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BAOJ Orthopedics
Repeated Knee Arthroscopic Surgeries and Manipulations for Knee Pain and Joint Stiffness Secondary to Melorheostosis: A Case Report
Chien-An Shih1, Chih-Kai Hong1, Chih-Wei Chang1, Wei-Ren Su1,2 and Chyun-Yu Yang1*
1Department of Orthopaedic Surgery, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan, Taiwan2Medical Device Innovation Center, National Cheng Kung University, Tainan, Taiwan
Chien-An Shih, et al, BAOJ Ortho 2016 1: 21: 006
*Corresponding author: Chyun-Yu Yang, Department of Orthopaedic Surgery, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University,138 Sheng Li Road, Tainan 704, Taiwan, Fax: +886-6-2766189, Tel: +886-6-2353535; E-mail: [email protected]
Sub Date: November 26, 2016, Acc Date: December 6, 2016, Pub Date: December 7, 2016.
Citation: Chien-An Shih, Chih-Kai Hong, Chih-Wei Chang, Wei-Ren Su and Chyun-Yu Yang (2016) Repeated Knee Arthroscopic Surgeries and Manipulations for Knee Pain and Joint Stiffness Secondary to Melorhe-ostosis: A Case Report. BAOJ Ortho 1: 006.
Copyright: © 2016 Chien-An Shih, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution Li-cense, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
BAOJ Ortho, an open access journal Volume 1; Issue 2; 006
Case Report
AbstractIntroduction
Melorheostosis is a rare osteosclerotic bone disease. It affects skeleton and adjacent tissues, causing pain and contracture. Typical radiographic findings are cortical hyperostotic changes, resembling melting wax dripping from a candle. Surgeries are usually conducted to manage the resultant contracture and pain. However, no previous studies mentioned the long-term surgical outcomes. We therefore would like to present the long-term outcome of a female patient with recurrent knee pain and joint stiffness secondary to melorhestosis after repeated knee arthroscopic surgeries and manipulations.
Case Report
Our patient is a 33-year-old female with recurrent right knee pain with mild to moderate ROM limitations due to intra-articular loose bodies secondary to melorheostosis. We performed a second arthroscopic surgery with manipulation, and noted that her knee joint space is still preserved 8 years after the first one. Her right knee flexion and extension was restored and pain was tolerable after repeated arthroscopic surgeries. At the last follow-up when she was 44, her knee ROM was 0 to 110 degrees and her pain score was 2 (VAS: 0-10).
Conclusion
Our case suggested that knee arthroscopic surgeries, manipulations, and intense rehabilitation program may be beneficial to relieve pain, restore range of motion, arrest osteoarthritis progression and improve long-term outcomes for those with melortheostosis and mild to moderate knee osteoarthritis or joint stiffness.
Key Words: Knee Pain; Contracture; Joint Stiffness; Melorheostosis; Arthroscopic Release; Recurrence
IntroductionFirst described in 1922 by Leri and Joanny [1], melorheostosis is a rare, benign, sclerosing, disease that affects the major axis of bones, and the surrounding tissues. The disease may cause progressive joint stiffness [2]. The incidence of the disease is 0.9 cases per million. The disease affects both sex and all ages equally, and is associated with functional morbidity [2,3]. Dysplasia mostly starts since childhood and typically involved unilateral limbs with
higher prevalence in lower extremities than upper extremities [2,4]. The genetic involvement is still unknown. The loss of LEMD3 gene (also known as MAN1) has been reported in patients with osteopoikilosis, an autosomal dominant skeletal dysplasia characterized by multiple hyperostotic areas in different parts of the skeleton, which often co-exist with melorheostosis [5,6].
Diagnosis is made based on radiographic appearance rather than specific biomarkers or histologic findings. The radiologic findings include hyperostotic endosteal involvement in children or the extra-osseous sclerosis feature in adults, characterized by the typical “molten wax” appearance [7]. Heterotropic bone formation as well as soft tissue calcification may lead to limb shortening in children, range of motion restriction, contracture, chronic pain and disability. Conservative treatment is often ineffective for this condition [8]. Surgeries, such as tendon lengthening, contracture release, excision of hyperostosis and even resurfacing procedure, are often used for managing intractable pain and deformity, in particular, in severe cases [9-12].
