Tag Archives: Foot

The Role of High Resolution Ultrasonography in Detection of Neglected or Missed Radiolucent Foreign Body in Foot and Ankle Region

by Reyaz Ahmad Dar (MS)1emailsm, Mubashir Maqbool Wani (MS)2emailsm, pdflrgMubashir Rashid Beig (MS)1, Muzaffer Ahmad Ganaie (MS)1

The Foot and Ankle Online Journal 6 (3): 2

A prospective case series was undertaken to assess the role of high resolution ultrasonography to detect radiolucent foreign bodies in the foot and ankle region. Out of 30 suspected foreign bodies, ultrasonography was able to detect 28 foreign bodies with 2 false negatives. The overall sensitivity was 93.33%. The false negatives were attributed to the foreign body being obscured by bone.

Key words: , foot, ankle, ultrasound,

Accepted: February, 2012
Published: March, 2013

ISSN 1941-6806
doi: 10.3827/faoj.2013.0603.002

Address correspondence to: Department of orthopaedics, SKIMS Medical college Srinagar Kashmir India – Pin 190018

1Department of orthopaedics, SKIMS Medical college Srinagar Kashmir India – Pin 190018
2Hospital for bone and joint surgery Barzulla Srinagar Kashmir India – Pin 190005

Missed or neglected foreign body and subsequent complications in the extremities is a challenging complaint in the orthopedic outpatient department. Most of these cases present with soft tissue mass, granuloma, abscess, corns, osteomyelitis, fasciitis, cellulitis, chronic discharging sinus, and tendon contracture with or without pain.[1,2,3] The initial investigation is usually done with a plain radiograph, which however, cannot detect radiolucent foreign bodies such as those of wood, plastic and rubber.

Of the other imaging modalities, xeroradiography provides better edge enhancement, but it requires special equipment and is inadequate in detecting radiolucent foreign bodies.[4,5]

Computerized tomographic (CT) scan has the ability to detect the radiolucent foreign bodies with limitations of ionizing radiation, cost and poor sensitivity in detecting small foreign bodies.[6,7] Magnetic Resonance Imaging (MRI) can detect radiolucent foreign bodies but has the limitations of being inaccessible, expensive, and a concern regarding magnetic foreign bodies as well as time consuming.


Figure 1 and Figure 2 High-resolution ultrasound of a foot suspected of having a foreign body.

There is an added disadvantage of not detecting foreign bodies with low signal intensity from tissues such as scar tissue, tendon and calcifications.[8,9] Sonography, on the other hand, is easily accessible, inexpensive and a time saving image modality.

We undertook our study on thirty patients who presented to our outpatient department at two hospitals with a definite history of foreign body injury to the foot and ankle region. Patients presented with varied signs and symptoms which included pain, soft tissue mass, abscess, corn, chronic discharging sinus with duration of symptoms ranging from four months to eight years.

Most of these patients were initially managed by primary care givers and missed or often self treated themselves removing only a part of foreign body and subsequently neglected. Our aim was to assess the role of foreign body detection in these patients with high resolution ultra sonography (USG).

Materials and Methods

Thirty symptomatic patients who had a definite history of foreign body injury of the foot and ankle region were included in this study. The symptoms of these patients varied from simple pain to chronic discharging sinus and all had a normal plain radiograph. All of them underwent high resolution ultra sonography of the affected part followed by surgical exploration.

Sonography was conducted by four specialist doctors who had a minimum of four years of experience in the radiology department. Sensitivity of USG was determined with respect to that found on surgical exploration.


Thirty consecutive patients presented to our outpatient departments from May 2008 to May 2012 with history of foreign body injury. Patients presented with persistent pain, soft tissue mass, granuloma, abscess or chronic discharging sinus with a normal radiograph. Nineteen patients were male. Twenty two patients were younger than twenty years of age. Twenty eight patients had symptoms in the foot; two had symptoms in the ankle region. Twenty three patients had a history of nail insertion in the foot through a rubber sole. There was thorn injury in six patients with five having it in the foot and one in the ankle region. One patient had injury to the ankle with a wood. Three patients had multiple surgical interventions for chronic discharging sinuses.

All these patients were sent to radiology for the high resolution ultra sonography of the affected part. In all our cases a frequency of 7.5 MHz to 13 MHz was employed. Foreign bodies were reported as hyperechoic masses with surrounding hypo echoic rim with an acoustic shadow in twenty eight patients (Fig. 1 and Fig. 2).


Figure 3 Foreign body seen at the time of surgery.

Two patients which were reported negative had chronic discharging sinus with one having it on the lateral malleolus and another on the dorsal aspect of the foot. All patients underwent surgical exploration under general or regional anaesthesia with tourniquet control. Preoperative methylene blue injection into the sinus was used in three patients with chronic discharging sinus. Foreign bodies were recovered from all the patients (Fig. 3 and Fig. 4). Two patients who were labeled by the sonologist of not having a foreign body had foreign bodies close to or obscured by the bone. One of the patients had injury to the right lateral malleolar area with a wooden foreign body with persistent sinus discharge, and on exploration the foreign body was found very close to and abutting the cortex. Another patient had a history of nail insertion through the sole of the shoe with persistent sinus discharge on the planter aspect of the foot, and on surgical exploration a piece of rubber was found abutting the second metatarsal shaft cortex on the dorsal aspect. Out of the total thirty suspected radiolucent foreign bodies, high resolution ultra sonography was able to detect the foreign body in 28 patients with two false negatives with an overall sensitivity of 93.33%.


