s sacroiliac joint pain and the exact diagnosis of Hodgkin lymphoma was shorter than the previous report, because the constitutional symptoms and lymph nodes occurred rapidly (4). In a case report, sacroiliitis and enthesopathies developed due to diffuse, well-differentiated lymphocytic lymphoma (5).However, the initial presentation of our patient was similar to sacroiliitis: ESR level was over 100 mm/h and remained high despite non-steroidal anti-inflammatory drug and sulfasalazine medication.The etiology of sacroiliitis may be classified into inflammatory and non-inflammatory conditions. In particular, secondary car-cinoma must take part in this list (2). The presentation of the patient was similar to spondyloarthritis, including ankylosing spondylitis, reactive arthritis, psoriatic arthritis, and enteropathic arthritis. We determined a high level of ESR, which was sus-picious for inflammatory sacroiliitis. If ESR and CRP levels are higher than expected, the clinician must check out the initial diagnosis. When the expected clinical response is not achieved, with the persistence of symptoms, the diagnosis must be recon-sidered. Also, constitutional symptoms may direct the clinician for an advanced examination with suspicion of malignancy. The histopathologic diagnosis may be surprisingly different; however, an MRI indicating edema of the sacroiliac joint seems to meet the definition of sacroiliitis issued by the Assessment of the Spondyloarthritis International Society (ASAS). Bereau et al. (6) reported a case with sacroiliitis according to an MRI study that was diagnosed with Hodgkin’s disease with radioguided biopsy of the iliac joint. Rheumatic syndromes associated with paraneoplastic syn-dromes include rheumatoid arthritis, Sjogren’s syndrome, sys-temic sclerosis, temporal arteritis, carcinoma polyarthritis, der-matomyositis, lupus erythematosus, and vasculitis. Sacroiliitis is not defined as a paraneoplastic syndrome. According to our 354Akçay Yalbuzdağ et al.Hodgkin Lymphoma Mimicking SacroiliitisAkçay Yalbuzdağ et al.Hodgkin Lymphoma Mimicking Sacroiliitisfindings, we do not consider the sacroiliac pain of our patient as a paraneoplastic phenomenon but the direct involvement of the iliac bone by lymphoma mimicking unilateral sacroiliitis. Even though we could not confirm the sacroiliac joint involvement by Hodgkin lymphoma with the histopathological findings, the PET results confirmed our hypothesis. Also, our report is similar to previous reports: one of them was of sacroiliac joint involvement of metastatic adenocarcinoma, and the other one was prostatic carcinoma mimicking sacroiliitis (2,7). The self-criticism of this report is that the histopathological sample should be taken dur-ing the initial visits with a suspicion of malignancy even a well clinical response achieved. ConclusionIn rheumatic syndromes, fever; weight loss; thromboembolic events; and neurological, endocrinological, gastrointestinal, or cutaneous disorders may be the signs of underlying neoplastic disease (8). In patients with rheumatic syndromes, ESR, com-plete blood cell count, and serum chemistries are almost always obtained during the routine evaluation. In respect to laboratory findings, elevated ESR and anemia may both present in malig-nancy and primary rheumatic syndromes. Both of these findings have limited value for the differential diagnosis between cancer-associated disorders and primary rheumatic diseases (9). More detailed investigations with a suspicion of malignancy are not recommended initially if there are no specific findings that are suggestive of malignancy. Therefore, the clinician must recog-nize the clues that may be the features of a malignancy associ-ated with rheumatic syndromes.In conclusion, sacroiliitis may be the consequence of a malig-nancy with sacroiliac joint involvement; however, morning stiff-ness and good response to non-steroidal anti-inflammatory drug treatment accompany the sacroiliac pain. The physician must be aware of a malignancy with elevated serum ESR levels over 100 mm/h, despite a clinical response achieved with medication. An unusual presentation of malignancies mimicking rheumatologic disorders can mask the exact diagnosis of the disease. Informed Consent: Written informed consent was obtained from pa-tient who participated in this case. Peer-review: Externally peer-reviewed. Author Contributions: Concept - Ş.A.Y.; Design - Ş.A.Y., A.M.E., A.B.S.; Supervision - C.Ç.; Funding - Ş.A.Y., A.M.E.; Materials - Ş.A.Y., A.M.E.; Data Collection and/or Processing - Ş.A.Y., A.M.E.; Analysis and/or Inter-pretation - A.B.S., Ş.A.Y., B.K.; Literature Review - Ş.A.Y.; Writer - Ş.A.Y.; Critical Review - C.Ç.; Other - C.Ç., A.B.S.Conflict of Interest: No conflict of interest was declared by the authors.Financial Disclosure: The authors declared that this study has received no financial support.Hasta Onamı: Yazılı hasta onamı bu olguya katılan hastadan alınmıştır. Hakem değerlendirmesi: Dış bağımsız.Yazar Katkıları: Fikir - Ş.A.Y.; Tasarım - Ş.A.Y., A.M.E., A.B.S.; Dene-tleme - C.Ç.; Kaynaklar - Ş.A.Y., A.M.E.; Malzemeler - Ş.A.Y., A.M.E.; Veri toplanması ve/veya işlemesi - Ş.A.Y., A.M.E.; Analiz ve/veya yorum - A.B.S., Ş.A.Y., B.K.; Literatür taraması - Ş.A.Y.; Yazıyı yazan - Ş.A.Y.; Eleştirel İnceleme - C.Ç.; Diğer - C.Ç., A.B.S.Çıkar Çatışması: Yazarlar çıkar çatışması bildirmemişlerdir.Finansal Destek: Yazarlar bu çalışma için finansal destek almadıklarını beyan etmişlerdir.References1. Fam AG. Paraneoplastic rheumatic syndromes. Baillieres Best Pract Res Clin Rheumatol 2000;14:515-33. [CrossRef]2. Humphrey SM, Inman RD. Metastatic adenocarcinoma mimicking unilateral sacroiliitis. J Rheumatol 1995;22:970-2. 3. Moghadam A, Talebi-Taher M, Dehghan A. Sacroiliitis as an initial presentation of acute lymphoblastic leukemia. Acta Clinica Belgica 2010;65:197-9. [CrossRef]4. Saviola G, Abdi Ali L, Trentanni C, Notarangelo LD, Desiati F, Lupi E, et al. Sacroiliitis as a manifestation of Hodgkin’ disease in young females. Clin Exp Rheumatol 2003;21:270. 5. Cohen MR, Carrera GE, Lundberg J. Rapidly progressive sacroi-liitis in a patient with lymphocytic lymphoma. Ann Rheum Dis 1993;52:239-40. [CrossRef]6. Bereau M, Prati C, Wendling D. Sacroiliac edema by MRI does not always indicate spondylarthritis. Joint Bone Spine 2011;78:646. [CrossRef]7. Manghani M, Loneragan R, Barnsley L. Metastatic prostatic carcino-ma mimicking a unilateral sacroiliitis. Australas Radiol 2007;51:92-4. [CrossRef]8. Naschitz JE, Rosner I, Rozenbaum M, Zuckerman E, Yeshurun D. Rheumatic syndromes: clues to occult neoplasia. Semin Arthritis Rheum 1999;29:43-55. [CrossRef]9. Staszaewski H. Hematological paraneoplastic syndromes. Semin Oncol 1997;24:329-33.355Figure 1. Bone marrow edema at the sacroiliac joint by magnetic resonance imaging 

Hodgkin's Lymphoma: Case Report of Mimicking Sacroiliitis

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