Intra‐articular Osteoid Osteoma of the Intermediate Cuneiform Bone Causing Articular Degeneration: A Rare Case and Literature Review

Abstract

Osteoid osteoma of the cuneiform bone is an exceedingly rare and easily missed cause of foot pain. The uncharacteristic and nonspecific radiographs of such intra‐articular osteoid osteoma further increase difficulty in making the diagnosis. To date, there has been no description of intra‐articular osteoid osteoma of the intermediate cuneiform bone causing articular degeneration in any published literatures. We present a case of intra‐articular osteoid osteoma of the intermediate cuneiform bone causing articular degeneration, who underwent curettage, allograft bone graft, and navicular‐cuneiform arthrodesis. The patient presented with radiographic bone union, full motor function recovery and pain‐free at the 22‐month follow‐up. This report adds to the existing literature. Intra‐articular osteoid osteoma of the intermediate cuneiform bone causing articular degeneration is an exceedingly rare and easily missed cause of foot pain. It proves a complicated and challenging task to identify intra‐articular osteoid osteoma. Clinicians should be particularly careful not to exclude the possibility of arthritis and, thus, vigilant when choosing the surgical option.

In this work, we report a patient with an intra‐articular osteoid osteoma of the intermediate cuneiform bone causing articular degeneration, who underwent curettage, allograft bone graft, and navicular‐cuneiform arthrodesis. The patient presented with radiographic bone union, full motor function recovery and pain‐free at the 22‐month follow‐up. Intra‐articular osteoid osteoma of the intermediate cuneiform bone causing articular degeneration is an exceedingly rare and easily missed cause of foot pain. It proves a complicated and challenging task to identify intra‐articular osteoid osteoma. Our work cautions clinicians against overlooking the possibility of arthritis caused by intra‐articular osteoid osteoma.

Introduction

Among benign bone tumors, osteoid osteoma is the third most common, comprising 2%–3% of all bone tumors and 10%–14% of benign bone tumors. ¹ , ² The most common cases of osteoid osteoma are in children and young adults, usually between 5‐ and 30‐years‐old. ³ Osteoid osteoma occurs mostly in the extremities and spine, in particular, the femur and tibia. The first reported case of osteoid osteoma was in 1935 ⁴ and the first reported osteoid osteoma of the cuneiform bone was in 1983. ⁵ Different forms of pain are its typical clinical manifestation, the most striking feature of which is intensified at night and relieved by salicylates or NSAIDs. Conservative treatment is a preferred option, for the chance that osteoid osteoma heals spontaneously. ⁶ However, a failed trial of non‐operative therapy leads to the need for surgical management. Minimally invasive procedures (for example, radiofrequency ablation [RFA], cryoablation, and microwave ablation), have been gradually favored compared with traditional surgery. ⁷ , ⁸ , ⁹ To the best of our knowledge, no osteoid osteoma of the cuneiform bone with articular cartilage injury has been reported. We report a patient with an intra‐articular osteoid osteoma of the intermediate cuneiform bone causing articular degeneration.

Case Report

A 19‐year‐old man complained of severe pain and swelling in the right foot for 9 months. Tenderness was observed at the site of the midfoot. No obvious abnormality was found in foot alignment. The remarkable clinical finding was nocturnal pain and pain relief after oral NSAIDs. However, a reduction in the drug's efficacy and increased pain drove him to seek help for further intervention. The VAS score was 7 points, and the AOFAS score was 41 points.

Blood and biochemical tests did not show any abnormality. Preoperative CT images showed a tiny soft‐tissue density‐like lesion surrounded by reactive sclerosis, which was located at the dorsal proximal intermediate cuneiform bone. Also, joint space narrowing was found in medial‐intermediate, intermediate‐lateral cuneiform, and navicular‐cuneiform joints. Non‐articular cortical bone thickened at the dorsal intermediate and lateral cuneiform bone, resulting from the periosteal reaction. Preoperative MRI images showed the nidus displays low‐to‐intermediate signal intensity on T2‐weighted images and low signal intensity on T1‐weighted images. To note, bone marrow edema was found in intermediate and lateral cuneiforms and extensive synovitis was found in medial‐intermediate, intermediate‐lateral cuneiform, and navicular‐cuneiform joints. After intravenous gadolinium, the nidus enhances on T1‐weighted images (TR 650/TE 8.93 msec) (Fig. 1).