Figure 4 Foreign body after removal.


The basic principle of ultra sound is the use of a transducer to penetrate tissues with ultrasonic waves at various frequencies. When the wave strikes the denser component of tissue, they bounce (echo) back to the transducer. The ultrasound can then interpret the speed and intensity of the sound wave to determine the location and composition of the object. Structures are plotted on the screen based on their depth and location relative to the transducer. Superficial structures are plotted at the top and deeper ones at the bottom of the screen. The larger the surface area toward the transducer the greater it will reflect. Sonographic features of the foreign bodies in the soft tissues have three components. Firstly, the appearance of the foreign body; secondly, the changes in the soft tissues surrounding the foreign bodies. Thirdly, the appearance of soft tissues distal to the foreign bodies.

All foreign bodies on ultrasonography appear as hyperechoic foci. The reflectivity depends on acoustic impedance of the foreign body which in turn varies with the density of the object. In general, metal, mineral, glass, wood, and rubber reflect sound, appearing white on the screen. The changes surrounding the foreign bodies are due to inflammatory reaction which may range from edema to abscess formation.

This reaction takes some time to develop and is shown as hypo echoic rim around the foreign body. Distal to the echo rich foreign body acoustic shadowing is noted. This is due to failure of the ultrasound to pass through the foreign body.[10,11]

Despite their size, foreign bodies are no small matter. When left untreated they cause pain, swelling, infection, nerve and tendon injury.[2,3,12] Although USG has been a well-established diagnostic tool for foreign bodies in the soft tissues, it has been underutilized in this part of the world. While evaluating the usefulness of USG in the detection of unsuspected foreign bodies followed by CT, MRI, bone and labeled red cell Scintigraphy, it has been found that the later investigations added no relevant information and were time consuming and costly.[12] The sensitivity of USG in detecting different foreign bodies has been reported to be 70% to 100%. Cases which turned out to be false negatives had either a very deep foreign body, gas around foreign body, or a foreign body too close to the bone [8,13,14,15] as was the case in two of our patients.

Several studies have demonstrated the effectiveness of USG in detecting non-opaque foreign bodies in the soft tissues. The power of USG is as important as the depth of penetration of wave into soft tissues. The shorter wave length with high frequency penetrates less as most of energy is absorbed by the medium.[15] The authors do not believe that the results could be different if the USG was done by the same radiologists. Differences in the comparative accuracy, sensitivity and specificity of foreign body detection by radiologist and USG technician has not been found to be statistically significant in the previous studies.[16]


The authors do not recommend replacing plain radiography with ultrasonography in the evaluation of suspected foreign bodies of the foot and ankle region. But Sonography should definitely be considered part of diagnostic work up of patients in whom we strongly suspect the presence of radiolucent foreign bodies based on history and symptomatology.


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Left Underlapping Third Toe in a Patient who Underwent Ventricular Assist Device Implantation: A Case Report and Literature Review

by Massimiliano Polastri, MSc, PT, Walter Trani, PT1, Mariano Cefarelli, MD2, Sofia Martìn-Suàrez, MD2

The Foot and Ankle Online Journal 5 (12): 2

This case report describes a rare abnormality of the forefoot in an adult who underwent implantation of a ventricular assist device. Toe deformities are not necessarily related to pain and/or functional foot limitations. An underlapping toe is a rarely, described disorder. Ventricular assist devices (VAD) are comprised of a set of tools that allows the system to substitute for the pump function of the heart in eligible patients. A 60-year-old Caucasian man affected by ischemic dilated cardiomyopathy underwent ventricular assist device implantation as a bridge to transplantation. The third toe abnormality reported here did not influence the ventricular assist device implantation or postoperative recovery in terms of exercising. An underlapping third toe can coexist in the presence of debilitating illness without causing particular physical difficulties.

Key words: Blood circulation, Forefoot, Gait, Heart transplantation, Quality of life, Rehabilitation, Toes abnormalities.

Accepted: November, 2012
Published: December, 2012

ISSN 1941-6806
doi: 10.3827/faoj.2012.0512.0002

Toe deformities are not necessarily related to pain and/or functional foot limitations. [1] Rare abnormalities such as overlapping toes are a condition for which there is no possibility for spontaneous improvement. [2]

In contrast, an underlapping toe is a rare and little-described disorder. Friend found that the fourth and fifth toes are the most involved in an underlapping toe abnormality even if the second or third toes are also affected. The combination of congenitally elongated toes and an acquired adductovarus is the major mechanism that produces this deformity. [3] Ventricular assist devices (VAD) are comprised of a set of tools that allows the system to substitute for the pump function of the heart in eligible patients.

The main body of the device includes a miniaturized titanium pump. The power cord of the device used in the case described here was connected to a titanium base fixed to the skull (parietotemporal). [4]

The system is powered by lithium and lead batteries—which have different durations—and is transported in a bag in a horizontal position so as not to cover the microphone alarm. Left ventricular assist devices (LVAD) are an effective strategy to prolong survival and improve quality of life. [5] The Interagency Registry for Mechanically Assisted Circulatory Support has been created to collect information about patients, devices, and outcomes, including adverse events.  [6] The main purpose of this report is to describe a rare abnormality of the forefoot in an adult who underwent implantation of a VAD.

