The Open Orthopaedics Journal




ISSN: 1874-3250 ― Volume 13, 2019
CASE REPORT

Fabella Syndrome Following De-Rotation Surgery to Correct a Femoral Malunion



Iain Rankin*, Haroon Rehman, George Patrick Ashcroft
NHS Grampian, Aberdeen Royal Infirmary, Aberdeen, UK

Abstract

Background:

The fabella is a sesamoid bone situated within the lateral head of the gastrocnemius tendon, close to the lateral femoral condyle, and adjoined to the fabellofibular ligament. It is a normal variant, found in up to 87% of patients. Fabella Syndrome describes traditionally posterolateral knee pain, occurring due to biomechanical pressure of the fabella against the lateral femoral condyle. Given its rarity, its diagnosis is often overlooked. We present a case of Fabella Syndrome with a modified surgical excision technique and review of the literature.

Methods and Results:

A thirty-four-year-old man presented with posterolateral knee pain following de-rotation surgery to correct a femoral malunion, from a previous femoral shaft fracture. Due to the patient’s complex orthopaedic history, Fabella Syndrome was not initially diagnosed. Fabellectomy eliminated all symptoms of knee pain, with no limitations in knee function.

Conclusion:

Review of the literature identified ten publications (evidence level IV) describing Fabella Syndrome. This is the first reported case of Fabella Syndrome secondary to femoral de-rotation surgery. The authors recommend fabellectomy as a definitive treatment for Fabella Syndrome, in keeping with published literature.

Keywords: Fabella, Fabella Syndrome, Modified Surgical Technique, Arthroscopy, Case report, Knee.


Article Information


Identifiers and Pagination:

Year: 2018
Volume: 12
First Page: 346
Last Page: 352
Publisher Id: TOORTHJ-12-346
DOI: 10.2174/1874325001812010346

Article History:

Received Date: 28/4/2018
Revision Received Date: 16/6/2018
Acceptance Date: 24/6/2018
Electronic publication date: 29/08/2018
Collection year: 2018

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© 2018 Rankin et al.

open-access license: This is an open access article distributed under the terms of the Creative Commons Attribution 4.0 International Public License (CC-BY 4.0), a copy of which is available at: (https://creativecommons.org/licenses/by/4.0/legalcode). This license permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.


* Address correspondence to this author at the NHS Grampian, Aberdeen Royal Infirmary, Aberdeen, UK; Tel: +441224 437848; E-mail: iain.rankin@doctors.org.uk





1. INTRODUCTION

The fabella is a sesamoid bone situated within the lateral head of the gastrocnemius tendon, close to the lateral femoral condyle, and adjoined to the fabellofibular ligament. Its presence is a normal variant, with a prevalence reported as ranging from 20 to 87% [1Tabira Y, Saga T, Takahashi N, Watanabe K, Nakamura M, Yamaki K. Influence of a fabella in the gastrocnemius muscle on the common fibular nerve in Japanese subjects. Clin Anat 2013; 26(7): 893-902.
[http://dx.doi.org/10.1002/ca.22153] [PMID: 22933414]
-7Ehara S. Potentially symptomatic fabella: MR imaging review. Jpn J Radiol 2014; 32(1): 1-5.
[http://dx.doi.org/10.1007/s11604-013-0253-1] [PMID: 24158650]
]. It is situated within the lateral head of gastrocnemius tendon, with the fabellofibular ligament (or ligament of Vallois) connecting it to its distal insertion at the fibular head [8Driessen A, Balke M, Offerhaus C, et al. The fabella syndrome - a rare cause of posterolateral knee pain: A review of the literature and two case reports. BMC Musculoskelet Disord 2014; 15: 100.
[http://dx.doi.org/10.1186/1471-2474-15-100] [PMID: 24666711]
]. It is bordered posteriorly by the oblique popliteal ligament [2Pritchett JW. The incidence of fabellae in osteoarthrosis of the knee. J Bone Joint Surg Am 1984; 66(9): 1379-80.
[http://dx.doi.org/10.2106/00004623-198466090-00009] [PMID: 6501334]
]. Anteriorly, it is bordered by the posterior capsule of the knee. Its location within the posterolateral aspect of the knee places it within intersecting lines of tensile stress and articulating with the lateral femoral condyle [5Müller W. The knee - form, function and ligament reconstruction. Knee - Form, Funct Ligament Reconstr 1982; 40, 96, 98, 192, 249, 252]

