Tag Archives: soft tissue mass

Longitudinal plantar approach for excision of interdigital perineural fibroma of the foot: A case series and literature review

by George Flanagan1, Ian Reilly1,2*pdflrg

The Foot and Ankle Online Journal 9 (1): 1

Background:  There is an array of clinically and academically derived opinion as to the correct surgical approach to the intermetatarsal space, most frequently to excise a symptomatic interdigital perineural fibroma (aka Morton’s Neuroma).  The purpose of this retrospective case series was to evaluate the patient outcomes, and sequelae following a longitudinal plantar incision for excision of interdigital perineural fibroma.
Method: This is a retrospective case series of forty-two patients who underwent excision of interdigital perineural fibroma utilizing a longitudinal plantar incision between 2012-2014.  All patients were reviewed at two weeks, four weeks and six months post operatively.  Patient and clinician outcome measures were captured by means of the Manchester-Oxford Foot Questionnaire (MOXFQ) and Patient Satisfaction Questionnaire 10 (PSQ-10).
Results: Forty two patients (forty eight excisions) were assessed both pre operatively and post operatively.  95.2 percent of patients felt their foot was better or much better at six months post operatively.  The average post-operative PSQ-10 score was 85.05 (SD +/- 14.3).  A significant reduction in all three MOXFQ domains was observed.  Four patients had surgical sequelae; only one patient reported no improvement in pain post operatively.  No patients reported worsening symptoms.
Conclusion:  Producing excellent patient outcomes with only minimal complications, this case series substantiates the growing literature showing that excision of interdigital perineural fibroma through a longitudinal plantar incision is both safe and effective.

Key words: interdigital perineural fibroma, soft tissue mass, neuroma, fibroma

ISSN 1941-6806
doi: 10.3827/faoj.2016.0901.0001

1 – Department of Podiatric Surgery, Northamptonshire Healthcare Foundation Trust, England
2 – Private Practice, Northamptonshire, England
* – Correspondence: pod.surgeon@virgin.net


Interdigital perineural fibroma (Morton’s neuroma, interdigital neuroma, Morton’s metatarsalgia) is both a commonly described pathology as well as a frequently encountered affliction.  Although commonly affiliated with Thomas Morton’s description of 1876 (Morton, 1876), of only the third intermetatarsal space, arguably historic precedence can be attributed to either Civinini, (1835), Durlacher, (1845) or Tubby, (1912).

The incidence of interdigital perineural fibroma (IPF) is three-fold in the middle aged female population in comparison to their male counterparts (Naraghi et al, 2014). Symptoms commonly include paroxysmal pain and paresthesia of the pedal web space (most commonly the third) and adjacent digits.  The preferred description of an ‘entrapment neuropathy’ is now frequently utilized (Hassouna & Singh, 2005).  Despite the plethora of literature which exists for this common condition, a continuum of disparity regarding its etiology remains.

Diagnosis is by a thorough mixture of both symptomology and clinical examination.  Musculoskeletal ultrasound and magnetic resonance imaging have been shown to be sensitive diagnostic tools (Sharp et al, 2003).  There exists a treatise of nomenclature relating to this condition.  Although many historic reports entitle IPF as a ‘tumor’, recent histopathologic studies have formalized its histology as a perineural fibroma (Pazzaglia et al, 1996), that of inflammation and fibroblastic proliferation around a nerve.  Despite this, most studies cite an IPF as a benign swelling occurring at the bifurcation of the plantar interdigital nerve.

If symptoms of IPF are refractory despite exhausting conservative treatment modalities, then surgical intervention is indicated (Akermark et al, 2008).  Numerous surgical approaches and procedures have been described for this condition.

Disaccord is prevalent regarding incision placement, particularly whether the incision should be approached dorsally or plantarly (Wilson & Kuwada, 1995).  Evidence does not favor any particular approach to incision, despite this, many practitioners surreptitiously avoid a plantar incision due to concerns surrounding cicatrix, particularly hypertrophic scar formation and intractable plantar keratosis (Hassouna & Singh, 2005).  This case series highlights the method and outcomes relating to patients (n=42) who underwent complete excision of IPF via a longitudinal plantar incision.

Methods

Between 2012-2014 consecutive patients (n=97) who had refractory symptoms despite conservative treatment intervention underwent surgical excision.

Inclusion and exclusion criteria

Of the 97 patients who underwent surgical excision:

Those patients who had surgery performed utilizing a dorsal approach (n=11), patients who had undergone simultaneous bilateral procedures (n=5) and patients who underwent excision in combination with other surgical procedures (n=9) were excluded from the study.  Patients for whom post-operative outcome data was unavailable (n=30) were also excluded.  Patients who had undergone excision of a perineural fibroma unilaterally and in isolation were subsequently included (n=42).  Successive outcome data for at least 6 months was a prerequisite.

Surgical technique

All surgery was undertaken or supervised by the second author (IR).  Patients were all admitted on a day surgery ambulatory basis.  General anesthesia with regional local anesthetic block was used in a minority of patients (4, 9.5%), the majority of patients underwent surgery with regional ‘ankle block’ anesthesia alone.  Standard surgical asepsis and infection control measures were observed.  Surgery took place within a laminar airflow (Howorth Exflow 90) operating theatre.  The procedure was performed utilizing a pneumatic ankle tourniquet set at 80-100 mmHg above the patients’ recorded systolic blood pressure.

A longitudinal plantar incision was made (Figure 1).  The incision extends from the edge of the adjacent web space distally to just beyond the level of the metatarsal heads proximally.  Vessel management was undertaken utilizing bipolar cautery.