Case Report

A 60 year-old Caucasian man affected by ischemic dilated cardiomyopathy underwent LVAD implantation (Jarvik Heart®, New York, NY, USA) as a bridge to transplantation. He had diabetes, dyslipidemia and was an ex-smoker.

He did not undergo myocardial revascularization after two episodes of acute myocardial infarction, and 8 years ago he was implanted with a single-chamber implantable cardioverter. The patient underwent pre-transplant screening for nearly 2 years. It was decided to apply a temporary LVAD due to his low cardiac ventricular function (ejection fraction, 22%) and significant pulmonary hypertension. This device has a compact axial flow impeller pump with an outflow Dacron graft for anastomosis to the descending thoracic aorta. The pump was inserted through a sewing cuff into the apex of the left ventricle. The adult model measured 2.5 cm in diameter by 5.5 cm in length. Its weight was 85 g with a displacement volume of 25 mL. The postoperative course was free of complications. Bilateral hallux valgus and an underlapping third toe on the left side were noted by observation of the patient in a standing position. (Fig.1) Deviation in the valgus of the right big toe was more evident, as was pronation of the first metatarsophalangeal joint (this condition probably avoided the hammer toe on the same side). The left foot was characterized by hammer toes (Fig. 2), and the congenital underlapping third toe was attached to the first toe through the distal portion of both toes. (Fig. 3)


Figure 1 Standing position. Right side: hallux valgus, hammertoes second to fifth. Left side: hammertoes first to fifth, hallux valgus, underlapping third toe.


Figure 2 Dorsal view of the left side: underlapping third toe.


Figure 3 Plantar view of the left side: the third toe is medially deviated (two red arrows) and attached to the first (four red arrows).

The patient had no difficulties ambulating and was free from pain. Thus, postoperative rehabilitation was centered on recovery of motor activity and reconditioning after the VAD implantation. The first line of the rehabilitative treatment in the sub-intensive setting was focused on encouraging the patient to perform exercises (even in a group) such as cycling, climbing stairs, and walking (even outside the pavilion); the patient’s performance of exercises was monitored. Furthermore, all motor activities were performed in association with respiratory exercises, such as deep breathing and incentive spirometry. The patient provided written informed consent for this study.


The absence of both foot pain and functional limitations at the initial examination was unexpected, but allowed the patient to adhere to the postoperative rehabilitation program, with excellent results. Augustine and Jacobs described hammertoes as the most common deformities of the foot. [7] Abnormalities of the forefoot, particularly in children are described in the literature. Smith, et al., found that an underlapping toe was common in a pediatric population of 44 newborns and proposed a simple algorithm for treatment. [8] In the mid-1960s, Greenberg discussed the possibility of resolving underlapping and contracted toes by plantar digital tenotomy, in the absence of shortening of the dorsal tissue and subluxation of the metatarsophalangeal. [9]

Similarly, Korn proposed a surgical approach for correction of a painful underlapping fifth toe and reported excellent outcomes of surgery. [10] Fattirolli, et al., discussed the importance of a customized rehabilitation program in patients undergoing VAD to enhance function and the quality of life. [4] A multidisciplinary approach is the ideal solution for long-term care during postoperative recovery. [11] Furthermore, the benefits of exercise training were reported by Bellotto, et al., who discussed the postoperative course of a patient with an implanted artificial heart. [12] Polastri investigated the role of postoperative rehabilitation after hallux valgus surgery, and surmised that a rehabilitative intervention is required to encourage both plantar pressure on the first ray and joint mobility. [13] If these are the objectives of hallux valgus surgery, what is advisable in terms of exercise in a case such as that we report here in which the deformities were not corrected? The answer to this question must consider the rationale of the treatment according to both the condition of the patients and their quality-of-life expectations. In fact, the patient described here was admitted so that his cardiac function issues could be addressed; the feet abnormalities (hallux valgus, hammer toes, and underlapping third toe) were an occasional finding of secondary importance considering his overall condition. The postoperative rehabilitation pathway, particularly in specialized settings, must be appropriate and centered on the patient’s needs with due consideration of their priorities. In this regard, the third toe abnormality reported here did not influence the VAD implantation or postoperative recovery in terms of exercising. The main limitation of this case report is the lack of quantification of the foot-joint deformities by means of range-of-motion measurements. However, the aim of this case study was to describe an unusual abnormality that does not require deep investigation. Furthermore, our findings should not be extended to a larger population. Nevertheless, this is to our knowledge the first report of feet deformities in a patient implanted with a VAD. In summary, an underlapping third toe can coexist in the presence of debilitating illness without causing particular physical difficulties.