Fig. 1.

Preoperative imagological data. (A–C, E–I) The location of osteoid osteoma was indicated by red arrowheads. (C) The tiny lesion in soft‐tissue density was surrounded by reactive sclerosis. (D) joint space narrowing was found in medial‐intermediate and intermediate‐lateral cuneiform joints. Periosteal reaction occurred at the dorsal intermediate and lateral cuneiform bone. (E, F, G) The nidus displays low‐to‐intermediate signal intensity on T2‐weighted images. Bone marrow edema was found in intermediate and lateral cuneiforms. (H) The nidus displays low signal intensity on T1‐weighted images. (I) After intravenous gadolinium, the nidus enhances on T1‐weighted images (TR 650/TE 8.93 msec). (J) Extensive synovitis was found in medial‐intermediate, intermediate‐lateral cuneiform, and navicular‐cuneiform joints.

Preoperative differential diagnosis was conducted. Preoperative CT and MRI excluded fracture, malignant tumor, tarsal coalition, tendonitis, tendon or ligament injuries. On the other hand, considering no definite injury history nor patient had not been in any vigorous sport activities, fracture or stress fracture was ruled out. In addition, chance of infection was unlikely in CT and MRI image result together with normal inflammatory biomarkers. Occasionally transient bone marrow edema may affect a large joint among the middle‐aged, might accompanied by regional migratory osteoporosis which was not consistent with our patient's characteristic. Besides, primary osteoarthritis is very unlikely in young patients. Secondary osteoarthritis can be a possible diagnosis. However, midfoot was an uncommon site for metabolic and inflammatory joint disease. Instead, tumor induced synovitis or osteoarthritis was probable. A major diagnostic point was nocturnal pain which can be relieved by NSAID. Therefore, the primary diagnosis of osteoid osteoma was arrived at, with both CT and MRI images support.

After careful evaluation and discussion with the patient, we chose curettage, allograft bone graft, and navicular‐cuneiform arthrodesis because of the patient's worry about donor site morbidity.

The procedure was performed under general anesthesia, patient was lying supine with thigh tourniquet applied. A 7‐cm longitudinal dorsal incision was made lateral to the extensor hallucis longus tendon. The dorsalis pedis artery and the deep peroneal nerve were protected and retracted laterally while extensor hallucis longus tendon retracted medially. The navicular and three cuneiform bones were disposed. A distractor was placed across the navicular‐cuneiform joint. A red lesion with soft and friable composition and a diameter of 0.5 × 0.5 cm was observed located at the dorsal proximal intermediate cuneiform bone. Additionally, extensive synovial hyperplasia and cartilage destruction were found during articular explorations. After adequate curettage and debridement of the lesion, corresponding cartilage was removed adequately. Subchondral bone was fenestrated using a small drill to facilitate the fusion. Allograft bone graft was packed to fill in the defect space. The joints were initially fixed by K‐wire when the alignment was checked with intraoperative fluoroscopy. A locking plate and screws were used as the final fixation. The incision was closed with layers of sutures. Sterile compression dressing was applied and the foot and ankle were immobilized with a splint.

The histology of the specimen confirmed the diagnosis of osteoid osteoma (Fig. 2). In first 6 weeks after surgery, the patient was on non‐weight‐bearing walking with walking boot and crutches. The ankle was placed in 90° dorsiflexion for the duration of non‐weight‐bearing. Suture was removed at 2 weeks after the surgery. Active muscle contraction and ankle flexion/extension exercises were performed shortly after the surgery with the numbers of exercise increasing to 150 times per day. At 6 weeks postoperatively (the first follow‐up visit), an X‐ray revealed an initial callus formation. We encouraged the patient to partial weight‐bearing walk and increase activity as tolerated. Gradually, the patient was transferred to full weight‐bearing and activities of daily living. The patient's rehabilitation exercises were instructed during follow‐up visits.

Fig. 2.

Histological examination showed variable mineralization and of a richly fibroblastic stroma. The fibrous stroma contains dilated blood vessels. No evidence of tumor necrosis or cellular atypia was found. The findings consistent with osteoid osteoma. (A) Low power view (hematoxylin and eosin stain, origin magnification ×40). (B) High power view (hematoxylin and eosin stain, origin magnification ×200).

