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Removal of an Unusual Pedal Ossicle in an Elite Distance Runner: A Case Report
CASE REPORTS
Removal of an Unusual Pedal Ossicle in an Elite Distance Runner: A Case Report Amol Saxena, DPM, FACFAS A 24-year-old female national champion 5000-meter runner presented with an approximately 1-year duration of left foot pain localized to the medial arch. She had received multiple diagnoses and had undergone many different treatments, all of which had failed to relieve her symptoms. Careful review of her radiographic studies revealed an unusual ossicle in the region of her symptoms. Surgical excision of the not previously described ossicle localized within the posterior tibial tendon distal to the navicular adjacent to the first cuneiform resulted in complete resolution of the patient’s longstanding symptoms, and allowed her to return to competition at an elite level. Level of Clinical Evidence: 4 ( The Journal of Foot & Ankle Surgery 48(2):191–195, 2009) Key Words: accessory ossicle, runner, tarsal navicular, tibialis posterior tendon
P edal ossicles occur in various locations within the foot and ankle, and they can cause symptoms that may necessitate their removal when nonsurgical treatment methods fail to satisfactorily alleviate pain and disability. In this article, we describe the case of an elite athlete who suffered with recalcitrant pain caused by a heretofore-undescribed pedal ossicle that responded well to surgical excision and resumption of her high level of athletic activity. Case Report A 24-year-old female national champion 5000-meter runner presented with an approximately 1-year duration of left foot pain localized to the medial arch. She had received multiple diagnoses and treatments, including navicular and first cuneiform stress fracture, and posterior tibial tendon tear, with 2 sessions of immobilization with non–weight bearing for 8 weeks, combined with electrical bone growth
Address correspondence to Amol Saxena, DPM, FACFAS, Department of Sports Medicine, Palo Alto Medical Foundation, 795 El Camino Real, Palo Alto, CA 94301. E-mail: [email protected]. Podiatrist, Department of Sports Medicine, Fellowship Director, Palo Alto Medical Foundation, Palo Alto, CA. Financial Disclosure: None reported. Conflict of Interest: None reported. Copyright © 2009 by the American College of Foot and Ankle Surgeons 1067-2516/09/4802-0015$36.00/0 doi:10.1053/j.jfas.2008.12.002
stimulation. She had also undergone numerous sessions of physical therapy, and she used foot orthotic devices, one pair of which had been prescribed by the author of this article. All of these interventions had failed to relieve her symptoms. The clinical exam was relatively unremarkable other than the patient’s location of pain. She had mildly pronated feet that were symmetrical without any medial prominence or swelling, and she displayed equal limb length, no evidence of Tinel’s sign upon percussion of the posterior tibial nerve and its branches, absence of any lumbosacral radicular pain patterns, full muscle strength, and she used appropriate shoe gear and foot orthoses. There was no evidence of hyperesthesia, hyperemia, or hypohidrosis that could have been indicative of complex regional pain syndrome. The patient consistently stated that her pain emanated from the medial arch, and she pointed to the area of her foot just distal to the tuberosity and body of the tarsal navicular, dorsal to the plantar fascia, distal to the tarsal tunnel, and these findings seemed to be inconsistent with all of the previous diagnoses that were historically ascertained. At the time of her presentation to our clinic, she was in possession of a number of diagnostic images that had been previously obtained. The patient presented with radiographic studies including a magnetic resonance image (MRI), which, as reported by the radiologist who interpreted the images, showed signal change consistent with rupture of the distal fibers of the VOLUME 48, NUMBER 2, MARCH/APRIL 2009
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FIGURE 1 (A) Sagittal T1-weighted magnetic resonance image showing a bony ossicle at the plantar-medial aspect of the first (medial) cuneiform. (B) Short TI inversion recovery (STIR) axial magnetic resonance image showing inflammation around the ossicle and surrounding posterial tibial tendon. (C) Axial T2-weighted magnetic resonance image showing increased signal of the accessory ossicle.