Fig1

Figure 1 Plantar longitudinal incision.

Fig2 

Figure 2 Highlighting digital bifurcation.

Fig3

Figure 3 Proximal tracking of nerve.

Fig4

Figure 4 Wound closure.

Utilizing blunt dissection carefully, navigating through the plantar fat pad, the pathological nerve is identified, perineural adhesions were bluntly released ensuring integrity of the surrounding soft tissue architecture.  Visualization and adhesion neurolysis was performed distally at the level of digital bifurcation (Figure 2).

Proximally blunt dissection was undertaken (Figure 3) until normal nerve diameter and visualization was encountered.

The plantar digital nerve was resected distally at its digital bifurcation, and proximally where approximately 10mm of normal nerve had been visualized.  The remaining proximal nerve stump was cauterized and displaced proximally.  Hemostasis was achieved again using bipolar cautery.  The surgical site was then flushed with saline and skin closure achieved with 3-0 monofilament polypropylene suture utilizing a horizontal mattress technique (Figure 4).

Post-operative management

Protocol for post-operative analgesia included co-codamol 30/500 one or two tablets QDS and diclofenac 50mg TDS (PRN).  Co-codamol was replaced for Tramadol 50mg QDS in cases of allergy or intolerance.  All patients were placed in a compression dressing utilizing a universal post-operative shoe (Benefoot) for four weeks, during which time they were allowed to weight bear for essential activities of daily living.  Patients received a wound review at two weeks and removal of sutures at four weeks.  Patients were seen finally for six month review.

Data capture and statistics

Patient reported outcome measures were recorded using paper questionnaires both pre-operatively on the day of surgery and post operatively at 6 month review.  Pre and post operatively patients were asked to complete the Manchester-Oxford Foot Questionnaire (MOXFQ) (Dawson et al, 2006) and post-operatively. In addition to the MOXFQ, patients completed The Patient Satisfaction Questionnaire 10 (PSQ-10) (Rudge & Tollafield, 2003).  Data was inputted electronically to the Podiatric Audit of Surgery and Clinical Outcome Measure (PASCOM-10) system.  During post-operative review clinician determined outcome data was also collected.

Statistics and calculations were achieved using Microsoft Excel (2010 Microsoft Corporation).

Results

Of the patients included in the study (n=42), 33 (78.5%) were female with a mean age of 52.9 (SD +/- 11.8) years (range 28-72) and 9 (21.5%) were male with a mean age of 54.8 (SD +/- 12.8) years (range 44-72).  In 25 (59.5%) cases the left foot was involved, leaving 17 (40.5%) incorporating the right foot.  Neuromas were excised in isolation in 20 (47.6%) cases from the 3rd intermetatarsal space and 16 (38.1%) from the 2nd intermetatarsal space, 6 (14.3%) cases involved both the 2nd and 3rd intermetatarsal space.  Histological confirmation of perineural fibroma was obtained in all samples excised, totaling 48 from 42 feet. (Example of excised sample sent to histology seen in Figure 5).

Fig5

Figure 5 Excised interdigital perineural fibroma.

Of the 42 patients in the study, 4 cases of surgical sequelae were recorded.  Sequelae were recorded during post-operative visits by the attending clinician utilizing electronic data capture. Of the 4 patients, 2 patients (4.7%) suffered with scar line hypertrophy, one patient required surgical revision of the scar with good long term results at six month follow up.  One patient (2.4%) had a suspected stump neuroma at 6 months post op, this patient achieved asymptomatic longevity with one infiltration of 40mg Depo-Medrone, subsequently not requiring further surgery. One patient (2.4%) had a suspected (not proven) superficial infection treated with a 7 day course of antibiotics with no long term negative effect.

Patient satisfaction was captured utilizing the PSQ10.  At 6 months post-surgery 40 (95.2%) patients felt that their foot was better or much better than compared to the original problem.  One patient (2.4%) noted that their foot felt ‘the same’ when compared to the original problem, it was this patient who subsequently required revision of hypertrophic scar formation.  One patient (2.4%) did not state how they felt.  When asked ‘would you have surgery again under the same circumstances?’, 40 (95.2%) said yes and 2 (4.8%) said no.  The majority of patients (13, 31%) were back into preferred footwear by 8 weeks, 2 patients (4.8%) by 2 weeks; 10 (21.4%) by 4 weeks; 12 (28.6%) by 6 weeks with 6 (14.3%) patients waiting until 6 months until they were normally shod.   The average PSQ10 score was 85.05 (SD +/- 14.3).

Figure 6 illustrates the MOXFQ score distribution across the 3 patient reported outcome domains.  In the walking / standing (WS) domain the average pretreatment score was 68.8 (SD +/- 21.5), the post treatment score reduced to 23.1 (SD +/- 27.4).  For social interaction (SI) scores reduced from 51.3 (SD +/- 22.3) to 14.4 (SD +/- 19.5) and in the pain (P) category, 67.1 (SD +/- 18) and 24.6 (SD +/- 22.3) respectively.  Scores in each domain exceeded the threshold (WS 12.8, SI 20.3 and P 4.6) for a clinically significant difference as defined by Dawson et al, (2007).

Fig6

Figure 6 Pre/post op comparative MOXFQ distribution.

Discussion

Interdigital perineural fibroma are a common clinical entity, despite this, there remains disparity both academically and subjectively about its etiology, pathophysiology and long term treatment.  However it is uniformly agreed that surgical intervention is required once conservative treatment has failed (Akermark et al 2013).  Despite this surgical consensus and the vast amount of literature pertaining to it there remains druthers amongst surgeons regarding the surgical approach which should be taken.