Knee contracture is not uncommon in patients with melorheostosis involving their knee joints [12-16]. Conservative treatments, including local analgesia, bracing, serial casting and physical therapy, often fail to manage the contracture long-term. Previously, a wide variety of open surgical procedures have been used, including fasiotomies, capsulotomies and soft tissue release with limited success [17,18]. With the advancement in medical technologies
BAOJ Ortho, an open access journal Volume 1; Issue 2; 006
Page 2 of 5Citation: Chien-An Shih, Chih-Kai Hong, Chih-Wei Chang, Wei-Ren Su and Chyun-Yu Yang (2016) Repeated Knee Arthroscopic Surgeries and Manipulations for Knee Pain and Joint Stiffness Secondary to Melorheostosis: A Case Report. BAOJ Ortho 1: 006.
and surgical techniques, arthroscopically assisted procedures are becoming more common. To the best of our knowledge, there were only three case reports on the arthroscopic treatment for knee pain and joint stiffness due to melorheostosis, and none of them documented long-term outcomes [12,15,16]. Hence, we would like to present a female patient with recurrent knee joint stiffness and pain secondary to melorheostosis who underwent arthroscopic arthrolysis, synovectomry and manipulations. A specific focus was placed on the long-term effects of the surgery.
Case ReportA 33-year-old woman with progressive knee pain attended our clinic. Her pain score was 8 using visual analogue scale (none: 0, mild: 1-3, moderate: 4-7, severe: 8-10). There was a reduction in her knee range from 0-130 degrees to 20-90 degrees in the last 2 years despite conservative treatment, including local analgesia, drugs, physical therapy and occupational therapy. Plain X-ray of the affected knee showed cortical hyperostosis that resemble the dripping of candle wax, particularly in proximal tibia and patella around knee anterior compartment (Figures 1A-D). It also showed low grade (Kellgren & Lawrence, K & L, grade 0-1) osteoarthritis. Hyperostosis were noted in right ankle joint involving right anterior distal tibia and dorsal talar neck (Figures 2A-B). Patient reported that the ankle pain was tolerable (VAS score: 2). A MRI study (Figure 3A-C) confirmed the diagnosis of melorheostosis. Due to the ineffectiveness of conservative treatments, we therefore performed arthroscopic arthrolysis, synovectomy and manipulation.
Under spinal anesthesia in supine position, four portals (anteromedial, anterolateral, superomedial and superolateral) were created to gain access to the anterior knee compartment. Hyperplastic synovial tissues and acute synovitis (Figure 4A) along with numerous osseous lesions were found on the right patella surface (Figure 4B), in the infra-patella space (Figure 4C) and the joint capsule (Figure 4D). Arthroscopic arthrolysis, synovectomy and debridement were performed to remove all fibrous and osseous tissue over both medial and lateral gutters, supra- and infra-patellar
space, intercondylar notch and lateral meniscus (Figure 4E). The outerbridge chondral lesion (grade III-IV) on both patellar medial and lateral facets were debrided (Figure 4F). Histologic findings were in line with the MRI findings, that is, in support of the diagnosis of melorheostosis. Manipulation was performed during the surgery along with the arthroscopic release. Her knee ROM achieved 0-150 degrees intra-operatively. She was able to walk fully-weight-bearing after the surgery. Intense continuous passive range of motion was performed following the surgery. Two weeks after the surgery, her knee range was 0-130 degrees, pain score was 2 (mild) and satisfactory score was 4 (1: bad, 2: fair, 3: good, 4: excellent) [19].
Nine years after the first surgery when she was 42, she revisited our clinic as her pre-tibial pain worsened and range of motion was reduced to 0-90 degrees in the last 6 months. Plain radiography showed recurrence of infra-patella ossifications (Figure 5A). The same procedure was performed again, and the osseous lesions were removed (Figure 5B). Compared with last surgery, there were less scar tissues this time. Two weeks after surgery,
Fig. 1 Pre-operative images show cortical hyperostosis in proximal tibia (A-B) and patella (C-D) around anterior knee compartment.
Fig. 2 Hyperostosis in right ankle joint: (A) right anterior distal tibia and (B) dorsal talar neck
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her knee ROM increased to 0-130 degrees. Like last surgery, the pain score was 2 (mild) and satisfaction score was 4 (excellent). Post-operative rehabilitation included passive and active range of motion exercises. In the last follow-up (2 years after the second surgery), her knee ROM was 0-110 degrees. Her pain score was 2 (mild) and satisfaction score was 3 (fair). Plain radiography of right knee showed a slightly enlarged infra-patellar osteophyte (Figure
Fig. 3 The MRI (A) axial, (B) sagittal, and (C) coronal section of right knee joint shows hyperostosis involving patellofemoral joint and infra-patella space.
Fig. 4 Arthroscopic findings of right knee shows (A) hyperplastic synovial tissue and acute synovitis and numerous osseous lesion were identified in the right patella facets (B), infra-patella space (C), joint capsule (D), and lateral meniscus(E). An outerbridge grade III-IV chondral lesion on both patellar medial and lateral facets was debrided with a shaver (F).
Fig. 5 (A) Arthroscopic findings showing recurrence of recurrence at infra-petallar region. (B) Excise osseous fragment.
6A-B). However, pain was still tolerable for her. The severity of knee osteoarthritis (as rated by the K & L grades) had remained the same in the last 12 years (Figure 6C).