The presence of a fabella is usually asymptomatic. Fabella Syndrome refers to posterolateral knee pain associated with the presence of a fabella. The presence of pain occurs due to biomechanical pressure of the fabella against the lateral femoral condyle. It is characterized by periodic posterolateral pain which is exacerbated during extension, where tension causes the fabella to compress against the lateral femoral condyle [9Weiner DS, Macnab I. The “fabella syndrome”: An update. J Pediatr Orthop 1982; 2(4): 405-8.
[http://dx.doi.org/10.1097/01241398-198210000-00010] [PMID: 6815224]
]. On occasion, its presence can result in irritation of the common peroneal nerve, causing neurologic symptoms of numbness or dysesthesia [10Mangieri JV. Peroneal-nerve injury from an enlarged fabella. A case report. J Bone Joint Surg Am 1973; 55(2): 395-7.
[http://dx.doi.org/10.2106/00004623-197355020-00019] [PMID: 4696171]
].

The presence of a fabella may be palpated during clinical examination. Lateral radiographs of the knee can confirm the presence of a fabella. CT and MRI imaging can delineate the position of a fabella in relation to the posterolateral femoral condyle. Ultrasound has been described as a useful adjunct to provide information regarding the posterolateral structures of the knee and their relation to a fabella [11Sekiya JK, Jacobson JA, Wojtys EM. Sonographic imaging of the posterolateral structures of the knee: Findings in human cadavers. Arthroscopy 2002; 18(8): 872-81.
[http://dx.doi.org/10.1053/jars.2002.32845] [PMID: 12368785]
, 12Draghi F, Danesino GM, Coscia D, Precerutti M, Pagani C. Overload syndromes of the knee in adolescents: Sonographic findings. J Ultrasound 2008; 11(4): 151-7.
[http://dx.doi.org/10.1016/j.jus.2008.09.001] [PMID: 23396316]
].

Fabella syndrome has been treated conservatively with manual therapy involving extension, flexion and rotation of the knee [13Zipple JT, Hammer RL, Loubert PV. Treatment of fabella syndrome with manual therapy: A case report. J Orthop Sports Phys Ther 2003; 33(1): 33-9.
[http://dx.doi.org/10.2519/jospt.2003.33.1.33] [PMID: 12570284]
]. Where conservative management has failed, operative treatment may be performed. Surgical techniques described include open, arthroscopic, and arthroscopic assisted open excision [8Driessen A, Balke M, Offerhaus C, et al. The fabella syndrome - a rare cause of posterolateral knee pain: A review of the literature and two case reports. BMC Musculoskelet Disord 2014; 15: 100.
[http://dx.doi.org/10.1186/1471-2474-15-100] [PMID: 24666711]
, 14Dannawi Z, Khanduja V, Vemulapalli KK, Zammit J, El-Zebdeh M. Arthroscopic excision of the fabella. J Knee Surg 2007; 20(4): 299-301.
[http://dx.doi.org/10.1055/s-0030-1248063] [PMID: 17993073]
, 15Provencher MT, Sanchez G, Ferrari MB, et al. Arthroscopy-assisted fabella excision: Surgical technique. Arthrosc Tech 2017; 6(2): e369-74.
[http://dx.doi.org/10.1016/j.eats.2016.10.011] [PMID: 28580255]
].

