CASO CLÍNICO Atypical localization of rheumatoid nodule: case report Bayram KB1, Guvendi EU1, Gurgan A1, Kocyigit H1, Askin A1, Rezanko T2 ACTA REUMATOL PORT. 2015;40:81-84 ABSTRACT between 45 and 88% in post-mortem studies4,5. Laryn- goscopic examination can demonstrate mucosal oede- Rheumatoid nodules can be seen in about 30% of pa- ma, hyperaemia, swelling of the arytenoids, intrinsic tients with rheumatoid arthritis. They are occasionally laryngeal muscle myositis, epiglottitis, crycoarytenoid localized subcutaneously, but they can rarely be seen in joint (CJ) involvement, reduction in mobility of the vo- visceral organs. Their appearance can be confused with cal cords and vocal cord rheumatoid nodules4-7. If an many clinical conditions when they have atypical lo- atypically localized space-occupying lesion detected in calizations. To exclude the presence of a malignancy, patients with complaints like throat ache, hoarseness; these lesions should always be investigated. We aimed primarily malignancy and benign laryngeal lesions are to discuss a patient with rheumatoid nodule localized considered, a rheumatoid nodule may not come to in close neighbourhood of hyoid bone, presumed as a mind8. This can cause confusion, misdiagnosis and ex- malignancy. posure of patients to unnecessary tests. In this case we aimed to discuss the case of a patient with a rheuma- Keywords: Hyoid bone; Malignancy; Rheumatoid no - toid nodule loca lized in close neighbourhood of the hy- dule; Mass. oid bone, that had presumed malignancy and to per- forme a review of the literature. INTRODUCTION CASE REPORT Rheumatoid arthritis (RA) is a chronic autoimmune di - sease with unknown aetiology, which is characterized Forty-nine years old female patient with a diagnosis of by peripheral symmetrical polyarthritis, typically with RA for 25 years, admitted to our clinic due to an in- joint and bone erosion that leads to deformity and des - crease in complaints of wide -spread joint pain and truction1. Most common extra-articular involvement in morning stiffness. After diagnosis, the patient had been patients with RA are subcutaneous rheumatoid no - treated with hydroxychloroquine, sulfasalazine, dules2. Subcutaneous nodules are usually seen on the leflunomide and corticosteroid during short periods. extensor surface of the proximal ulna and in areas She had not been receiving any treatment in last 2 years subjec ted to pressure such as sacrum, Achilles tendon, and had dificulties in performing everyday activities occipital area and hand tendons. Visceral rheumatoid due to the weakness, fatigue and pain. She referred dys- nodu les are detected in areas such as lung, heart, la - phagia in swallowing, sore throat and hoarseness for rynx and vocal cords3. four months. She did not describe mouth and eye dry- The prevalence of laryngeal involvement in RA ness, rashes, aphthous lesions, diarrhea, abdominal ranges from 13 to 75% in various clinical studies, and pain or an infection recently. She had no previous his- tory of surgery or major trauma and hadn’t been used any other drugs for a systemic di sease. 1. Physical Medicine and Rehabilitation, Katip Celebi University On physical examination the patient’s body tempera - Ataturk Training and Research Hospital; o 2. Pathology, Katip Celebi University Ataturk Training and ture was 36,1 C; blood pressure 120/70 mmHg; heart Research Hospital rate 80 beats per minute. Examination of locomotor ÓRGÃO OFICIAL DA SOCIEDADE PORTUGUESA DE REUMATOLOGIA 81 ATYPICAL LOCALIZATION OF RHEUMATOID NODULE: CASE REPORT gative. In neck MRI lingular tonsil volumes had increased. A tissue that had the same signal characteristics as thy- roid gland was discovered at the right of hyoid bone, between strep muscles. The patient was referred to the endocrinology clinic and an USG guided FNA biopsy was performed to the mass. The cytological exa - mination of the obtained smears revealed a large num- ber of isolated atypical cells with eosinophilic, vacuo- lated or clear cytoplasm, ‘’histiocytic’’ cells, some of them were suggestive for signet-ring cells, in a amor- phous eosinophilic material background (Figure 1). Mitotic figure and anisonucleosis was observed in some cells. A small number of multinucleated giant FIGURE 1. Hypercellular smear shows a predominantly isolated cell histiocytes were also seen. Cytomorphological single dispersed cell population. Cytoplasm is mostly finely findings were primarily considered as a neoplasm with vacuolated or ‘’ histiocyte-like’’. Some cells have eccentric nuclei and abundant eosinophilic cytoplasm, mitotic figure a cystic component and metastatic carcinomas (signet (arrow). Sparse multinucleated cells were also observed. There ring or renal cell carcinoma) were also suspected. is an