1. Badlissi F, Dunn JE, Link CL, Keysor JJ, McKinlay JB, Felson DT. Foot musculoskeletal disorders, pain and foot-related functional limitation in older person. J Am Geriatr Soc 2005 53: 1029-1033. [PubMed]
2. Hulman S. Simple operation for the overlapping fifth toe. Br Med J 1954 2: 1506-1507. [PubMed]
3. Friend G. Correction of elongated underlapping lesser toes by middle phalangectomy and skin plasty. J Foot Surg 1984 23: 470-476. [PubMed]
4. Fattirolli F, Bonacchi M, Burgisser C, Cellai T, Francini S, Valente S, Sani G, F. Cardiac rehabilitation of patients with left ventricular assist device as “destination therapy”. Monaldi Arch Chest Dis 2009 72: 190-199. [PubMed]
5. Maciver J, Ross HJ. Quality of life and left ventricular assist device support. Circulation 2012 126: 866-874. [PubMed]
6. Rector TS, Taylor BC, Greer N, Rutks I, Wilt TJ. Use of left ventricular assist device as destination therapy in end-stage congestive heart failure: a systematic review. 2012, Washington (DC), Department of Veterans Affairs. URL: http://www.ncbi.nlm.nih.gov/books/NBK99059/pdf/TOC.pdf. [PDF] (accessed 18 August 2012). [Website]
7. Augustine DF, Jacobs JF.V Restoration of toe function with minimal traumatic procedures including advanced diaphysectomy. Clin Podiatry 1985 2: 457-470. [PubMed]
8. Smith WG, Seki J, Smith RW. Prospective study of a noninvasive treatment for two common congenital toe abnormalities (curly/varus/under lapping toes and overlapping toes). Pediatr Child Health 2007 12: 755-759. [PubMed]
9. Greenberg HH. Plantar digital tenotomy for underlapping and contracted toes. J Am Podiatry Assoc 1966 56: 65-66. [PubMed]
10. Korn SH. The lazy S approach for correction of painful underlapping fifth digit. J Am Podiatry Assoc 1980 70: 30-33. [PubMed]
11. Pistono M, Corrà U, Gnemmi M, Imparato A, Caruso R, Balestroni G, Tarro Genta F, Angelino E, Giannuzzi P. Cardiovascular prevention and rehabilitation for patients with ventricular assist device from exercise therapy to long-term therapy. Part II: long-term therapy. Monaldi Arch Chest Dis 2011 76: 136-145. [PubMed]
12. Bellotto F, Compostella L, Agostoni P, Torregrossa G, Setzu T, Gambino A, Russo N, Feltrin G, Tarzia V, Gerosa G. Peripheral adaptation mechanisms in physical training and cardiac rehabilitation: the case of a patient supported by a CardioWest total artificial heart. J Card Fall 2011 17(8): 670-675. [PubMed]
13. Polastri M. Postoperative rehabilitation after hallux valgus surgery: a literature review. FAOJ 2011 4(6): 4. [Website]

Address correspondence to: Massimiliano Polastri, Physical Medicine and Rehabilitation, Bologna, University Hospital Authority, Sant’Orsola-Malpighi Polyclinic, Via G. Massarenti, 9. 40138 –Bologna, Italy.

1  Physical Medicine and Rehabilitation, Bologna University Hospital Authority, Sant’ Orsola-Malpighi Polyclinic, Bologna, Italy.
2  Cardiac Surgery Department, Sant’ Orsola-Malpighi Polyclinic, Bologna University, Bologna, Italy.

© The Foot and Ankle Online Journal, 2012

Arteriovenous Malformation: An Unusual Reason for Foot Pain in Children

by Kunze, B.1 , Kluba, T.1, Ernemann, U.2, Miller, S.3

The Foot and Ankle Online Journal 2 (12): 1

The incidence of vascular anomalies is rare, and they are mainly localized in the head or upper extremity. We report the case of an 8 year-old boy with arteriovenous high-flow malformation of the foot. Presentations of diagnostic and therapeutic opportunities as well as post surgical clinical follow-up are included.

Key words: Arteriovenous malformation, angiography, foot, children.

Accepted: November, 2009
Published: December, 2009

ISSN 1941-6806
doi: 10.3827/faoj.2009.0212.0001

Vascular anomalies are classified into two main types: vascular malformations and benign vascular endothelium tumors. Vascular malformations (e.g. venous, arteriovenous and capillary malformations) are congenital abnormalities with manifestation during childhood or adolescence due to various stimuli (e.g. trauma, hormonal changes). They arise from defects in vascular tissue during embryonic development. Spontaneous regression of these anomalies is rare. Vascular tumours (e.g. haemangioma, Gorham-Stout-disease) are characterized by cellular hyperplasia and the expression of growth factors but they can also spontaneously become involuted. [1-6] “High” and “low-flow” malformations can be differentiated by their haemodynamic characteristics and the vascular architecture. [7,8]

Case Report

We report the case of an 8 year-old boy who presented in 2008 with a one-year history of progressive left foot pain and local swelling without previous trauma. Weight-bearing activities exacerbated the pain.

On clinical examination, a plantar soft tissue swelling with local paraesthesia and hypersensitivity was seen. The left foot was held in a relieving pes equinus posture.

Magnetic resonance imaging (MRI) of the foot was performed. The swelling showed up as a vascular malformation with high uptake of contrast medium which stretched out intrafascially, intramuscularly and subcutaneously from the metacarpophalangeal joints to the tarsal bone. (Figs. 1 and 2)

Figure 1 Sagittal MRI of the left foot with hypervascularized tumor on T2 (arrow).

Figure 2 MR image of the left foot with hypervascularized tumor on T1.

An open biopsy was performed and histological examination confirmed the radiological diagnosis. To minimize the trauma of treatment for the boy, selective percutaneous as well as endovascular transarterial embolisation of the malformation were discussed. After percutaneous puncture and illustration of the malformation by injection of contrast medium, a venous lacuna with a varicose vein of plantar venous arc was seen. (Fig. 3)

Figure 3 Percutaneous puncture and illustration of the malformation by injection of contrast medium.

Additionally in projection of the third toe a high-flow arteriovenous malformation was depicted supplied in an en-passant fashion by an interdigital artery and arteries to the back of the foot. (Fig. 4) Due to these arterial collaterals a percutaneous embolisation was not performed because of the risk of necrosis of the toe.