2. CASE REPORT

A 34-year-old gentleman was first referred to orthopaedic services with a history of persistent knee pain, located over the right femoral condyle near the origin of the lateral collateral ligament. Eleven years prior to presentation, the patient was involved in a road traffic accident where he sustained a fracture of the right femoral neck and ipsilateral shaft. Initial management consisted of open reduction, fixation of the femoral neck fracture with cannulated screws and the ipsilateral shaft fracture with plating. The patient later had a revision of the plate to a femoral nail. Union was subsequently achieved with the femoral shaft fracture; however, a significant external rotation deformity was noted, and discomfort to the knee.

The patient was referred on to our tertiary referral orthopaedic centre for femoral de-rotation surgery. An initial CT scan performed revealed an external rotation deformity of 45 degrees. The patient procedure included removal of femoral nail, osteotomy with de-rotation surgery, and subsequent exchange nail. The femoral nail was fixed proximally and locked into place distally, following the corrective 45 degrees of internal rotation achieved to the distal femur during osteotomy, performed under intraoperative radiographic guidance. The post-operative rehabilitation regime consisted of initial touch weight bearing only, with gradual increments in weight bearing status. At six months follow-up, the patient complained of distal lateral femur pain. A repeat CT scan was performed, revealing a delayed femoral union. This was initially thought to be the cause of the patient’s symptoms. The patient was admitted for dynamization of the femoral nail with an injection of bone graft substitute, and iliac crest graft. Despite eventual union, the patient continued to complain of pain at the distal lateral femur, with a cracking sensation on movement now noted.

Repeat clinical examination revealed a palpable crepitus over the distal iliotibial band with a snapping sensation, as it appeared to catch. A palpable small, solid swelling was noted at the posterolateral right knee. Ultrasound and CT revealed intra-articular loose bodies lying within the lateral para-patellar gutter. The patient subsequently underwent arthroscopy with removal of loose bodies. Despite this, at six-months follow-up to arthroscopy, the patient continued to complain of posterolateral knee pain. MRI showed no evidence of abnormality to the popliteus tendon or muscle, but highlighted the fabella embedded within the lateral head of gastrocnemius at the posterolateral corner of the knee. (Image 1). The fabella was also notable on radiographs (Image 2) and CT (Image 3). Ultrasound scan confirmed the fabella as mobile on movement and associated with tenderness. The diagnosis of Fabella Syndrome was made, and the patient planned for a Fabellectomy procedure.

An initial arthroscopy was performed to review the knee joint, and to potentially assist in surgical excision of the fabella. A complete diagnostic arthroscopy was performed, with the fabella unable to be identified through the posterior capsule. After arthroscopy, open fabellectomy was performed. In view of the patient’s extensive scarring from prior orthopaedic procedures, a short 2cm incision was made directly over the palpable fabella (Image 4). The common peroneal nerve was identified and spared. Subsequent incision left a cuff of biceps femoris to protect the nerve. Gastrocnemius was split and incised directly over the fabella, which was removed in entirety (Image 5). Histopathology confirmed the diagnosis.

The patient was followed-up at two and ten months postoperatively. At both follow-ups, he described the complete resolution of his posterolateral knee pain.

Image (1)
MRI revealing fabella embedded within the lateral head of gastrocnemius.


Image (2)
Review of sagittal radiographs revealing fabella.


Image (3)
Review of coronal CT revealing fabella.


Image (4)
Minimally invasive approach used directly over palpable fabella. Most posterior line utilised. Also marked are Gerdy’s tubercle and the head of the fibula.


Image (5)
The excised fabella.