isonucleosis in some cells and a signet ring-like cell Clini cal, radiological examination and excision of mass (arrow, inset) (Hematoxylin&Eosin X200) for definitive diagnosis was recommended. The biopsy results were discussed at the Otolaryngolo gy, Urology and General Surgery clinics. system revealed tenderness in the joints of the hands There were no malignancy findings in gastroscopy and and feet, bilateral swelling of the 1st and 2nd meta - colonoscopy. No pathological findings were detected carpal joint and bilateral Z deformity of the thumb. In in computed tomography (CT) of all abdomen and neck palpation, a hard mobile 2x2 cm lymphade no - high-resolution CT of lung. Ear, nose and throat (ENT) pathy was discovered on the left side, about the level Clinic performed a biopsy with flexible laryngoscopy. of C3. Rheumatoid nodu l es were present on the right In pathological exa mination a cell organization that olecranon. No further changes were observed on exa - may accompany inflammatory cystic neoplasm was mination. observed. Total excision of the mass was recommend- The basic laboratory tests including complete blood ed. Pathological exa mination of the excisional biopsy count, erythrocyte sedimentation rate (ESR), blood showed features of a rheumatoid nodule (Figures 2 biochemistry findings, rheumatoid factor (RF), liver and 3). The patient’s medical treatment was planned function tests and urinary analysis were within nor- and scheduled a follow-up in our rheumatic diseases mal limits. Disease activity score (DAS-28) was calcu- clinic. lated as 4.41. Upon detection of thyroid-stimulating hormone (TSH): 5.65 uIU/ml (0.35-5.50), FT3: 2.24 pg/ml (2.3-4.2), FT4: 1.2 ng/dL (0.88-1.72) thyroid DISCUSSION ultrasonography (USG) and thyroid antibody tests were planned. In USG slight coarsening was observed Rheumatoid nodules are seen in approximately 30% of in the parenchyma of the thyroid gland but no nodules RA patients. They can be detected on the compression detected. A mass lesion about 13x9.33 mm was loca - exposed areas like elbows, forearms, fingers, tendons, ted posterior to the hyoid bone. The lesion was he - sacrum, heel bone and visceral organs (liver, kidney, terogeneous, hypoechoic and its borders were indis- lung, heart), larynx, pharynx, trachea, vocal cords, pe- tinguishable. In differential diagnosis necrotic lymph nis, vulva and breast3,7,9-13. Most nodules are node and complicated cystic lesion was considered. asymptoma tic, but those located under pressure areas Magnetic resonance imaging (MRI) examination, thy- such as periosteum or tendons, can be painful2,3. No - roid scintigraphy and fine-needle aspiration biopsy dules are more common in RA patients who have RF (FNA) was planned. Diffuse hyperplasia was found in and anti-cyclic citrullinated peptide (anti-CCP) posi- thyroid scintigraphy. Thyroid autoantibodies were ne - tivity, bone erosions and extra articular manifestations. ÓRGÃO OFICIAL DA SOCIEDADE PORTUGUESA DE REUMATOLOGIA 82 BAYRAM KB ET AL FIGURE 3. Rheumatoid nodule: nodular lesion infiltrating the striated muscle; fibrin deposition and necrobiosis is seen in centre of nodule surrounded by histiocytes, proliferated FIGURE 2. The mass-forming diffuse cellular infiltration vascular endothelial cells, lymphocytes, plasma cells and nearby hyoid bone and muscle tissue (Hematoxylin&Eosin occasional giant cells (Hematoxylin&Eosin X40, inset X100) X40) Larynx rheumatoid nodules may present with many No dules are gene rally seen in long-term disease3. Also, symptoms. Dyspnoea, stridor, hoarseness, fullness in increased rheumatoid nodule formation has been re- the throat and pain in swallowing are some of ported with methotrexate treatment14. them5,12,13. In patients with RA if one of these findings The pathogenesis of rheumatoid nodules is not suggesting larynx involvement are present, radio- clearly understood. However, has been hypothesized graphs may be used for showing most common in- that trauma to pressure points on the body, with sub- volvement erosive CJ arthritis. However CT is consi - sequent small haemorrhages, results in accumulation dered the method of choice. The main findings that of RF complexes at the site of the injury. RF comple - include CJ thickening or erosions, arytenoid subluxa- xes have a chemotactic role in immune response by tion and asymmetry of the glottis or aryepiglottic folds activating local monocytes and developing local vas- can be better displayed by CT4,16. We primarily exa - culitis. Involvement of T and B-lymphocytes increa ses mined our patient with a neck MRI, because of sus- the immune response15. This explains the formation of pected cystic or necrotic lymph node component of rheumatoid nodules also in the internal organs and in the mass lesion.