posterior tibial tendon distal to the navicular attachment and irregularity of proximal plantar aspect of the first cuneiform. There was no signal change within the body of the navicular consistent with stress fracture, nor any abnormality of the plantar fascia and musculature (Figure 1). A technetium (99Tc) bone scan showed increased uptake in the medial navicular area and was interpreted as either a fracture or enthesiopathy of the posterior tibial tendon. A computerized 192
THE JOURNAL OF FOOT & ANKLE SURGERY
tomographic (CT) scan report was also read as negative for navicular and first cuneiform stress fracture, but did not state any other abnormalities (Figure 2). The author’s interpretation of the radiographic studies was of a sesamoidlike ossicle of the posterior tibial tendon within the distal attachment to the plantar-proximal aspect of the first cuneiform (exactly at the patient’s pain location), with tearing of the surrounding posterior tibial tendon fibers. This did not ap-
FIGURE 3 Lateral oblique radiographic view showing a bony ossicle at the plantar-medial aspect of the first (medial) cuneiform.
FIGURE 2 (A) Sagittal computerized tomographic image showing a bony ossicle at the plantar-medial aspect of the first (medial) cuneiform. (B) Axial computerized tomographic image showing a bony ossicle just distal to the tarsal navicular.
pear to be an avulsion fracture because the margins of the ossicle were smooth and there was no adjacent signal change at the first cuneiform as viewed with MRIs. Despite having undergone multiple radiographic studies, including plain radiographs, she had yet to be imaged with an oblique view of the symptomatic foot. The author was able to demonstrate this ossicle on a lateral oblique view of the foot (Figure 3). Interestingly, review of the literature failed to reveal an eponym for this ossicle. Because of the patient’s lack of relief from nonsurgical treatment and worsening symptoms such that she could not train effectively, the decision was made to proceed with surgical excision of the ossicle and repair of the posterior tibial tendon insertion.
FIGURE 4 Intraoperative view showing a bony ossicle at the plantar-medial aspect of the first (medial) cuneiform, and tearing of the posterior tibial tendon at its insertion.
Surgical Technique A curvilinear incision was created on the medial aspect of the left foot to expose the navicular tuberosity and the articulation with the first cuneiform. The posterior tibial tendon was identified and explored. The attachment to the navicular tuberosity appeared relatively secure but distally inferior to the navicular-cuneiform articulation; there was tearing noted of the tendon’s fibers; and a firm, smooth, rounded ossicle was visualized (Figure 4). The ossicle was excised (Figure 5), and close inspection failed to reveal any jagged or irregular margins that would be suggestive of a VOLUME 48, NUMBER 2, MARCH/APRIL 2009
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FIGURE 5 Excised ossicle.
FIGURE 6 Postoperative weight-bearing lateral radiograph showing anchor for reinforcement of posterior tibial tendon repair.
fracture. The plantar ligamentous and tendinous fibers were repaired with 2– 0 Fiberwire (Arthrex, Inc., Naples, FL) with the foot in a supinated position. To further add to the stability of the posterior tibial tendon, a bone anchor was placed in the inferior aspect of the navicular, since she was a high-level athlete and optimum strength would be required (Figure 6). Thereafter the wound was closed in anatomical layers. Postoperatively, the patient was placed in a below-theknee cast-boot, with the foot immobilized in a slightly supinated position and the ankle at 90°. She remained non–weight bearing on the operated foot for 3 weeks, and then bore weight in the cast-boot for 2 more weeks, at which time she was pain-free. The patient initiated range-of-motion exercises with her foot at 3 weeks postoperative, and used a stationary bike with the cast-boot, beginning at 2 weeks postsurgery. The patient removed the cast-boot to swim at 4 weeks postsurgery. At 5 weeks following the operation, she discontinued use of the cast-boot and returned to an athletic shoe with a foot orthosis and she initiated formalized physical therapy, including singlelegged strengthening. At 7 weeks postsurgery, she was able 194
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FIGURE 7 A runner using the Alter-G Treadmill (Menlo Park, CA), which was used by the patient described in this report when she resumed running. Use of this pressurized treadmill allowed the patient to gradually increase her effective body weight from an initially reduced weight, up to her actual body weight, from 7 to 10 weeks postsurgery.