There was no postoperative complication. The first follow‐up was conducted at 6 weeks postoperatively. Since then, follow‐ups have been conducted every 3 months. The latest follow‐up was performed 22 months after surgery. The patient was satisfied with full motor function recovery and pain‐free. 22‐month postoperative X‐ray showed good bony fusion with no tumor recurrence (Fig. 3).

Fig. 3.

22‐month postoperative X‐ray showed good bony fusion with no tumor recurrence.

Discussion

As far as we know, only 20 cases of osteoid osteoma of the cuneiform bone have been reported in the literature, among which only three cases were located in the intermediate cuneiform bone (Table 1). However, no osteoid osteoma of the cuneiform bone causing articular cartilage destruction has been reported. Our case presented an intra‐articular osteoid osteoma of the intermediate cuneiform bone causing articular degeneration. After treatment with curettage, allograft bone graft, and navicular‐cuneiform arthrodesis, the patient showed satisfactory results without recurrence.

TABLE 1.

Osteoid osteoma of the cuneiform bone previously reported in the literature

Study lead author	Patient	Location	Treatment
Shereff, 5 1983	3	One each in medial, intermediate, and lateral cuneiform bone	Excision
Ambrosia, 36 1985	1	Lateral cuneiform bone	Excision and autologus bone from the iliac crest
Catani, 37 1994	1	Lateral cuneiform bone	En bloc excision and autologus bone from the iliac crest
Zouari, 38 2008	1	Medial cuneiform bone	CT‐guided percutaneous laser photocoagulation
Le corroller, 39 2011	1	Lateral cuneiform bone	Percutaneous radiofrequency ablation
Neumann, 40 2012	1	Medial cuneiform bone	CT‐guided percutaneous radiofrequency thermoablations
De Palma, 41 2013	1	Cuneiform bone (unspecified)	CT‐guided percutaneous radiofrequency thermoablation
Bourgault, 42 2014	1	Lateral cuneiform bone	CT‐guided radiofrequency thermocoagulation
Houdek, 43 2014	1	Lateral cuneiform bone	Curettage or radiofrequency ablation
Angelini, 44 2014	5	Cuneiform bone (unspecified)	Three percutaneous drill resection and Two radiofrequency
Ayas, 45 2020	1	Lateral cuneiform bone	Excision
Smolle, 3 2021	2	Medial cuneiform bone	Curettage or radiofrequency ablation
Payo‐ollero, 11 2021	1	Intermediate cuneiform bone	Curettage and bone grafting

Intra‐articular osteoid osteoma is of rare occurrence, which accounts for 5.2% of all locations. ¹⁰ Radiographs of intra‐articular osteoid osteoma are nonspecific, for example, joint effusion and bone marrow edema. Particularly, lack of the intense perifocal sclerotic margin tends to cause diagnostic difficulties. Sometimes, classical symptoms may not present, such as nocturnal pain or a positive salicylate test. ¹¹ , ¹² Therefore, intra‐articular osteoid osteoma with misleading clinical and radiological findings is likely to contribute to a significant delay in diagnosis (on average 26.6, 14.1 months later than in non‐articular locations). ¹³

Several differential diagnoses should be noticed. First, trauma should be highly suspicious which injury history and imaging examinations will help to identify. Second, stress fracture, especially in athletes, is another differential diagnosis. Which is unlikely in our patient as he was not active in sports activities. Third, congenital malformation, such as tarsal coalition, may be a cause, clinical foot mal‐alignment, and further imaging examinations will help to confirm the diagnosis. Fourth, infection such as skin rupture, purulent secretion, sinus tract, elevation of inflammatory biomarkers (C‐reactive protein, erythrocyte sedimentation rate, for instance), and imaging examinations (abscess, bone destruction, sequestrum, for instance) will confirm the diagnosis. Fifth, metabolic and inflammatory joint diseases with their own predilection sites, respective, rarely occurs in midfoot. Blood and biochemical investigations are helpful. Finally, in view of the young age, malignant tumor should be watched for too. The mass rapid growing in a short time and imaging findings (extensive cortical erosion, the mass invading the surrounding structure, rich blood flow, for instance) may indicate malignancy.