Numerous approaches to the intermetatarsal space have been described in the literature, including minimally invasive techniques (Zelent at al 2007). Minimally invasive surgical approaches to excision of IPF have been available for a number of years (Barrett & Pignetti, 1996), however such approaches are often utilized when performing procedures alternative to excision of the pathological nerve such as decompression of the deep intermetatarsal ligament (Dellon, 1992).  Excision of the fibrosed nerve in toto is the most common procedure of choice (Jain & Mannan, 2013), for this, four main approaches to the intermetatarsal space have been described, which include; the longitudinal plantar, transverse plantar, web space approach and longitudinal dorsal.

Patient satisfaction has shown to be high following IPF excision (Thomson et al, 2004).  Despite this, reports state satisfaction rates as low as 65% (Nashi et al, 1997) and as high as 100% (Colgrove et al, 2000) utilizing various surgical approaches and methods.  As such Thomson’s Cochrane review (Thomson et al, 2004) concluded that there was insufficient evidence to assess formally the effectiveness of both operative and conservative treatment for IPF.  Further appraisal of the literature also shows discourse on failure rates following surgical intervention, ranging from 3% (Giannini et al, 2004) to as high as 24% (Younger & Claridge, 1998).

Akermark et al (2013) performed one of the first prospective randomized controlled trials providing comparative data for dorsal vs longitudinal plantar approaches to excision of IPF.  Patient satisfaction and pain reduction were comparable between both groups.  A reduction in daily activity restriction was noted in both dorsal and plantar groups, 67% and 77% respectively.  Pain reduction was 96% in the plantar incision group and 97% in the dorsal group. Other studies citing similar satisfaction rates are available.  Akermark et al, (2008) in an earlier study found 86% satisfaction rates following plantar incision, Jerosch et al, (2006) showed 92% satisfaction following longitudinal plantar incision.  Wilson & Kuwada, (1995) in a 15 year follow up study of 29 patients (59 neuromas) found preference for the plantar transverse incision when compared to a dorsal incision, with 68% satisfaction rate following dorsal incision and 100% satisfaction in the plantar approach group.  Barbosa et al, (2005) & Nery et al, (2012) also advocated a plantar transverse incision reporting 89.5% and 89.4% satisfaction respectively. Similar favorable data exists for surgery performed utilizing a longitudinal dorsal incision.  Lee et al, (2011) followed patients over 10 years, 61.4% of patients were completely satisfied, however they broke down a further 38% that were satisfied with reservations, totaling an overall 99.4% satisfaction rate.

Coughlin & Pinsonneault, (2001) using a median follow up of sixty nine months found patient satisfaction high at 84% of 66 patients (74 neuromas), good / excellent results were also compiled into satisfaction rates in studies by Giannini et al, (2004) and Kasparek & Schneider, (2013) citing 77% and 76.5% respectively.  Whilst no significant difference has been concluded in the literature, particularly that comparing multiple surgical approaches (for which there are few) patient satisfaction does seem to be slightly higher in those studies utilizing the plantar incision (both longitudinal and transverse).  Two very recent studies are also showing high patient satisfaction rates following the plantar approach (Killen et al, 2015 & Kundert et al 2015), this study adds to that data, showing a patient satisfaction rate of 95.2%.  Despite there being no significant difference in patient outcomes with either approach, as noted previously in this article, there seems to be a subjective preference for dorsal incision due to concerns regarding surgical sequelae (McKeever, 1952).

Whilst concern is derived from the notion of increased risk of intractable plantar keratosis and hypertrophic scarring following a plantar approach to excision of IPF, there remains a multitude of literature showing relatively comparable rates of surgical sequelae for both dorsal and plantar approaches.  Akermark et al, (2013) studied 97 patients and found 5 complications following IPF excision through a plantar incision and 6 through a dorsal incision.  Of the plantar group three cases of small ‘pea sized’ hypertrophic scar formation and one large hypertrophic scar were noted, all were revised with superficial excision and included in the final satisfaction results.  Of the 6 complications from the dorsal group these included one resected artery as oppose to IPF (confirmed by histology), one infection, one dehiscence, one deep vein thrombosis and two patients with recalcitrant pain requiring revision surgery through a plantar incision.  Patients reported sensory loss 85% of the time in the dorsal group compared to 66% in the plantar group.  Jerosch et al, 2006 noted 2.8% of patients with problematic scars and 1.7% infection in a large cohort of 356 patients.  Barbosa et al, 2005 found one patient of a nineteen cohort to have dehiscence, with no further complications cited.  Kundert et al, 2015 in a midterm follow up study also found favorable scar sequelae with a 5.3% symptomatic scar rate in 51 feet.  Like Akermark et al, (2013), Wilson & Kuwada, (1995) compared complication rates between both dorsal and plantar approaches, in a 15 year follow up they found a 6.7% complication rate for plantar incision and a 17% complication rate for the dorsal approach.