3. DISCUSSION

Fabella syndrome is traditionally described as a cause of pain in the posterolateral aspect of the knee. A literature review revealed ten publications describing fabella syndrome [8Driessen A, Balke M, Offerhaus C, et al. The fabella syndrome - a rare cause of posterolateral knee pain: A review of the literature and two case reports. BMC Musculoskelet Disord 2014; 15: 100.
[http://dx.doi.org/10.1186/1471-2474-15-100] [PMID: 24666711]
, 9Weiner DS, Macnab I. The “fabella syndrome”: An update. J Pediatr Orthop 1982; 2(4): 405-8.
[http://dx.doi.org/10.1097/01241398-198210000-00010] [PMID: 6815224]
, 13Zipple JT, Hammer RL, Loubert PV. Treatment of fabella syndrome with manual therapy: A case report. J Orthop Sports Phys Ther 2003; 33(1): 33-9.
[http://dx.doi.org/10.2519/jospt.2003.33.1.33] [PMID: 12570284]
, 14Dannawi Z, Khanduja V, Vemulapalli KK, Zammit J, El-Zebdeh M. Arthroscopic excision of the fabella. J Knee Surg 2007; 20(4): 299-301.
[http://dx.doi.org/10.1055/s-0030-1248063] [PMID: 17993073]
, 16Okano E, Yoshioka T, Yanai T, et al. Fabella syndrome as an uncommon cause of posterolateral knee pain after total knee arthroplasty: A case report and review of the literature. Case Rep Orthop 2016; 2016: 4328462.
[http://dx.doi.org/10.1155/2016/4328462] [PMID: 27418991]
-20Matsuzaki A. Compression syndrome of the common peroneal nerve Das Kompressionssyndrom des Nervus peronaeus communis 1996; 8: 73-9.]. All publications were evidence level IV, with six case reports and four case series, the largest of which described 16 patients [9Weiner DS, Macnab I. The “fabella syndrome”: An update. J Pediatr Orthop 1982; 2(4): 405-8.
[http://dx.doi.org/10.1097/01241398-198210000-00010] [PMID: 6815224]
]. Across each publication, posterior knee pain was present in all patients. Other described features include posterolateral knee swelling, pain on direct pressure of the fabella, pain on extension of the knee, clicking sensations, and common peroneal nerve dysfunction. Fabella syndrome has been described secondary to total knee arthroplasty [16Okano E, Yoshioka T, Yanai T, et al. Fabella syndrome as an uncommon cause of posterolateral knee pain after total knee arthroplasty: A case report and review of the literature. Case Rep Orthop 2016; 2016: 4328462.
[http://dx.doi.org/10.1155/2016/4328462] [PMID: 27418991]
, 21Jaffe FF, Kuschner S, Klein M. Fabellar impingement: A cause of pain after total knee replacement. A case report. J Bone Joint Surg Am 1988; 70(4): 613-6.
[http://dx.doi.org/10.2106/00004623-198870040-00020] [PMID: 3356730]
-25Segal A, Miller TT, Krauss ES. Fabellar snapping as a cause of knee pain after total knee replacement: Assessment using dynamic sonography. AJR Am J Roentgenol 2004; 183(2): 352-4.
[http://dx.doi.org/10.2214/ajr.183.2.1830352] [PMID: 15269024]
], however, we describe the only known documented case of fabella syndrome occurring secondary to femoral de-rotation surgery.

The diagnosis of fabella syndrome is not always immediately clear, particularly in the presence of concurrent orthopaedic pathology - as highlighted in this case. Despite its innocuous appearance and rarity, the diagnosis should not be overlooked. A detailed history should include the presence of posterolateral knee pain, with the possibility of other symptoms as described above. Standard clinical examination must include palpation of the posterolateral structures of the knee. Reproducible pain upon applying pressure to the fabella suggests fabella syndrome as the underlying cause. Other pathologies including osteochondral fragments, foreign bodies, Baker’s cyst, meniscal tears, and localized pigmented villonodular synovitis, should be excluded prior to diagnosis. Standard anteroposterior and lateral radiographs of the knee should be utilised to reveal most bony fabella, with MRI utilised to aid in the diagnosis of smaller or cartilaginous fabellae. A Japanese cadaveric study found 66% of subjects to have the presence of a fabella - 29.3% of subjects had an entirely bony fabella, with the remainder displaying osteochondral or cartilaginous fabella [4Kawashima T, Takeishi H, Yoshitomi S, Ito M, Sasaki H. Anatomical study of the fabella, fabellar complex and its clinical implications. Surg Radiol Anat 2007; 29(8): 611-6.
[http://dx.doi.org/10.1007/s00276-007-0259-4] [PMID: 17882346]
].