Figure 4 Percutaneous puncture and illustration of the malformation by injection of contrast medium.

During the course of further treatment planning MR angiography was performed and revealed a hypertrophy of the posterior tibial artery and of the lateral part of the plantar artery. The distal part of Ramus profundus of A. plantaris medialis was the focus of the malformation after its passage through the abductor hallucis.

Expected circulatory disturbance of the third toe and the probability of an incomplete occlusion of the malformation using transarterial embolisation limited the therapeutic options.

Finally, marginal surgical resection of the malformation was performed. Histological examination confirmed the diagnosis of arteriovenous malformation with a central thrombus.

Only eight weeks after surgery, the boy could bear his full weight on his foot and the local pain as well the swelling were significantly reduced. (Fig. 5) At the one year follow-up the boy had returned to sporting activity. No signs of local recurrence were evident.

Figure 5  Clinical control six months after surgery.


Vascular anomalies occur with an incidence of 1-10/100000. They are apparent in 1 – 2.6% of neonates. [9] Cephalic localizations are most common, followed by the trunk, lower and upper extremities. In rare cases vascular malformations may be part of a syndrome complex like Klippel-Trenaunay-Weber and Sturge-Weber syndromes or genetic disorders, so when evaluating these abnormalities a family history should be performed. [10,11,12]

The presented case describes a high-flow arteriovenous malformation of the foot. This localization appears to be rare with only few reports in literature.

Whereas a third of vascular tumors (e.g. haemangioma) are visible at birth, the remaining 70% appears in the first years of childhood. They are characterized by a rapid growth in the first years but can also spontaneously become involuted.

In contrast, congenital malformations increase in size as the child grows. [13] According to their pathophysiology and vascular architecture low flow and high flow malformations are distinguished. Low flow lesions comprise capillary, lymphatic and venous malformations. High flow arteriovenous malformations are characterized by a high blood flow in a direct connection between arteries and veins without capillaries. The Schobinger staging system of arteriovenous malformations includes four grades of severity: dormancy, expansion, destruction and decompensation. [14] Clinical manifestation may occur during adolescence and due to various stimuli (e.g. trauma, hormonal changes).

In the presented case, the cause of expansion and clinical manifestation could not be determined.

Depending on the localization, arteriovenous malformations present with different symptoms: local pain, swelling, skin ulceration, length discrepancy of extremities or severe bleeding with neurological complications. [13,15,16] After clinical examination further diagnostic investigations are recommended. Useful non-invasive methods to define the extent of the lesion are ultrasonography and duplexsonography. Additional non-invasive imaging modalities are magnetic resonance imaging and MR angiography including dynamic sequences with high temporal resolution. Transarterial catheter angiography is required in order to visualize the architecture of the vascular malformation and the arterial supply and venous drainage of the surrounding structures prior to treatment. [7,17,18,19,20] Depending to the extension of the malformation and the associated involvement of surrounding tissue, minimally invasive interventions or surgical resection may be necessary.

This report describes an unusual localization of an arteriovenous malformation. Nevertheless, the possibility of its existence should be considered in cases with the symptoms presented here. To facilitate rapidly diagnosis and to avoid unnecessary treatment, interdisciplinary therapy is recommended.


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Address correspondence to: Dr. Torsten Kluba
Department of Orthopaedics, University of Tuebingen, Hoppe-Seyler-Str. 3, 72076 Tübingen, Germany. Email: Torsten.Kluba@med.uni-tuebingen.de
Tel.+ 49 7071 2986685,Fax + 49 7071 294091.

Department of Orthopaedics, University of Tuebingen Hoppe-Seyler-Str. 3, 72076 Tübingen, Germany.
Department of Neuroradiology, University of Tuebingen Hoppe-Seyler-Str. 3, 72076 Tübingen, Germany.
Department of Radiology, University of Tuebingen Hoppe-Seyler-Str. 3, 72076 Tübingen, Germany.

© The Foot and Ankle Online Journal, 2009

Congenital Variations Discovered in the Clinical Presentation of Hyperkeratosis of the Hand and Foot: A report of 2 cases

by Al Kline, DPM1

The Foot & Ankle Journal 2 (1): 3

Case presentations describing a congenital variation of palmoplantar keratosis are presented. The majority of these conditions are autosomal dominant with associated nail dystrophy. A variant condition is described with little palmar keratosis; however, finger nail and toe nail dystrophy is the most common identifying feature. Gene identification and treatment protocol are presented. Fortunately, these conditions are rare. A good knowledge of these conditions will help in proper diagnosis and treatment.

Key words: Palmoplantar keratosis (PPK), hand, foot, congenital, hyperkeratosis

This is an Open Access article distributed under the terms of the Creative Commons Attribution License.  It permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. ©The Foot & Ankle Journal (www.faoj.org)

Accepted: December, 2008
Published: January, 2009

ISSN 1941-6806
doi: 10.3827/faoj.2009.0201.0003

Congenital hyperkeratosis is an uncommon condition of the foot. There are a number of congenital conditions that cause hyperkeratotic lesions of the foot. Fortunately, these conditions remain rare in the population. These lesions can be divided into diffuse and punctuate. There is also a subclass of hereditary diseases defined as either epidermolytic or nonepidermolytic. Conditions characterized by palmoplantar keratosis (PPK) are the most causes of congenital hyperkeratosis. These conditions include Unna-Thost disease, Vohwinkel’s syndrome, Papillon-Lefèvre syndrome and pachyonychia congenita. [1,2,3,4]