Similar to Akermark et al, (2013) the plantar group had two cases of problematic scar formation; however, this mirrored the dorsal group who also reported two patients with pathological scar formation.  In addition to the two patients suffering with scar tenderness, the dorsal group also had six reported cases of recurrent neuroma, one hematoma and one deep vein thrombosis.  Nery et al, (2012) found a lower incidence of complication compared to other studies (6.9% patients reported a ‘fair result’), however unlike other studies focusing on plantar incisions they had a 3.8% stump re-occurrence, confirmed via histology following revisional surgery.  Studies focusing on the longitudinal dorsal approach show no significant difference in complication rates when compared to plantar incision, however the specific type of surgical sequelae differs with approach.  Lee et al, (2011), Wilson & Kawada, (1995) and Akermark et al, (2013) all report significant discomfort related to sensory loss following IPF excision through a dorsal approach with two of these studies directly comparing dorsal and plantar incisions.  The results of the two studies (Wilson & Kuwada, (1995) & Akermark et al, 2013) which directly compare multiple surgical approaches have already shown the surfeit of sequelae which may arise following a dorsal approach, in addition to this, Coughlin & Pinsonneault, (2001) also found problems related to ‘uncomfortable’ paresthesia post operatively as well as six superficial infections, one deep infection, a case of complex regional pain syndrome and adjacent metatarsophalangeal joint instability.  In this case series the complication rate deduced was 9.6% (4 patients) which averaged between the literature scrutinized, however given the small sample size an elevated complication percentage is expected.  Of the four patients with complications, two patients had satisfactory outcomes, one patient chose not to comment and one patient (2.4%) was deemed to have a failed outcome; ultimately culminating in a 95.2% satisfaction rate.

Conclusion

Whilst we have shown that there is a risk of cicatrix’s following surgery approached through the plantar surface of the foot, research infers that there is no evidence to suggest inferior patient outcomes following this approach, particularly when compared to alternative surgical approaches.  In fact the risk of adverse scarring following this approach is comparable with data derived from cohorts utilizing a dorsal incision, thus negating the argument of mechanically induced scar hypertrophy.  In fact an interesting outcome of the Coughlin & Pinsonneault, (2001) study was the 15% incidence of post-operative intractable plantar keratosis development around an adjacent metatarsophalangeal joint following a dorsal incision. This could be a potential side effect of division of the deep intermetatarsal ligament, which requires further research in the future.

This case series produced excellent patient outcomes, in common with recent literature; a significant reduction in pain with increased mobility and social inclusion.  The existing literature already justifies the longitudinal plantar approach to excision of intermetatarsal perineural fibroma, showing a low (and less varied) complication rate: this case series substantiates this.

References

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Second Metatarsophalangeal Joint Pigmented Villonodular Synovitis: A case report

by John A. Rialson, DPM, Eric J. Heit, DPM, FACFAS

The Foot and Ankle Online Journal 4 (9): 2

Pigmented Villonodular Synovitis (PVNS) is an uncommon condition of the foot. PVS of the foot accounts for 2% of all cases. We present the case of a 26 year-old male who was found to have pigmented villonodular synovitis of the second metatarsophalangeal joint. Although a high incidence is reported in the literature, the patient underwent surgical excision with no known reoccurrence to date. Findings from magnetic resonance imaging, surgical presentation and literature review is presented.

Key words: Pigmented villonodular synovitis, metatarsophalangeal joint, foot surgery, soft tissue mass.

Accepted: August, 2011
Published: September, 2011

ISSN 1941-6806
doi: 10.3827/faoj.2011.0409.0002


Pain to the second metatarsophalangeal joint (MTP) is a common complaint encountered in the foot and ankle surgeon’s office. While reasons for this presentation are vast, a soft tissue mass could be a cause of this pain. Proper work up of the patient that includes a thorough history and physical, basic radiographs, and advanced imaging studies can aid in an accurate diagnosis.

Pigmented villonodular synovitis (PVNS) is a fairly rare disease. The incidence of PVNS is 1.8 patients per million people. [1] Pigmented villonodular synovitis is a proliferative disorder of synovial tissue in the joint, tendon sheath, and/or bursa.

The common locations for PVNS are the knee, hip and ankle. Two percent of these lesions are in the foot. [2] Bone and joint destruction can occur with this disease which makes early, accurate diagnosis and treatment essential. Furthermore, despite appropriate management by surgical excision [3], PVNS has a high recurrence rate. In the foot and ankle, recurrence rate has been reported as 14.3%. [4]

Case Report

A 26 year-old male presented to the institution of the senior author with right foot pain that he had for a few weeks. The patient was unsure of the cause for the pain, but did remember feeling a pop to his right foot while performing yoga. He initially did not seek care as he was not in a lot of discomfort, but progressively his second MTP joint became more painful.

The patient was able to point to his second MTP joint as is primary location of pain. Upon presentation, the area was so painful that he was using a crutch to take the pressure off the area. No other forms of treatment have been attempted prior to evaluation.

Clinical evaluation of the area revealed mild edema over the dorsal aspect of the second MTP joint without erythema or warmth. There was no significant deformity or instability, and neurovascular status was intact with an otherwise benign exam. Initial diagnostic imaging consisted of anteroposterior, medial oblique, and lateral foot radiographs, which were normal. There were no obvious signs of Freiberg’s infraction. The only finding was a very mild joint abnormality with a subchondral cyst that was appreciated on the oblique view.

The initial diagnosis was capsulitis of the second MTP joint with possible osteochondral defect. Initial treatment consisted of CAM boot application and discussion of further imaging if not improving. The plan was to attempt this treatment for three weeks and reevaluate his foot with the option to obtain an magnetic resonance imaging (MRI).

The patient presented for follow up after the initial visit and stated that overall his condition felt worse. After the initial treatment, he experienced about five days of pain relief but developed increased pain since the initial relief. Once again, pain was localized to the second MTP joint. Clinically, there was a reduction of swelling to the area; however, with the decrease in swelling, a subcutaneous palpable mass was now present on the dorsal aspect of the second MTP joint. The contour of the metatarsal could also then be palpated with no obvious findings. The decision was then to obtain an MRI scan of the right foot.