In most of the reported cases, immediate and persistent pain relief was produced with fabellectomy. Conservative treatment with physiotherapy was found to be successful in a minority of cases. Zipple et al describe in detail the technique of manual mobilisation of the fabella to achieve pain relief [13Zipple JT, Hammer RL, Loubert PV. Treatment of fabella syndrome with manual therapy: A case report. J Orthop Sports Phys Ther 2003; 33(1): 33-9.
[http://dx.doi.org/10.2519/jospt.2003.33.1.33] [PMID: 12570284]
]. The largest case series described five out of sixteen patients (31%) responding to conservative treatment. The remaining eleven patients required surgery – all of which obtained immediate and persistent relief of symptoms [9Weiner DS, Macnab I. The “fabella syndrome”: An update. J Pediatr Orthop 1982; 2(4): 405-8.
[http://dx.doi.org/10.1097/01241398-198210000-00010] [PMID: 6815224]
].

Surgical excision has been described arthroscopically, arthroscopically assisted and directly via an open technique. Dannawi et al described arthroscopic excision of the fabella in two patients [14Dannawi Z, Khanduja V, Vemulapalli KK, Zammit J, El-Zebdeh M. Arthroscopic excision of the fabella. J Knee Surg 2007; 20(4): 299-301.
[http://dx.doi.org/10.1055/s-0030-1248063] [PMID: 17993073]
]. Provencher et al describe an arthroscopic assisted fabella excision, with initial arthroscopy to help delineate the borders of the fabella, prior to open excision [15Provencher MT, Sanchez G, Ferrari MB, et al. Arthroscopy-assisted fabella excision: Surgical technique. Arthrosc Tech 2017; 6(2): e369-74.
[http://dx.doi.org/10.1016/j.eats.2016.10.011] [PMID: 28580255]
]. The remaining fabellectomy procedures described within the literature review utilised an open technique. As per previous authors, an arthroscopic evaluation of the knee prior to excision of the fabella was performed to aid in delineating the borders of the fabella, which was not possible in this case. Subsequently, open excision was performed. In keeping with published literature, fabellectomy provided immediate and persistent pain relief.

No cases of Fabella Syndrome secondary to femoral de-rotation surgery were identified within the published literature. The patient, in this case, required a significant femoral correction (45 degrees) with consequent alteration in the relative tensions of the surrounding soft tissues in the post-operative period. The resultant Fabella Syndrome was thought to be caused secondary to the increased tensile stress of the fabella upon the lateral femoral condyle, as in keeping with other causes of Fabella Syndrome [5Müller W. The knee - form, function and ligament reconstruction. Knee - Form, Funct Ligament Reconstr 1982; 40, 96, 98, 192, 249, 252]. Increased soft tissue tension of the fabellofibular ligament, oblique popliteal ligament, and the lateral head of gastrocnemius tendon were all hypothesised to contribute towards an increase in the tensile stress of the Fabella articulating with the lateral femoral condyle and resultant Fabella Syndrome.

An alternative diagnosis within the differential of this patient following de-rotation surgery is that of common peroneal nerve irritation [10Mangieri JV. Peroneal-nerve injury from an enlarged fabella. A case report. J Bone Joint Surg Am 1973; 55(2): 395-7.
[http://dx.doi.org/10.2106/00004623-197355020-00019] [PMID: 4696171]
]. The common peroneal nerve may have been stretched following de-rotation surgery, to correct a 45-degree femoral malunion in this patient. A differential in the diagnosis could include common peroneal nerve irritation subsequent to this. As fabellectomy provided total resolution of the patient’s symptoms, with sparing of the peroneal nerve (which was protected intraoperatively), the diagnosis of Fabella Syndrome was made.

CONCLUSION

This paper describes the first reported case of Fabella Syndrome secondary to femoral de-rotation surgery. In keeping with the published literature of Fabella Syndrome secondary to other causes, fabellectomy provided total resolution of symptoms. The authors recommend fabellectomy as a definitive treatment for Fabella Syndrome secondary to femoral de-rotation surgery.

