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OSTEOID OSTEOMA IN A YOUNG ATHLETE--A CASE REPORT FROM THE PERSPECTIVE OF A PRIMARY HEALTH CARE PEDIATRICIAN: OSTEOID OSTEOMA KOD MLADOG SPORTISTE--PRIKAZ SLUCAJA IZ UGLA PEDIJATRA U PRIMARNOJ ZDRAVSTVENOJ ZASTITI.

Introduction

Osteoid osteoma (OO) is a benign osteoblastic tumor that was first described as a separate entity by Jaffe in 1935 [1]. It is usually of 1.5 - 2 cm in diameter and characterized by well demarcated central area (osteoid nidus) in a highly loose, well vascularized connective tissue. Surrounding the nidus is the zone of sclerotic bone and the central part of the nidus is often calcified.

This tumor does not have the potential for distant metastases but can lead to functional and esthetic deficits [2, 3]. About 50% of lesions occur in the proximal meta-diaphysis of the long bones, most commonly femur or tibia [4, 5]. OO accounts for 10% of all symptomatic benign bone tumors and 5% of all bone tumors. It mostly occurs between 10 and 30 years of age and affects males 2 to 4 times more often than females [2, 3]. The main symptom is pain that worsens at night and is relieved by non-steroidal anti-inflammatory drugs (NSAIDs). The exact mechanism of pain is unknown but increased production of prostaglandins and increased nerve fiber density are found in the nidus [6]. In particular, prostaglandin E2 (PGE 2) is increased up to 1.000 times in comparison with the normal bone and is presumed to be the cause of severe pain seen in patients with lesions [7]. Strong expression of cyclooxygenase-1 (COX-1) and cyclooxygenase-2 (COX-2) in the osteoblasts within OO suggests an important role of COX in PGE production, thus explaining good response to NSAIDs [8].

Case Report

We report a case of an 11-year-old boy who visited the Pediatric Outpatient Clinic, part of the Primary Health Care Center complaining about pain in the right thigh in the past 3 months. He started complaining about the pain after a basketball training which he practiced regularly for 3 years, 3 times a week for 1.5 hours. His medical history showed that he had an annual sports physical exam by a pediatrician a month before the first symptoms occurred, without abnormalities at physical examination, electrocardiogram (ECG) and routine laboratory at that time, normal height of 150 cm and weight of 39 kg, and body mass index (BMI) of 17.33 kg/[m.sup.2]. Initially, the pain was nonspecific, occurred occasionally and lasted for a short time, but sometimes woke him up at night. During the day he felt pain only if he received an accidental kick in the right foot, even if it was of low intensity (unintentionally, passing by). One month after the pain started, he stopped training, and even walking became difficult. His appetite decreased. The NSAIDs relieved the pain, but for a short time, especially at night. Two weeks before seeking medical attention, the pain worsened at night and sometimes he had to take NSAIDs twice a night. He denied any history of trauma or fever. At the first examination by his pediatrician, he was cautious when walking, pale, and felt severe pain on right thigh palpation (pain was evaluated as 7/8 on the Wong Baker Faces Pain Rating Scale). There was no leg deformity, no local swelling, and the skin above the painful area was normal. The X-ray of the right femur in two directions was performed and showed a homogeneous shadow of 80 x 20 mm in the proximal and medial part of the right femur body. In the upper third part of the described shadow an illumination of 10 mm x 3 mm was seen (Figure 1). OO was suspected and the patient was referred to an orthopedist.