Our case presented as a tiny lesion in soft‐tissue density surrounded by reactive sclerosis and other uncharacteristic imaging manifestations, which was easily misdiagnosed and overlooked. Luckily, we noted a characteristic history of nocturnal pain which was effectively relieved by NSAIDs. Again, careful scrutiny was given, leading to the correct diagnosis of osteoid osteoma. Interestingly, we noted periosteal reaction occurred at the dorsal intermediate and lateral cuneiform bone. Rarely has literature reported periosteal reaction taking place in extraarticular cortical bone, even adjacent bone. ¹⁴ Finally, the consequent histopathology confirmed our diagnosis.

Percutaneous ablative therapies, with RFA most represented, are commonly used due to their cost‐effectiveness, low invasiveness, safe, convenience, and low recurrence rate. ¹⁵ Nevertheless, the most concerning complications are neurovascular injury, cartilage injury, and skin burns. Although ablation of osteoid osteoma close to the articular surface or nerve seems generally effective and safe in the short term, ¹⁶ , ¹⁷ , ¹⁸ , ¹⁹ it can be quite challenging with certain risks. ²⁰ Thus, a margin of safety is at least >1 cm from the lesion to the neurovascular bundle or skin surface. ²¹ The installation of 10% dextrose solution in the subcutaneous plane or the joint cavity may be beneficial. ²² Furthermore, MR‐guided focused ultrasound ablation therapy (MRgFUS) emerges as a new non‐invasive radiology revolution with little damage to the neurovascular bundle or articular cartilage. ²³ , ²⁴ , ²⁵ In addition, relatively large lesions measuring >1 cm in size might have higher odds of recurrence. ²⁶ , ²⁷ The use of a multi‐pronged probe or multiple probe placements is recommended. ²²

Arthroscopy is another popular procedure with specific advantages of clear tumor visualization, minimal invasion, and sample gathering. What is more, arthroscopy allows the handling of many other conditions simultaneously. Studies have demonstrated successful applications of arthroscopy in osteoid osteoma. ²⁸ , ²⁹ , ³⁰ , ³¹ Interestingly, for intra‐ and juxta‐articular localization of osteoid osteoma, arthroscopy‐assisted radiofrequency ablation was proposed. ³¹ , ³² , ³³ This technique synthesizes both of the strengths. However, further studies with higher evidence quality are warranted to validate the effectiveness and efficiency of this method.

In our case, the intra‐articular osteoid osteoma was located at the dorsal proximal intermediate cuneiform bone with extensive cartilage destruction. It was anticipated that simply dealing with osteoid osteoma and neglecting arthritis would result in treatment failure. Also, the proximity between the dorsalis pedis artery, skin surface, and the lesion restricted the percutaneous ablative techniques. Moreover, the narrow operating space made it practically impossible for arthroscopic management. Hence, we chose traditional open surgery. Taking into account that overly cautious handling of the nidus might increase the hazards of recurrence or residual pain, ³⁴ , ³⁵ we performed adequate curettage and debridement, followed by allograft bone graft and navicular‐cuneiform arthrodesis. the patient showed satisfactory results without recurrence. At the final follow‐up, the pain was abolished and a good functional result was achieved.

Conclusion

Intra‐articular osteoid osteoma of the intermediate cuneiform bone causing articular degeneration was an exceedingly rare and easily missed cause of foot pain. It proved a complicated and challenging task to identify intra‐articular osteoid osteoma. Clinicians should be particularly careful not to exclude the possibility of arthritis and, thus, vigilant when choosing the surgical option.

Author Contributions

ZhongMin Shi and Cheng Chen conceptualized the study. Yan Su and ShaoLing Fu collected patients' information and reviewed the literature. Cheng Chen and ShaoLing Fu drafted the manuscript. ZhongMin Shi and Yan Su revised the manuscript. All authors read and approved the final version.

Conflicts of Interest Statement

The authors declare no conflicts of interest. The authors report no conflicts of interest concerning the materials or methods used in this study or the findings specified herein.

Ethics Statement

The study was approved by the Institutional Ethics Committee of Shanghai Jiao Tong University Affiliated Sixth Peoples Hospital. Written informed consent was obtained from the patient for the publication of this manuscript and any accompanying images. The study was conducted in compliance with the Helsinki Declaration.

Data Availability Statement

The original contributions presented in the study are included in the article, further inquiries can be directed to the corresponding author/s.