to run on a special treadmill (Alter-G, Alter-G Inc., Menlo Park, CA) that allows patients to “reduce” their bodyweight (Figure 7). This specialized treadmill uses a pressurized chamber to provide a counterforce to the patient’s body weight, thereby unweighting the individual during the training regimen. In effect, the counter pressure reduces the patient’s effective weight on the running surface of the treadmill. Our patient ran on this treadmill initially at 70% of her bodyweight, progressively increasing to 95% over a 3-week period. Although the precise protocol for use of this equipment is still being established, patients should not exhibit any “limping” or antalgic postural guarding of the injured extremity, while running. At 10 weeks postsurgery, she was able to begin training outdoors. At 7 months postsurgery, she won her first competition following the operation. At 9 months postsurgery, she broke her first American record indoors, and by 1 year postsurgery, she had broken the American 5000-meter record outdoors. Thereafter, she went on to win the national championships and finished eighth in the world championships. After 2 years of postoperative follow-up, she won a medal in the 2008 Olympics. After the Olympics she had mild posterior tibial tendon symptoms that resolved with physical therapy, taping, and mild additional orthoses correction. She is currently training at full capacity with no limitations. Discussion Accessory ossicles of the foot are common. They can occur as osseous, incompletely ossified, and cartilaginous forms (1, 2). Authors have reported good results with exci-
sion of the commonly found accessory navicular and os trigonum (3, 4). The rehabilitation component for the treatment of excised accessory bones of the foot and ankle typically takes 3 or more months postsurgery before individuals can comfortably to return to their preoperative activity level (4, 5). The accessory bone excised in the patient presented in this case study did not appear to have been previously described in the published biomedical literature, and because of this, there was no information available to guide our choice of treatment and her subsequent rehabilitation efforts. Furthermore, the symptomatic bone appeared to be an ossicle, as it was embedded within the distal slip of the posterior tibial tendon. Even further, the smooth, round margins of the excised bone suggested ossicle, as compared with an avulsion fracture fragment that would have most likely presented with sharp and irregular margins without being embedded. Coughlin (2) described 3 accessory bones associated with the first cuneiform. One medial ossicle, known as os paracuneiforme, occurs near the medial aspect of the naviculocuneiform joint, and not plantar, as was the case in the patient described in this report. The second, termed os cuneometatarsale I tibiale, occurs near the medial aspect of the first metatarsocuneiform joint. An ossicle known as os cuneo-I metatarsale-I plantare occurs on the plantar aspect of the first metatarsocuneiform joint. Coughlin’s extensive and thorough review of accessory ossicles fails to reveal a bone similar in location to the one described in this case study. A small bipartite first cuneiform could possibly be
mistaken for the bony lesion observed in the patient described in this article; however, involvement of the insertion of the posterior tibial tendon is not described in association with a bipartite first cuneiform. In conclusion, elite and nonelite athletes may have atypical causes for foot pain. When patients with atypical and prolonged symptoms fail to respond to standard means, the practitioner should be alerted to the possibility of an unusual or anomalous skeletal structure. In this case report, surgical excision of a previously undescribed pedal ossicle associated with the insertion of the tibialis posterior tendon distal to the tarsal navicular, and adjacent to the first cuneiform, resulted in complete resolution and a full recovery in an elite runner. References 1. Champagne IM, Cook DL, Kestner SC, Pontisso JA, Siesel KJ. Os subfibulare. Investigation of an accessory bone. J Am Podiatr Med Assoc 89:520 –524, 1999. 2. Coughlin MJ. Sesamoids and accessory bones of the foot. In Surgery of the Foot & Ankle, ed 8, pp 531– 610, edited by MJ Coughlin, RA Mann, CA Saltzman, Mosby, St. Louis, MO, 2006. 3. Abramowitz Y, Wollstein R, Barzilay Y, London E, Matan Y, Shabat S, Nyska M. Outcome of resection of a symptomatic os trigonum. J Bone Joint Surg Am 85-A:1051–1057, 2003. 4. Kopp FJ, Marcus RE. Clinical outcome of surgical treatment of the symptomatic accessory navicular. Foot Ankle Int 25:27–30, 2004. 5. Requejo SM, Kulig K, Thordarson DB. Management of foot pain with accessory bones of the foot: two clinical case reports. J Ortho Sports Phys Ther 30:580 –594, 2000.