The orthopedic physical exam revealed a muscle atrophy of the right thigh and a difference between the right and left thigh muscle volume of 1.5 cm. Hospitalization was indicated by the orthopedist. Computed tomography (CT) of the right thigh confirmed a nidus and open surgical resection under general anesthesia was performed 2 weeks after the pediatrician's examination. The patient recovered rapidly and was discharged after 7 days of hospitalization. Weight bearing and physical activities were restricted. The patient was able to walk with crutches with the affected foot touching the ground. Five days after discharge, the pain was completely relieved and he stopped taking NSAIDs. Exercises for muscle strengthening progressively improved the range of motion through eight weeks and he was gradually involved in sports activities afterwards. The histopathological analysis confirmed the diagnosis of OO (dg. Neoplasm benignum ossium longorum extremitatis inferioris osteoid osteoma) barely 4 months after the onset of pain. After 12 months of follow up, no local recurrence was observed and the difference between the right and left thigh volume was less than 1 cm. Nine months after surgery and 13 months since the routine pediatric visit, the patient had normal height and weight (155 cm, 41 kg, BMI 17.07 kg/[m.sup.2]).

Discussion

Osteoid osteoma is a benign tumor that commonly affects male adolescents and young adults. The proximal femur and tibia are the commonest locations of OO. The typical clinical presentation is nocturnal pain that responds well to NSAIDs. However, the pain is associated with various disorders which is one of the reasons why OO may be misdiagnosed and correct diagnosis delayed (median 16 months, range 8--36 months) [9, 10]. In children, OO may be mimicking different diseases such as infantile cortical hyperostosis, osteomyelitis, Perthes disease, leg length discrepancy, healing stress fractures, tuberculosis, neuromuscular conditions, as well as malignant tumors (osteosarcoma, Ewing sarcoma) [11]. The diagnosis of OO can be a challenging process especially in young athletes. The etiologic factors may be the overuse injury and muscle strain. Some reports present similar clinical cases in athletes with femoral neck stress fractures and intra-articular OO of the femoral neck that may further delay the correct diagnosis [12]. Other unusual localizations, such as metacarpal, hallux, talus or intra-articular are also associated with delayed diagnosis or misdiagnosis of OO [13-16]. Primary health care pediatricians always have a dilemma whether to perform an X-ray of the painful area or not. Radiography is sufficient for establishing the diagnosis of OO if the nidus is clearly expressed, as in this case, so the initial diagnosis was confirmed barely 4 months after the onset of pain. However, there are vague, more calcified cases where nidus overlaps with the surrounding bone sclerosis on the

X-rays. Even in case of clear radiographic findings, CT is a gold standard in the preoperative diagnosis of OO [17]. The therapeutic approach depends on the localization, as well as on the severity of symptoms or a potential deformity. NSAIDs may sometimes lead to withdrawal of symptoms and resolution of the tumor [18]. Unsuccessful treatment with NSAIDs or intolerance to analgesics leads to surgical treatment. Open surgical resection is gold standard, but nowadays, minimally invasive surgical methods, such as percutaneous radiofrequency ablation and percutaneous excision, are also becoming important modalities [19, 20]. Maric et al. showed that modified percutaneous techniques provide fast recovery after the procedure and significantly decrease length of hospital stay compared with traditional surgical methods (2,43 [+ or -] 0,53 versus 10 [+ or -] 7.79 days) [21, 22]. However, even after complete nidus removal, anatomical changes and sequelae may progress and become symptomatic, so long-term follow-up is therefore essential [23]. Histopathological analysis sets the final diagnosis, and as in our case, confirming the clinical and imaging findings. Removal of nidus leads to resolution of pain, so except during the short post-operative period, analgesia is no longer needed, which was also the case in our patient [24].

Conclusion

In differential diagnosis of chronic leg pain in patients under the age of 18 years, primary health care pediatricians rely on medical history and physical examination, whereas after that plain radiography of the painful area should be considered. If the nidus is clearly expressed, the possibility of diagnosing osteoid osteoma timely and providing adequate treatment increases. In the presented case, the correct diagnosis was confirmed barely 4 months after the onset of pain.

References

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(4.) Lenke LG, Sutherland CJ, Gilula LA. Osteoid osteoma of the proximal femur: CT-guided preoperative localization. Orthopedics. 1994;17(3):289-92.