ETHICS APPROVAL AND CONSENT TO PARTICIPATE

Not applicable.

HUMAN AND ANIMAL RIGHTS

No Animals/Humans were used for studies that are base of this research.

CONSENT FOR PUBLICATION

Full patient written consent has been provided and available on request.

CONFLICT OF INTEREST

The authors declare no conflict of interest, financial or otherwise.

ACKNOWLEDGEMENTS

Declared none.

REFERENCES

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[http://dx.doi.org/10.1002/ca.22153] [PMID: 22933414]
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[http://dx.doi.org/10.1016/j.jus.2008.09.001] [PMID: 23396316]
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[http://dx.doi.org/10.2519/jospt.2003.33.1.33] [PMID: 12570284]
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[http://dx.doi.org/10.1055/s-0030-1248063] [PMID: 17993073]
[15] Provencher MT, Sanchez G, Ferrari MB, et al. Arthroscopy-assisted fabella excision: Surgical technique. Arthrosc Tech 2017; 6(2): e369-74.
[http://dx.doi.org/10.1016/j.eats.2016.10.011] [PMID: 28580255]
[16] Okano E, Yoshioka T, Yanai T, et al. Fabella syndrome as an uncommon cause of posterolateral knee pain after total knee arthroplasty: A case report and review of the literature. Case Rep Orthop 2016; 2016: 4328462.
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[http://dx.doi.org/10.2214/ajr.183.2.1830352] [PMID: 15269024]

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Sean L. Kitson
(Almac Sciences, Northern Ireland)

"In principle, all scientific journals should have open access, as should be science itself. Open access journals are very helpful for students, researchers and the general public including people from institutions which do not have library or cannot afford to subscribe scientific journals. The articles are high standard and cover a wide area."


Hubert Wolterbeek
(Delft University of Technology, The Netherlands)

"The widest possible diffusion of information is critical for the advancement of science. In this perspective, open access journals are instrumental in fostering researches and achievements."


Alessandro Laviano
(Sapienza - University of Rome, Italy)

"Open access journals are very useful for all scientists as they can have quick information in the different fields of science."


Philippe Hernigou
(Paris University, France)

"There are many scientists who can not afford the rather expensive subscriptions to scientific journals. Open access journals offer a good alternative for free access to good quality scientific information."


Fidel Toldrá
(Instituto de Agroquimica y Tecnologia de Alimentos, Spain)

"Open access journals have become a fundamental tool for students, researchers, patients and the general public. Many people from institutions which do not have library or cannot afford to subscribe scientific journals benefit of them on a daily basis. The articles are among the best and cover most scientific areas."


M. Bendandi
(University Clinic of Navarre, Spain)

"These journals provide researchers with a platform for rapid, open access scientific communication. The articles are of high quality and broad scope."


Peter Chiba
(University of Vienna, Austria)

"Open access journals are probably one of the most important contributions to promote and diffuse science worldwide."


Jaime Sampaio
(University of Trás-os-Montes e Alto Douro, Portugal)

"Open access journals make up a new and rather revolutionary way to scientific publication. This option opens several quite interesting possibilities to disseminate openly and freely new knowledge and even to facilitate interpersonal communication among scientists."


Eduardo A. Castro
(INIFTA, Argentina)

"Open access journals are freely available online throughout the world, for you to read, download, copy, distribute, and use. The articles published in the open access journals are high quality and cover a wide range of fields."


Kenji Hashimoto
(Chiba University, Japan)

"Open Access journals offer an innovative and efficient way of publication for academics and professionals in a wide range of disciplines. The papers published are of high quality after rigorous peer review and they are Indexed in: major international databases. I read Open Access journals to keep abreast of the recent development in my field of study."


Daniel Shek
(Chinese University of Hong Kong, Hong Kong)

"It is a modern trend for publishers to establish open access journals. Researchers, faculty members, and students will be greatly benefited by the new journals of Bentham Science Publishers Ltd. in this category."


Jih Ru Hwu
(National Central University, Taiwan)


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