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Removal of an Unusual Pedal Ossicle in an Elite Distance Runner: A Case Report Amol Saxena, DPM, FACFAS A 24-year-old female national champion 5000-meter runner presented with an approximately 1-year duration of left foot pain localized to the medial arch. She had received multiple diagnoses and had undergone many different treatments, all of which had failed to relieve her symptoms. Careful review of her radiographic studies revealed an unusual ossicle in the region of her symptoms. Surgical excision of the not previously described ossicle localized within the posterior tibial tendon distal to the navicular adjacent to the first cuneiform resulted in complete resolution of the patient’s longstanding symptoms, and allowed her to return to competition at an elite level. Level of Clinical Evidence: 4 ( The Journal of Foot & Ankle Surgery 48(2):191–195, 2009) Key Words: accessory ossicle, runner, tarsal navicular, tibialis posterior tendon
P edal ossicles occur in various locations within the foot and ankle, and they can cause symptoms that may necessitate their removal when nonsurgical treatment methods fail to satisfactorily alleviate pain and disability. In this article, we describe the case of an elite athlete who suffered with recalcitrant pain caused by a heretofore-undescribed pedal ossicle that responded well to surgical excision and resumption of her high level of athletic activity. Case Report A 24-year-old female national champion 5000-meter runner presented with an approximately 1-year duration of left foot pain localized to the medial arch. She had received multiple diagnoses and treatments, including navicular and first cuneiform stress fracture, and posterior tibial tendon tear, with 2 sessions of immobilization with non–weight bearing for 8 weeks, combined with electrical bone growth
Address correspondence to Amol Saxena, DPM, FACFAS, Department of Sports Medicine, Palo Alto Medical Foundation, 795 El Camino Real, Palo Alto, CA 94301. E-mail: [email protected]. Podiatrist, Department of Sports Medicine, Fellowship Director, Palo Alto Medical Foundation, Palo Alto, CA. Financial Disclosure: None reported. Conflict of Interest: None reported. Copyright © 2009 by the American College of Foot and Ankle Surgeons 1067-2516/09/4802-0015$36.00/0 doi:10.1053/j.jfas.2008.12.002
stimulation. She had also undergone numerous sessions of physical therapy, and she used foot orthotic devices, one pair of which had been prescribed by the author of this article. All of these interventions had failed to relieve her symptoms. The clinical exam was relatively unremarkable other than the patient’s location of pain. She had mildly pronated feet that were symmetrical without any medial prominence or swelling, and she displayed equal limb length, no evidence of Tinel’s sign upon percussion of the posterior tibial nerve and its branches, absence of any lumbosacral radicular pain patterns, full muscle strength, and she used appropriate shoe gear and foot orthoses. There was no evidence of hyperesthesia, hyperemia, or hypohidrosis that could have been indicative of complex regional pain syndrome. The patient consistently stated that her pain emanated from the medial arch, and she pointed to the area of her foot just distal to the tuberosity and body of the tarsal navicular, dorsal to the plantar fascia, distal to the tarsal tunnel, and these findings seemed to be inconsistent with all of the previous diagnoses that were historically ascertained. At the time of her presentation to our clinic, she was in possession of a number of diagnostic images that had been previously obtained. The patient presented with radiographic studies including a magnetic resonance image (MRI), which, as reported by the radiologist who interpreted the images, showed signal change consistent with rupture of the distal fibers of the VOLUME 48, NUMBER 2, MARCH/APRIL 2009
191
FIGURE 1 (A) Sagittal T1-weighted magnetic resonance image showing a bony ossicle at the plantar-medial aspect of the first (medial) cuneiform. (B) Short TI inversion recovery (STIR) axial magnetic resonance image showing inflammation around the ossicle and surrounding posterial tibial tendon. (C) Axial T2-weighted magnetic resonance image showing increased signal of the accessory ossicle.