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(7.) Greco F, Tamburrelli F, Ciabattoni G. Prostaglandins in osteoid osteoma. Int Orthop. 1991;15(1):35-7.

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(10.) Turkmen I, Alpan B, Soylemez S, Ozkan FU, Unay K, Ozkan K. Osteoid osteoma of the great toe mimicking osteomyelitis: a case report and review of the literature. Case Rep Orthop. 2013;2013:234048 .

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(12.) Cordova CB, Dembowski SC, Johnson MR, Combs JJ, Svoboda SJ. Osteoid osteoma of the femoral neck in athletes: two case reports differentiating from femoral neck stress injuries. Sports Health. 2016;8(2):172-6.

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(14.) Xarchas KC, Kyriakopoulos G, Manthas S, Oikonomou L. Hallux osteoid osteoma: a case report and literature review. Open Orthop J. 2017;11:1066-72.

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(16.) Rolvien T, Zustin J, Mussawy H, Schmidt T, Pogoda P, Ueblacker P. Intra-articular osteoid osteoma as a differential diagnosis of diffuse mono-articular joint pain. BMC Musculoskelet Disord. 2016;17(1):455.

(17.) Starcevic S, Nikolic D, Tatic V, Popovic Z. [Osteoid osteoma-ten-years' experience in therapy]. Vojnosanit Pregl. 2004;61(1):9-14.

(18.) Yokouchi M, Nagano S, Shimada H, Nakamura S, Setoguchi T, Kawamura I, et al. Early complete remission of osteoid osteoma with conservative medical management. Pediatr Rep. 2014;6(1):5311.

(19.) Roqueplan F, Porcher R, Hamze B, Bousson V, Zouari L, Younan T, et al. Long-term results of percutaneous resection and interstitial laser ablation of osteoid osteomas. Eur Radiol. 2010;20(1):209-17.

(20.) Burger IM, McCarthy EF. Phalangeal osteoid osteomas in the hand: a diagnostic problem. Clin Orthop Relat Res. 2004;(427):198-203.

(21.) Maric MD, Djan I, Maric LD, Madic D, Petkovic D, Velickovic S, Gajdobranski Dj. Metatarsalgia caused with osteoid osteoma in active gymnast: case report. HealthMED. 2011:5(6 Suppl 1):2183-5.

(22.) Maric D, Djan I, Petkovic L, Vidosavljevic M, Sopta J, Maric D, et al. Osteoid osteoma: fluoroscopic guided percutaneous excision technique--our experience. J Pediatr Orthop B. 2011;20(1):46-9.

(23.) Jang WY, Lee SH, Cho IY. Progressive femoroacetabular impingement after complete excision of osteoid osteoma in adolescents: a report of two cases. Skeletal Radiol. 2017; 46(4):553-7.

(24.) Assenmacher S, Voggenreiter G, Klaes W, Nast-Kolb D. [Osteoid osteoma--a diagnostic and therapeutic problem?]. Chirurg. 2000;71(3):319-25.

Rad je primljen 25. II 2018.

Recenziran 25. VI 2018.

Prihvacen za stampu 3. X 2018.

BIBLID.0025-8105:(2018):LXXI:9-10:319-322.

Health Center Indija, Indija

Vesna PETROVIC, Tanja ROZEK MITROVIC and Danilo VISNJEVAC

Corresponding Author: Dr Vesna Petrovic, Dom zdravlja Indija, 23320 Indija, Srpskocrkvena 3, E-mail: dr.vesna.petrovic@gmail.com

https://doi.org/10.2298/MPNS1810321P
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Title Annotation:CASE REPORTS/PRIKAZI SLUCAJEVA
Author:Petrovic, Vesna; Mitrovic, Tanja Rozek; Visnjevac, Danilo
Publication:Medicinski Pregled
Article Type:Report
Date:Sep 1, 2018
Words:1988
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