The official findings of the MRI are as follows: “. . . markedly abnormal thickening and enhancement of the synovium surrounding the second metatarsophalangeal joint, with minimal associated joint fluid seen. Within the second MTP joint, there is abnormal signal within the metatarsal head with potential collapse of the cortical surface.” (Figs. 1A, 1B and 1C) The radiologist’s impression was a differential consideration to include a severe synovitis or other primary synovial abnormality such as PVNS. Also mentioned was no involvement of other joints to suggest a polyarticular arthropathy such as rheumatoid arthritis.

  

Figures 1ABC MR T1 image of the second MTP joint showing the soft tissue mass. (A)  An Axial image showing the mass to the second metatarsal. (B)  A STIR image of the soft tissue mass as well as showing the signal to the metatarsal head. (C)

With the above findings, the results of the MRI and treatment options were discussed with the patient. A surgical excision was planned and performed without complications. Intraoperatively after the incision and initial dissection, a lobular pigmented soft tissue mass, consistent with PVNS, was encountered. (Fig. 2) The soft tissue mass was dissected, removed, and sent for pathologic examination. (Fig. 3) Upon removing the mass, a cartilage defect to the second metatarsal head was appreciated. (Fig. 4) This finding was anticipated with the MRI but with carefully examination was a substantial deficit. The decision was made to fenestrate the head of the second metatarsal to promote fibrocartilage growth. (Fig. 5)

Figure 2 An intraoperative picture of the soft tissue mass consistent with PVNS.

Figure 3 The soft tissue mass removed from the second MTP joint and sent for examination. 

Figure 4 Operative finding of the cartilage defect to the head of the second metatarsal.

Figure 5 Operative photo showing the cartilage defect to the second metatarsal after fenestration.

The pathology report confirmed the diagnosis consistent with PVNS. He had an uneventful post operative course and at last exam had no tenderness with palpation. The second digit was in a plantigrade position and had good range of motion across the MTP joint. The patient is now over one year after the procedure and there has been no known reoccurrence to date.

Discussion

Soft tissue masses of the foot and ankle often present with complaints of pain and/or irritation with footwear. Pigmented villonodular synovitis, while most common in the knee, hip and ankle, can present in the foot, although they are most commonly seen in the rearfoot and ankle2. Nabeshima, et al., does describe a case report of PVNS to the first MTP joint. [9]

Carpintero, et al., [2] reported eight cases of PVNS in the foot. Five of the eight cases were reported in the rearfoot and the remaining three cases in the forefoot. The locations they reported were the talus, first cuneiform, first and fifth metatarsals, and middle phalanx. Interestingly, they also reported that there was radiographic bone involvement in six patients.

Similarly, Ghert, et al., [5] documented the rarity of PVNS in the foot and ankle with a review of six cases from 1978-1997. Of the six of their cases, two were isolated to the ankle joint. The remaining four cases involved the subtalar joint, midfoot and two cases in the metatarsal region. Lisfranc amputation was a treatment for one of the cases.

The invasive nature of PVNS is also an important aspect. This is described with bone involvement being found in seven of eight cases that Rochwerger et al. reported between 1981 and 1997. [6] Surgical treatment consisted of arthrodesis in six cases, one ankle synovectomy, and one toe amputation.

In our patient’s case, as well as in the literature, the use of MRI aids in the diagnosis of PVNS. The description of PVNS on MRI may have certain findings consistent with other synovial lesions. On T1-weighted images, the lesions will have low or intermediate signal intensity. Pigmented villonodular synovitis will also demonstrate low signal intensity with T2-weighted and gradient-echo images. The theory is that the hemosiderin in PVNS produces this low signal intensity on T2-weighted imaging. [7]

The final diagnosis of PVNS can be confirmed with pathological examination. Specimens may appear villous, nodular, or villonodular with often prominent hemosiderin deposition seen in most cases. [8] Histological findings of PVNS are hyperplastic synovium, giant cells, and mononuclear histiocytes with varying extent of hemosiderin deposition. [3,7,8]

While the literature states the PVNS is rare in the foot and of unknown etiology [2], some theories of etiology that are often debated are an inflammatory process, neoplasia, or lipid metabolism disorder. [8] The neoplastic theory is becoming more strongly supported by the discovery of cytogenetic aberrations. Mild repetitive trauma with hemorrhage into the joint also could explain the inflammatory nature and the hemosiderin of PVNS. One could speculate that this may be the origin in our patient with his yoga history.

References

1. Myers BW, Masi AT. Pigmented villonodular synovitis and tenosynovitis: a clinical epidemiologic study of 166 cases and literature review. Medicine 1980 59: 223-238.
2. Carpintero P, Gascon E, Mesa M, Mesa M, Jimenez C, Lopez U. Clinical and radiographic features of pigmented villonodular synovitis of the foot: Report of eight cases. JAPMA 2007 97: 415-419.
3. Brien EW, Sacoman DM, Mirra JM. Pigmented villonodular synovitis of the foot and ankle. Foot Ankle Int 2004 25: 908-913.
4. Sharma H, Jane MJ, Reid R. Pigmented villonodular synovitis of the foot and ankle: Forty years of experience from the Scottish Bone Tumor Registry. J Foot Ankle Surgery 2006 45: 329-336.
5. Gher MA, Scully SP, Harrelson JM. Pigmented villonodular synovitis of the foot and ankle: A review of six cases. Foot Ankle Int 1999 20: 326-330.
6. Rochwerger A, Groulier P, Curvale G, Launay F. Pigmented villonodular synovitis of the foot and ankle: A report of eight cases. Foot Ankle Int 1999 20: 587-590.
7. Llauger J, Palmer J, Monill JM, Franquet T, Bague S, Roson Nl. MR imaging of benign soft-tissue masses of the foot and ankle. Radiographics 1998 18: 1481-1498.
8. Murphy MD, Rhee JH, Lewis RB, Fanburg-Smith JC, Flemming DJ, Walker EA. Pigmented villonodular synovitis: Radiologic-pathologic correlation. Radiographics 2008 28:1493-1518.
9. Nabeshima Y, Mori H, Mitani M, Nagura I, Ozaki A, Fujii H, Doita M. Diffuse pigmented villonodular synovitis in the metatarsophalangeal joint of the Hallux: A Case Report. J Foot Ankle Surgery 2009 48: 573-576.