posterior tibial tendon distal to the navicular attachment and irregularity of proximal plantar aspect of the first cuneiform. There was no signal change within the body of the navicular consistent with stress fracture, nor any abnormality of the plantar fascia and musculature (Figure 1). A technetium (99Tc) bone scan showed increased uptake in the medial navicular area and was interpreted as either a fracture or enthesiopathy of the posterior tibial tendon. A computerized 192
THE JOURNAL OF FOOT & ANKLE SURGERY
tomographic (CT) scan report was also read as negative for navicular and first cuneiform stress fracture, but did not state any other abnormalities (Figure 2). The author’s interpretation of the radiographic studies was of a sesamoidlike ossicle of the posterior tibial tendon within the distal attachment to the plantar-proximal aspect of the first cuneiform (exactly at the patient’s pain location), with tearing of the surrounding posterior tibial tendon fibers. This did not ap-
FIGURE 3 Lateral oblique radiographic view showing a bony ossicle at the plantar-medial aspect of the first (medial) cuneiform.
FIGURE 2 (A) Sagittal computerized tomographic image showing a bony ossicle at the plantar-medial aspect of the first (medial) cuneiform. (B) Axial computerized tomographic image showing a bony ossicle just distal to the tarsal navicular.
pear to be an avulsion fracture because the margins of the ossicle were smooth and there was no adjacent signal change at the first cuneiform as viewed with MRIs. Despite having undergone multiple radiographic studies, including plain radiographs, she had yet to be imaged with an oblique view of the symptomatic foot. The author was able to demonstrate this ossicle on a lateral oblique view of the foot (Figure 3). Interestingly, review of the literature failed to reveal an eponym for this ossicle. Because of the patient’s lack of relief from nonsurgical treatment and worsening symptoms such that she could not train effectively, the decision was made to proceed with surgical excision of the ossicle and repair of the posterior tibial tendon insertion.
FIGURE 4 Intraoperative view showing a bony ossicle at the plantar-medial aspect of the first (medial) cuneiform, and tearing of the posterior tibial tendon at its insertion.
Surgical Technique A curvilinear incision was created on the medial aspect of the left foot to expose the navicular tuberosity and the articulation with the first cuneiform. The posterior tibial tendon was identified and explored. The attachment to the navicular tuberosity appeared relatively secure but distally inferior to the navicular-cuneiform articulation; there was tearing noted of the tendon’s fibers; and a firm, smooth, rounded ossicle was visualized (Figure 4). The ossicle was excised (Figure 5), and close inspection failed to reveal any jagged or irregular margins that would be suggestive of a VOLUME 48, NUMBER 2, MARCH/APRIL 2009
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FIGURE 5 Excised ossicle.
FIGURE 6 Postoperative weight-bearing lateral radiograph showing anchor for reinforcement of posterior tibial tendon repair.
fracture. The plantar ligamentous and tendinous fibers were repaired with 2– 0 Fiberwire (Arthrex, Inc., Naples, FL) with the foot in a supinated position. To further add to the stability of the posterior tibial tendon, a bone anchor was placed in the inferior aspect of the navicular, since she was a high-level athlete and optimum strength would be required (Figure 6). Thereafter the wound was closed in anatomical layers. Postoperatively, the patient was placed in a below-theknee cast-boot, with the foot immobilized in a slightly supinated position and the ankle at 90°. She remained non–weight bearing on the operated foot for 3 weeks, and then bore weight in the cast-boot for 2 more weeks, at which time she was pain-free. The patient initiated range-of-motion exercises with her foot at 3 weeks postoperative, and used a stationary bike with the cast-boot, beginning at 2 weeks postsurgery. The patient removed the cast-boot to swim at 4 weeks postsurgery. At 5 weeks following the operation, she discontinued use of the cast-boot and returned to an athletic shoe with a foot orthosis and she initiated formalized physical therapy, including singlelegged strengthening. At 7 weeks postsurgery, she was able 194
THE JOURNAL OF FOOT & ANKLE SURGERY
FIGURE 7 A runner using the Alter-G Treadmill (Menlo Park, CA), which was used by the patient described in this report when she resumed running. Use of this pressurized treadmill allowed the patient to gradually increase her effective body weight from an initially reduced weight, up to her actual body weight, from 7 to 10 weeks postsurgery.