Unna-Thost disease was first described in 1880 by Herrmann Arthur Thost and again described in 1883 by Paul Gerson Unna. [5,6,7] Synonyms for the disease include Brünauer-Fuhs-Siemens syndrome, Brauer’s syndrome and Brünauer’s syndrome. [7] It is a disease of autosomal dominant origin characterized by severe palmoplantar keratosis. Deep fissures and hypohidrosis with thickened skin of the palms of the hands and soles of the feet usually occur within the first year after birth. [1] Vohwinkel’s syndrome or keratoderma hereditaria mutilans is a rare autosomal dominant condition first described in 1929. [2] Clinical manifestations first appear in infants and then proceed through childhood into adulthood. Keratosis described as honeycomb and starfish-like in appearance is common. In the later stages of the disease, pseudoainhum or auto amputation of the digits can occur due to constricting bands of keratosis around the digit.

In fact, pseudoainhum is characteristic in a number of hereditary hyperkeratosis. [1,2,3,4] Papillon-Lefèvre syndrome (PLS), also known as Mal de Meleda , was first described by two French physicians in 1924. [3,8,9] It is an extremely rare genodermatosis inherited as an autosomal recessive trait, affecting children between the ages of 1-4. [3] Psoriatic-like plaques involving the palms, soles and elbows are described that worsen in winter and are often hyperhidrotic resulting in a foul odor. [3] Pachyonychia congenita (PC) is a rare genodermatosis that affects the nails of all the toes and fingers. [4] Other names and synonyms of this condition are called congenital dyskeratosis and pachyonychia ichthyosiformis.

Most cases appear within the first or second years of life, although cases of late onset have been reported in the second and third decades. (which is termed PC tarda) [4] Diagnostic features include symmetrical thickening of skin, dysmorphic nails and hyperkeratotic skin lesions. The disease fits into two major types: Jadassohn-Lewandowsky syndrome (JLS-1) and Jackson-Lawler syndrome (JLS-2). It is an autosomal dominant trait. Some recessive forms have been described. JLS-1 is characterized by nail hypertrophy, nail dystrophy, PPK, follicular keratosis and oral leukokeratosis and is the most common form of PC (56.2%). [4] JLS-2 usually lacks oral leukokeratosis and is commonly associated with epidermolytic bullae of the palms and soles (24.9%). [4]

Secondary symptoms are commonly associated with hereditary hyperkeratosis. This is called complex keratodermas. Unna-Thost disease is commonly associated with secondary fungal and bacterial infections. [1] Corneal opacities, pilitorti, hearing loss, hypohidrosis and dental abnormalities have also been described. [7]

Vohwinkel’s syndrome can be associated with deafness, cancer, cardiomyopathy and adrenal insufficiency. [2] PLS is often associated with severe periodontitis which usually starts at the age of three or four. [3] This is seen with gingivitis and rapid destruction of the periodontium when deciduous teeth proceed normally. Other associated conditions of PLS include pyogenic liver abscesses with impaired immunodeficiency. [3] The most common secondary associated symptom with PC is oral leukokeratosis with associated periodontitis and loss of teeth. [4] The teeth develop normally and are lost within 1 year.

Gene Identification

Often, the clinical presentation is not as straight forward as the texts present. Clinical evaluation can present with diffuse as well as punctuate keratodermas and associated nail dystrophy. Clinical diagnosis is usually made by presentation and secondary associated conditions in complex keratodermas.

Recent molecular biological studies indicate the presence of two variants of Vohwinkel’s syndrome, an ichthyosis-associated variant, associated with an insertional mutation of the loricrin gene, and a deafness-associated variant, associated with a missense mutation of the connexin-26 gene. [2] In PLS, there is a reported loss-of-function mutation affecting both alleles of the cathepsin-C gene identified on chromosome 11q14.1-q14.3.3 In Pachyonychia congenita (PC) mutations in the KRT16 and KRT 17 gene encoding keratins K6a and K16 (KRT16) that can trigger JLS-1 and JLS-2 respectively have recently been identified. [4]

Case Studies

Case 1: A 9 year-old female presents to our office with diffuse and punctate hyperkeratosis of both feet. (Fig. 1) Clinically, the hyperkeratosis is located on the soles of both feet with associated severe nail dystrophy to all fingers and toes.


Figure 1  A 9 year old-female with severe plantar regions of hyperkeratosis (A and B).  The toe nails are severely dystrophic (C).  This condition began in infancy and is congenital.

Interestingly, there is little palmar hyperkeratosis. (Fig. 2) The patient’s father, grandmother and aunt are affected with the same condition. All have PPK with associated fingernail and toenail dystrophy and discoloration. The patient presented with the condition at birth. The patient’s grandfather and uncle are asymptomatic. The condition is characterized by extreme pain.


Figure 2  Although there is severe plantar congenital hyperkeratosis, the hands show very little palmar keratosis (A and B).

The patient’s father has been treated with narcotics for a number of years. Most of the adults in the family abuse tobacco. The father, grandmother and aunt have undergone multiple surgical debridements in attempts to reduce keratosis. This included surgical debridement of deep keratomas, removal of nail plates and beds and metatarsal head compression osteotomies and 5th metatarsal head resections. To date, surgery was only temporarily effective.

Retinoids were not used at the time. The family did not exhibit any complex symptoms and dentition was normal. No oral leukokeratosis was seen on examination. The entire family has diffuse hyperkeratosis and hyperkeratosis of all nails affecting both hands and feet symmetrically.