DiscussionWe presented a female patient with recurrent right knee pain and joint stiffness due to melorheostosis. She received two arthroscopies and manipulations over 12 years. This patient reported tolerable knee pain after the first operation until 8 years later. In the second surgery, there were less scar tissues around the knee joint. The radiography 12 years later showed no changes in the severity of right knee osteoarthritis despite an increase ossifications on tibia plateau eminence.
Arthroscopic surgery has gained popularity and has been widely used in surgical procedures for knee problems. The applications of knee arthroscopy are wide ranging including debridement, intra-articular tumor excision, reconstructive surgery, distal femur or proximal tibia fracture reduction surgery and extra-articular procedures such as cystic debridement, abscess drainage. The successful rates of these procedures are high [20-23]. In people with melorheostosis and severe contracture, traditional methods such as manipulations, casts, open extensive soft tissue release, open capsulotomies, osteotomies, arthrodesis and correction with Ilizarov frame, often have uncertain results, low successful rates or high recurrence rates [17, 24-26].
Athroscopicarthrolysis have been successfully used in treating patients with knee contracture after knee trauma, knee osteoarthritis, movement disorders and total knee arthroplasties (TKA) [27-31]. Good pain relief was generally reported after arthroscopic synovectomy and arthroscopic removal of the loose bodies. However, the outcomes after arthroscopic surgeries were less predictable in knee joint stiffness secondary to melorheostosis. Recurrence may occur due to its continuous soft tissue contracture characteristic [12,15,16]. There are limited reports on using arthroscopic surgeries for knee melorheostosis. Minarro et al
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Page 4 of 5Citation: Chien-An Shih, Chih-Kai Hong, Chih-Wei Chang, Wei-Ren Su and Chyun-Yu Yang (2016) Repeated Knee Arthroscopic Surgeries and Manipulations for Knee Pain and Joint Stiffness Secondary to Melorheostosis: A Case Report. BAOJ Ortho 1: 006.
Fig. 6 Plain radiography of right knee lateral view: (A) 2 years after 2nd debridement, (B) after 2nd debridement, and (C) before 1st debridement.
reported findings on a 23-year-old female who presented with anterior knee pain, very mild flexion contracture (5-7 degrees) and low grade osteoarthritis. During arthroscopy, a small 2 x 2 cm calcifications was removed and hyperplastic synovial membrane was debrided [15]. Six months later, this patient was asymptomatic with no recurrent flexion deformity. Similarly, Claramunt et al [16] described a 59-year-female with knee pain and severe right knee joint stiffness (ROM: 10-60 degrees) secondary to melorheostosis which affected both the anterior and posterior knee compartments. Immediately after the arthroscopic arthrolysis, her post-operative knee ROM improved to 0-130 degrees. Three weeks after the surgery, she achieved ROM of 0-110 degrees with intensive rehabilitation. However, her ROM diminished to 5-90 degrees over the following six months. In the last follow-up 15 month after the first surgery, the ROM was maintained, and her medial knee pain remained tolerable. Moulder et al [12] also reported a 40-year-old woman with advanced knee osteoarthritis and severe knee joint stiffness (30-40 degrees). Over 9 years, this patient received manipulation under anesthesia and two arthroscopic surgeries to remove loose bodies. However, she lost further range in her knee joint. Consequently, she received total knee arthroplasty and then intensive rehabilitation. Six weeks after TKA, her right knee ROM was still limited (0-65 degrees). She continued to receive further rehabilitation. In the last follow-up at 18 months, her ROM increased to 0-80 degrees. Post-operative pain status was not reported in this study.
Melorheostosis is often accompanied by periods of remission and exacerbation. There is no effective conservative treatment [4,8]. The effect of oral medications, such as NSAIDS, nifedipine, bisphosphonates and pamidronate, was unclear [8,32]. Significant contracture and bone deformity are more likely to require surgeries [4]. Successful rates and long term outcomes of fasiotomies, capsulotomies and open soft tissue release are variable between studies [17,18]. Our case study and two previous studies [15,16] suggested that arthroscopic arthrolysis and manipulation could be an effective procedure in the treatment of knee contracture. Yet, in patients with more severe joint stiffness, recurrence can occur for which further surgeries would be necessary. In some cases,
especially those with advance knee osteoarthritis, the benefits of knee arthroscopic surgeries and manipulations are be limited [12], and resurfacing surgery may be necessary [12,33] Aggressive intensive post-operative rehabilitation may help prevent recurrence of contracture [12,33]. Currently, it remains unclear which sub-group of patients with melorheostosis and knee contracture would benefit from arthroscopic surgeries. Further studies are needed to evaluate the relationship between the outcomes after knee arthroscopies or resurfacing surgeries and disease severity.
Our case suggested that knee arthroscopic surgeries, manipulations, and intense rehabilitation program may be beneficial to relieve pain, restore range of motion, arrest osteoarthritis progression and improve long-term outcomes for those with melortheostosis and mild to moderate knee osteoarthritis or joint stiffness.
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