Address correspondence to: Swedish Podiatric Surgical Residency Program, Swedish Medical Center, 747 Broadway Seattle, WA 98122
1  Submitted while Chief Resident, Swedish Podiatric Surgical Residency Program, Swedish Medical Center, Seattle, WA
2  Attending Surgeon, Swedish Podiatric Surgical Residency Program, Virginia Mason Medical Center, Seattle, WA

© The Foot and Ankle Online Journal, 2011

Lymphangioma of the Foot: A case report

by Steven F. Boc, DPM,FACFAS,FACFAOM , Panagiotis Panagakos, DPM , Soorena Sadri, DPM 

The Foot and Ankle Online Journal 4 (2): 1

Vascular Malformations (VM) are localized or diffuse errors of embryonic development that may affect any segment of the vascular tree, including arterial, venous, capillary and lymphatic vessels. Lymphangiomas are rare vascular lymphatic malformations (LM). The rate of occurrence in the foot and ankle regions has been scarcely reported. Lymphangiomas are slow flow vascular malformations and are histological classified into capillary, cavernous and cystic. Treatment options for lymphangiomas include schlerotherapy, the use of angiogenesis inhibitors; however complete surgical resection is the recommended treatment.

Key words: Lymphangioma, tumor, lymphatic malformations, vascular malformation, soft tissue mass, hemangioma, foot, vascular anomaly.

Accepted: January, 2011
Published: February, 2011

ISSN 1941-6806
doi: 10.3827/faoj.2011.0402.0001


Lymphangiomas are rare soft tissue vascular malformations that can appear on the body. [1,2] Lymphangiomas of the foot and ankle is a rare occurrence.[3] Lymphangiomas are equally distributed among genders and races. In the literature, their incidence in children is estimated to amount to 6% of all benign tumors. In about half of the patients the disease is already obvious at the time of birth. The tumor commonly appears as a ballotable mass and the overlying skin is usually normal, but may have a bluish hue. Lymphangiomas of the extremity can cause diffuse swelling or localized swelling or gigantism with soft tissue and skeletal overgrowth. They are commonly misdiagnosed as Hemangiomas. [1,4,5,6]

Vascular malformations (VM) are localized or diffuse errors of embryonic development that may affect any segment of the vascular tree, including arterial, venous, capillary and lymphatic vessels. During Embryogenesis the development of the vascular system occurs by two separate but related processes; vasculogenesis and angiogenesis. Development of the lymphatic system begins in the sixth to seventh week of gestation, approximately four weeks after the onset of vasculogenesis. Existing veins give rise to lymph sacs which then bud lymphatic capillaries in a centrifugal manner. There is an in situ differentiation of lymphangioblasts from mesenchymal cells into lymphatic endothelial cells with subsequent recruitment of these cells into developing lymphatic vessels.

Vasculogenesis, angiogenesis and lymphangiogenesis are subject to precise regulation of growth factors, intercellular and extracellular matrix signaling molecules including vascular endothelial growth factor (VEGF) and the VEGF receptor families, angiopoietins and the Tie-2 receptor, transforming growth factor-β and its receptor, PDGF-B and its receptor, the Notch and Jagged families of membrane associated molecules and the integrin family of cell surface receptors. The integrins which mediate interactions within the extracellular matrix are important in the formation of the vascular and lymphatic systems. Mutations or deletions in specific integrin subtypes can lead to abnormal lymphatic development. [4,5] In 2007 Skandalakis, et al., described the formation of the lymphatic system which they clearly stated is an enigma. They report the formation of a lymphangiomas as a very rare phenomenon. [2]

The greatest impediment for the diagnosis and treatment of vascular anomalies has been the confusing terminology. [5] A biologic classification system introduced in 1982 based on studies correlating physical findings, natural history and cellular features has clarified most of the terminologic disorder. There are two major types of vascular anomalies: tumors and malformations. Vascular tumors are endothelial neoplasms characterized by increased cellular proliferation. Hemangioma is the most common and is almost exclusive to infants. Vascular malformations are the result of abnormal development of vascular elements during embryogenesis and fetal life. These may be single forms (capillary, arterial, lymphatic or venous) or a combination. Vascular malformations do not generally demonstrate increased endothelial turnover. They are designated according to the predominant channel type as capillary malformations, lymphatic malformations (LMs), venous malformations (VMs), arteriovenous malformations, and complex forms such as capillary-lymphatico-venous malformations. Malformations with an arterial component are fast-flow while the remainders are slow-flow. [4,5,6]

Case Report

A 25 year-old female presented to our office in April of 2009 with complaints of multiple discolored and enlarged painful masses on the plantar aspect of her left foot. (Figs 1A, 1B, and 1C) The masses have been present since early childhood but recently have caused her pain while ambulating. Radiographs were obtained upon initial presentation but were unremarkable. Aspiration of the masses was attempted in the office. The aspirate contained dark red fluid which was sent for culture, which came back negative for any infective process. It was decided at this point to send the patient for a series of diagnostic exams. Bilateral lower extremity venous ultrasound resulted in no evidence of a deep vein thrombosis (DVT). Bilateral ankle brachial indexes were unremarkable as well.