The daughter is now treated routinely treated with Retinoid creams, keratolytic agents, debridement and accommodative padding on a regular basis. It appears that most family members affected had the condition worse on the soles of the feet than the hands. Although there is very little palmar keratosis in the 9 year-old female, both the nails of the hands and feet are affected. The patient’s hands reveal discolored, dystrophic changes to the finger nails, but very little palmer keratosis.

Case 2: A 37 year-old female presents with severe plantar keratoderma and secondary inflammation and interdigital bacterial infection. She has allergies to iodine and seafood. The patient has diffuse plantar foot keratosis with nail involvement. (Fig. 3)


Figure 3  A 39 year-old female with diffuse keratosis to both feet (A).  Secondary infectious tinea is also observed along the medial border of the foot (B).  In this variant form, the hyperkeratosis is more diffuse rather than punctate along the soles of the feet. 

However, her hands appeared to be almost spared of the condition. There is some distal darkening just under the distal region of the finger nail. (Fig. 4).


Figure 4  As in case 1, this 39 year-old female has very little palmar keratosis.  Distal nail discoloration is observed (A and B).

Her family history shows that most family members were affected by the disease. Her grandmother had the condition, but not the grandfather. They had eight children, 4 boys and 4 girls.

All of the boys inherited the disease and only one girl. Three girls were unaffected. Her mother (who is unaffected) and father (who is one of the boys affected) had 2 boys and 4 girls. Of this group, her 2 oldest sisters have the condition and one brother is also affected.

Only one male was unaffected. Some of the siblings have the condition much worse than the others.


Treatment regimes are based on surgical techniques, medications and ancillary treatments designed to decrease painful keratosis. Surgical debridement or paring of keratosis is effective, but only temporary. Surgical bone debridement under regions of intense hyperkeratosis rarely works in our experience. There is very little information in the literature concerning full thickness surgical removal of tissue and skin grafting. Unfortunately, hyperkeratotic areas may return as soon as 1-2 weeks following simple debridement. Medications designed to improve PPK include keratolytic agents and oral or topical retinoids. The most common keratolytic agent used today is Vanamide®. Vanamide is a keratolytic, emollient cream designed to soften the skin. This can be used topically under occlusion for the best results. We have used Vanamide under occlusion for 3 or 4 days before the office visit. It helps most in the debridement of keratosis by softening or hydrating the region of hard keratosis. Vanamide’s main ingredient is 40% urea, so it is especially useful in nail as well as skin debridement. [10] Probably, the most widely used oral retinoid is Accutane or Isotretinoin. Retinoids are a class of medications derived from vitamin A and used to treat various skin conditions from psoriasis to warts to skin cancers. Oral retinoids were first released for use in the United States and Europe in 1982. [11] Oral accutane is most commonly used for cystic acne. Etretinate and Acitretin are more commonly used in the treatment of hyperkeratosis. [11] Oral retinoids should be carefully used in females. The drugs are teratogenic causing serious birth defects.[11] However, when carefully used, they have been found to be very effective in the treatment of various PPK disorders. [2] There have also been reports of elevated liver enzymes while taking oral retinoids, so careful monitoring of the liver enzyme panel is recommended. [11]

Topical and ancillary treatments can include saline soaks, topical Vaseline under occlusion, adding bleach to bath water, antibacterial soaps and a host of others too numerous to mention.


Hereditary hyperkeratotic disorders appear to be heterogenous in nature. In the case studies presented, the autosomal dominant gene has no predilection for males or females and is randomized, passing the trait to some siblings while sparing others. Autosomal dominant carriers have a 50:50 chance of passing this gene on to their siblings and the individuals spared will not have the ability to pass on this disease and will not be carriers. [12] It also appears that genetic polymorphisms and mutations continue to occur in varied cases. This would explain why some individuals have the condition worse and some have milder forms of the disease. It can be safe to say that the majority of PPK disorders can have variant forms and severity. As varied as this disease can present, treatment results can also vary. We have found that aggressive debridement with use of topical keratolytics with oral retinoids provide some of the best results. Surgical procedures should only address regions that are most problematic and don’t respond readily to conservative treatment regimes. Educational instruction and understanding should include a thorough discussion with your patients that results can vary and may be unsuccessful or only temporary. Discussing the disease and treatment options will enable better care of the patient with this frustrating condition.


1. Kline A. Keratotic lesions of the footH. Podiatry Internet Journal 1 (1): 8, 2006.
2. Bari AU. Keratoderma hereditarium mutilans (Vohwinkel syndrome) in three siblings. Dermatology Online Journal. 12 (7): 10, 2006.
3. Janjua SA, Khachemoune A. Papillon-Lefèvre syndreom: Case report and review of the literature. Dermatology Online Journal. 10 (1): 13, 2004.
4. Caproni M, Fabbri P. Pachyonychia congenita Orphanet Encylopedia., (online PDF) accessed 21/12/2008.
5. Thost A. Über erbliche Ichtyosis palmaris et plantaris cornea. Dissertation. Heidelberg, 1880.
6. Unna PG. Über das Keratoma palmare et plantare hereditarium. Vierteljahrsschrift für Dermatologie und Syphilis. Wien. 15: 231, 1883.
7. Unna Thost Syndrome. Who named it? (online), accessed 21/12/2008.
8. Papillon MM, Lefèvre P. Deux cas de kératodermie palmaire et plantaire symétrique familiale (maladie de Meleda) chez le frère et la soeur. Coexistence dans les deux cas d’altérations dentaires graves.
Bulletin de la Société française de dermatologie et de vénéorologie. 4 (31): 82-87, 1924.
9. Papillon MM. Lefèvre syndrome. Who named it? (online), accessd 22/12/2008.
10. Vanamide Cream (online PDF) , accessed 21st December 2008.
11. Chan A, Hanna M, Abbott M, Keane R. Oral retinoids and pregnancy. The Medical Journal of Australia. 165: 164-167, 1996.
12. The Universe of Genetic Testing, online resource. [online] , accessed date 21/12/2008.