to run on a special treadmill (Alter-G, Alter-G Inc., Menlo Park, CA) that allows patients to “reduce” their bodyweight (Figure 7). This specialized treadmill uses a pressurized chamber to provide a counterforce to the patient’s body weight, thereby unweighting the individual during the training regimen. In effect, the counter pressure reduces the patient’s effective weight on the running surface of the treadmill. Our patient ran on this treadmill initially at 70% of her bodyweight, progressively increasing to 95% over a 3-week period. Although the precise protocol for use of this equipment is still being established, patients should not exhibit any “limping” or antalgic postural guarding of the injured extremity, while running. At 10 weeks postsurgery, she was able to begin training outdoors. At 7 months postsurgery, she won her first competition following the operation. At 9 months postsurgery, she broke her first American record indoors, and by 1 year postsurgery, she had broken the American 5000-meter record outdoors. Thereafter, she went on to win the national championships and finished eighth in the world championships. After 2 years of postoperative follow-up, she won a medal in the 2008 Olympics. After the Olympics she had mild posterior tibial tendon symptoms that resolved with physical therapy, taping, and mild additional orthoses correction. She is currently training at full capacity with no limitations. Discussion Accessory ossicles of the foot are common. They can occur as osseous, incompletely ossified, and cartilaginous forms (1, 2). Authors have reported good results with exci-
sion of the commonly found accessory navicular and os trigonum (3, 4). The rehabilitation component for the treatment of excised accessory bones of the foot and ankle typically takes 3 or more months postsurgery before individuals can comfortably to return to their preoperative activity level (4, 5). The accessory bone excised in the patient presented in this case study did not appear to have been previously described in the published biomedical literature, and because of this, there was no information available to guide our choice of treatment and her subsequent rehabilitation efforts. Furthermore, the symptomatic bone appeared to be an ossicle, as it was embedded within the distal slip of the posterior tibial tendon. Even further, the smooth, round margins of the excised bone suggested ossicle, as compared with an avulsion fracture fragment that would have most likely presented with sharp and irregular margins without being embedded. Coughlin (2) described 3 accessory bones associated with the first cuneiform. One medial ossicle, known as os paracuneiforme, occurs near the medial aspect of the naviculocuneiform joint, and not plantar, as was the case in the patient described in this report. The second, termed os cuneometatarsale I tibiale, occurs near the medial aspect of the first metatarsocuneiform joint. An ossicle known as os cuneo-I metatarsale-I plantare occurs on the plantar aspect of the first metatarsocuneiform joint. Coughlin’s extensive and thorough review of accessory ossicles fails to reveal a bone similar in location to the one described in this case study. A small bipartite first cuneiform could possibly be
mistaken for the bony lesion observed in the patient described in this article; however, involvement of the insertion of the posterior tibial tendon is not described in association with a bipartite first cuneiform. In conclusion, elite and nonelite athletes may have atypical causes for foot pain. When patients with atypical and prolonged symptoms fail to respond to standard means, the practitioner should be alerted to the possibility of an unusual or anomalous skeletal structure. In this case report, surgical excision of a previously undescribed pedal ossicle associated with the insertion of the tibialis posterior tendon distal to the tarsal navicular, and adjacent to the first cuneiform, resulted in complete resolution and a full recovery in an elite runner. References 1. Champagne IM, Cook DL, Kestner SC, Pontisso JA, Siesel KJ. Os subfibulare. Investigation of an accessory bone. J Am Podiatr Med Assoc 89:520 –524, 1999. 2. Coughlin MJ. Sesamoids and accessory bones of the foot. In Surgery of the Foot & Ankle, ed 8, pp 531– 610, edited by MJ Coughlin, RA Mann, CA Saltzman, Mosby, St. Louis, MO, 2006. 3. Abramowitz Y, Wollstein R, Barzilay Y, London E, Matan Y, Shabat S, Nyska M. Outcome of resection of a symptomatic os trigonum. J Bone Joint Surg Am 85-A:1051–1057, 2003. 4. Kopp FJ, Marcus RE. Clinical outcome of surgical treatment of the symptomatic accessory navicular. Foot Ankle Int 25:27–30, 2004. 5. Requejo SM, Kulig K, Thordarson DB. Management of foot pain with accessory bones of the foot: two clinical case reports. J Ortho Sports Phys Ther 30:580 –594, 2000.
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