Figure 1A, 1B and 1C Initial presentation of lymphangioma on plantar aspect of foot.(A),  A more oblique view of the lesion. (B). A more lateral view of the lesions. (C)

Magnetic resonance imaging (MRI) was performed for the left foot which showed an enhancing soft tissue mass involving the quadrates plantae, flexor digitorum brevis and flexor hallucis muscle bellies consistent with a hemangioma. (Figs. 2A, 2B and 2C) Due to the extensive nature of the soft tissue masses and severe pain it was determined that surgical excision be performed. The patient received a popliteal nerve block pre operatively for pain control. One plantar incision was used to access the most painful soft tissue masses. It was noted that once the ankle tourniquet was inflated the masses decreased in size.

Figure 2A, 2B and 2C MRI of lymphangioma; sagittal view (A), Axial view (B) and Coronal view. (C)

The masses were excised and sent to pathology for evaluation. (Figs. 3A, 3B and 3C) The final report by the pathologist was dense fibrocollagenous tissue, nerve bundles and few dilated endothelial lined spaces and enlarged lymphatic sacs suggestive of a lymphangioma. (Figs. 4A and 4B)

Figure 3A, 3B and 3C Intra operative removal of the lymphangioma (A and B).  Removed lymphangioma on the back table. (C)

Figure 4A and 4B 10x hematoxylin & eosin slide, shows the presence of many dilated vascular channels lined by flattened endothelial cells and containing lymphatic fluid. (A)  The 40x hematoxylin & eosin slide, shows the presence of many dilated vascular channels lined by flattened endothelial cells and containing lymphatic fluid.

The patient returned the next day to the emergency department in excruciating pain and unable to bear weight on the left foot. There were no signs of infection present at this time (Fig. 5A). Patient was admitted for pain management and was discharged 4 days later when her pain was controlled by simple oral analgesics. Patient presented to the office three weeks after excision with a dehiscence of the surgical site. She was admitted at this point and an incision and drainage with primary closure was performed during this hospital stay (Fig. 5A and 5B). She returned approximately 2 weeks after her second admission to our office.

Figure 5A and 5B One day excision of the lymphangioma. (A)  The patient returned to the operating suite for operative incision and drainage of additional serous fluid with primary closure.(B)

At this point there were no signs of infection and the sutures were removed. (Figs. 6A and 6B) The surgical site appeared healed and no evidence of recurrence of the lymphangioma was apparent at this point. The patient was lost to follow up after this office visit.

Figure 6A and 6B 2 weeks following the Incision and drainage and foot appears to be healing.  (A and B)

Discussion and Conclusion

Lymphangiomas are usually noted at birth or within 2 years of life. [1] Prenatal ultrasonography can detect relatively large lesions as early as the second trimester, although lymphangiomas are frequently misdiagnosed as other pathologic entities. Lymphangiomas most commonly occur in the cervicofacial region, axilla/chest, mediastinum, retroperitoneum, buttock and perineum.

The tumors most commonly appear as ballotable masses the overlying skin is usually normal, but may have a bluish hue. Less common dermal involvement manifests as puckering or deep cutaneous dimpling. Lymphangiomas in the subcutis or submucosa manifest as tiny vesicles. Intravascular bleeding is evident by tiny, dark red domed shaped nodules. [5]

Radiologic documentation is best performed by MRI. Ultrasound is a useful auxiliary agent to confirm the presence of macrocystic lymphangiomas. Lymphangiomas, like hemangiomas and most vascular malformations, demonstrate hyperintense signal intensity in T2 weighted and turbo STIR images. Lymphangiomas demonstrate rim enhancement after contrast application. Microcystic lesions have an intermediate signal on T1 sequences and an intermediate to high signal on T2 sequences. Macrocystic lesions show low intensity in T1 and high intensity in T2. Conventional contrast lymphangiography is rarely performed. [1,4,7]

Histologically, lymphangiomas consist of ecstatic lymphatic channels, occasionally containing a mild lymphocytic infiltrate. Classification by histologic appearance includes three subtyes: capillary, cavernous and cystic. The capillary form is composed of small thinned walled lymphatic vessels. Cavernous consists of large lymphatic channels with adventitial coats.

The third and most common type, cystic lymphangioma is composed of macroscopic lymphatic spaces. These lesions are often multiseptated, which is suggestive of infiltration across the tissue planes. [1,3]

Lymphangioma rarely affects the foot and ankle region. Wu in 1996 described a case of a 12 year-old female with a lymphangioma of the anterior aspect of her ankle. He promotes the use of Lymphangiography for diagnosis and recommends for complete surgical excision for treatment. They usually appear in the cervicofacial area up to 75% of the time. [3] In 2004, Ly, et al., described another case of lymphangioma in a 2 year-old girl’s foot. They recommend Doppler sonography of the mass which reveals no remarkable blood flow. They further describe how to differentiate a lymphangioma from a hemangioma. On MRI, lymphangiomas are characterized by the absence of feeding vessels and lack of intense contrast enhancement. Because of the infiltrating nature of the lesion, complete excision rates are only 18-50%. [1] It appears that there have not been a reported percentage of lymphangioma occurrences of the foot and ankle region. In 2009, Itakura, et al., reported 114 cases of lymphangiomas, which of only 2 occurred in the foot. According to their findings the incidence of lymphangioma of the foot is 1.75%. [8]

A purely osseous lymphangioma is a very rare lesion. Bickel and Borders first described lymphangioma of bone in 1947. They described a case of a 5 year girl with an intraosseous lymphangioma of her Ilium. They speculated that as to whether this tumor could have risen in the soft tissue surrounding the bone and caused secondary erosion of the ilium. [9] In 1977, Jumbelic described the 5th reported case in bone, which was located in the medullary cavity of a long bone, the humerus of a 3 year-old girl. [10] In 1968, Rosenquist and Wolfe reported one of the first rare cases of an intraosseous lymphangioma of a lower extremity bone. They described a 3 year-old male with lymphangioma of his femur, tibia and fibula.

Although lymphangiomas are mainly a soft tissue pathology they can occur within bone, but remain extremely rare in the lower extremities. [11]

The two main complications of that can arise from lymphangiomas are intralesional bleeding and infection. Lymphangiomas often swell in the event of a viral or bacterial infection. Most often this is a harmless event likely related to change in flow or alterations. Bacterial cellulitis, however, is more dangerous and requires prolonged intravenous antibiotics. The two main strategies used to treat lymphatic anomalies are schlerotherapy and surgical resection. Schlerotherapy works through obliteration of the lymphatic lumen. Macrocystic lymphangiomas is more likely than microcystic tissue to shrink after an injection of sclerosant. Ethanol is considered to be the most effective sclerosing agent for low-flow malformations, success rates have been reported between 20% to 65%. Intralesional bleomycin has a reported success of approximately 80% with lymphangiomas of the head and neck. OK-432, a lyophilized mixture of attenuated group A Streptococcus pyogenes of human origin, has also been reported to have dramatic results reported. Resection is the only way to potentially eliminate the lymphangioma. Complete resection is the operative goal. Resection should be staged and the surgeon should focus on a defined anatomic region. Postoperative wound complications are common, including drainage, seroma and infection. A compressive dressing should be applied immediately post operatively to prevent complications. Even with an intensive approach to resection, the recurrence rate is reported to be 40% after an incomplete excision and 17% after a macroscopically complete excision. Angiogenesis inhibitors are at the forefront of medical research for the treatment of venous malformations and neoplasms. Blocking new blood vessel formation may limit the ability of the malformation or neoplasm to fully develop. Approximately 20 angiogenesis inhibitors are being tested in human trials. Research for the development of new therapeutic strategies for vascular tumors by inhibiting angiogenesis is underway. [4,5,6]

In conclusion, lymphangiomas are rare in the foot and ankle region. A thorough history and physical are necessary to help distinguish from other benign soft tissue tumors. [5] Imaging studies such as radiographs and MRI are highly recommended to aid in the diagnosis of these tumors. Surgical resection of the mass is recommended for the treatment with the goal of complete excision. The specimen should be sent to pathology for confirmation of the diagnosis. Surgical resection was recommended for this patient. Overall, the patient had minor post operative complications with no sequela. Our recommendation on the day of surgical intervention is to administer a proximal regional block in the popliteal fossa for a longer duration of pain relief post operatively and to admit the patient for observation and pain management. Infection is a common complication for this procedure and the patient should receive pre operative antibiotics and possibly continue them for one week post resection to avoid this complication. The patient was followed for approximately 6 weeks post resection of the tumor and was doing satisfactory, no pain was present upon stance or ambulation. Unfortunately the patient was lost to follow-up due to her relocation to another country.

References

1. Ly Q, Gilbert B, Davis S, Beall D, Richardson R. Lymphangioma of the foot. Am J Roent 2005 184:205-206.
2. Skandalakis J, Skandalakis L, Skandalakis P. Anatomy of the lymphatics. Surg Oncol Clin N Am. 2007 16:1-16
3. Wu K. Lymphangioma of the ankle region. J Foot Ankle Surg 1996 35(3):263-65
4. Christison-Lagay E, Fishman S. Vascular anomalies. Surg Clin N Am 2006 86:393-425.
5. Marler J, Mulliken J. Current management of hemangiomas and vascular malformations. Clin Plastic Surg 2005 32:99-116.
6. Dohil M, Baugh W, Eichenfield L. Vascular and pigmented birthmarks. Ped Clin N Am 2000 47(4):783-812.
7. Damron T, Beauchamp C, Rougraff B, Ward W. Soft-tissue lumps and bumps. JBJS 2003 85A:1142-55.
8. Itakura E, Yamamoto H, Oda Y, Furue M, Tsuneyoshi M. VEGF-C and VEGFR-3 in a series of lymphangiomas: is superficial lymphangioma a true lymphangioma? Virchows Arch 2009 454(3):317-259.
9. Bickel W, Broders A. Primary lymphangioma of the ilium: Report of a case. JBJS1947 29A:517-22.
10. Jumbelic M, Feuerstein IM, Dorfman HD. Solitary intraosseous lymphangioma: A case report. JBJS 1984 66A:1479-481
11. Rosenquist C, Wolfe D. Lymphangioma of bone. JBJS1968 50A:158-6.


Address correspondence to: Steven F. Boc, DPM, FACFAS, FACFAOM, 235 North Broad Street, Philadelphia, PA 19107.

1 Director of Podiatric Medicine and Surgery Residency Program, Hahnemann University Hospital/Associate Professor Department of Surgery Drexel College of Medicine.
2 PGY3 Podiatric Surgery and Medicine Resident, Hahnemann University Hospital/Drexel College of Medicine.
3 PGY2 Podiatric Surgery and Medicine Resident, Hahnemann University Hospital/Drexel College of Medicine.

© The Foot and Ankle Online Journal, 2011