Address correspondence to: Al Kline, DPM
3130 South Alameda, Corpus Christi, Texas 78404.

1 Adjunct Clinical Faculty, Barry University School of Podiatric Medicine. Private practice, Chief of Podiatry, Doctors Regional Medical Center. Corpus Christi, Texas, 78411.

© The Foot & Ankle Journal, 2009

Chondromyxoid Fibroma of the Foot: An Uncommon Presentation

by Anil Thomas Oommen, MS (Ortho), DNB (Ortho)1 , Vrisha Madhuri, MS (Ortho), MCh (Ortho)2 , Noel Malcolm Walter, MD3

The Foot & Ankle Journal 2 (1): 2

We report a chondromyxoid fibroma of the proximal phalanx of the left third toe in a 35 year old woman, a rare site for this tumor. The radiological appearance of a trabeculated lytic lesion with sclerotic margins and soft tissue extension raised the possibility of other entities such as intraosseous epidermoid cyst, aneurysmal bone cyst, chondroblastoma, osteoid osteoma and chondrosarcoma as well as infections like pyogenic, tuberculous and leprous dactylitis. However the typical microscopic picture of lobules of cartilage separated by fibrocellular tissue and scattered osteoclasts confirmed the diagnosis. The tumor was successfully treated by curettage and bone grafting using a dorsal and plantar 2 incision technique and has no recurrence at 4 year follow up. In this article we present our experience with this case and discuss the differential diagnosis of a solitary lytic lesion in the phalanges of the toes.

Key words: Foot, chondromyxoid fibroma, cartilaginous tumor, lytic lesion toe, curettage

This is an Open Access article distributed under the terms of the Creative Commons Attribution License.  It permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. ©The Foot & Ankle Journal (www.faoj.org)

Accepted: December, 2008
Published: January, 2009

ISSN 1941-6806
doi: 10.3827/faoj.2009.0201.0002

A 35 year old housewife presented with a swelling on the dorsal aspect of the right third toe which had been present for 10 years with gradual increase in size. On examination an ovoid swelling measuring 1.5 x 1.5cm and extending from the metatarso-phalangeal joint up to the middle of the toe was noted. There was fullness on the plantar aspect of the third toe which extended to the web space on either side with the two adjacent toes being pushed aside as a result. Radiological examination revealed a trabeculated lytic lesion with sclerotic margins involving half the length of the proximal phalanx of the third toe. ( Fig. 1 )


Figure 1  Eccentric expansile well defined lytic lesion showing intra lesional trabeculae and  thinned medial cortex of the proximal phalanx of the third toe. Soft tissue swelling around the base of 3rd toe and widening of the webspaces is noted.

A needle biopsy was reported as benign cartilaginous tumor. Through a combined dorsomedial and plantar approach the soft tissue component was excised. The tumor was thoroughly curetted after making a window over the dorsomedial aspect of the proximal phalanx. The plantar approach was added to ensure complete clearance of soft tissue extension which was predominantly on the plantar aspect. The cavity was then filled with cancellous bone graft harvested from the ipsilateral proximal tibia.

Microscopic evaluation of the curetted tissue revealed a tumor composed of lobules of cartilage separated by fibrocellular tissue. (Fig. 2) Osteoclast-like cells were scattered along the interface between these tissues.(Fig. 3) There was no histological evidence of malignancy. At 4 years follow up there is no recurrence. (Figs. 4,5 )


Figure 2 Lobules of cartilage separated by fibrocellular tissue. (HPE H&E x 100)


Figure 3  Osteoclast like cells along the edge of chndromyxoid areas. (HPE H&E x 200)


Figure 4  Radiograph 4 years post op AP shows sclerosis and healed lesion.


Figure 5  Oblique radiograph showing healed lesion 4 years post op.


Chondromyxoid fibroma (CMF) is a rare neoplasm constituting less than 1% of all bone tumors. [1,2] It is derived from skeletal connective tissue cells which demonstrate the capacity to produce chondro-myxoid matrix in a distinctive histological pattern. The peak incidence is in the second and third decades of life. The most common bones affected are those of the lower extremities and in only 5% of cases are the toes involved. [2]

CMF in the long bones is usually metaphyseal and eccentrically located with well defined sclerotic margins on radiological evaluation. Lesions in the small bones are osteolytic with scalloped margins, an appearance which overlaps with Non-Ossifying Fibroma. Calcification within the lesion is visible in most cases. [3,4] There is attenuation, expansion or erosion of the overlying cortex. It usually occurs in the metaphyseal side of the growth plate which is situated proximally in the phalanges.

Intra-medullary tumours and tumour-like lesions of the toe phalanges are rare. Among benign lesions enchondroma is probably the most common and may be clinically and radiologically indistinguishable from CMF. [5]

The diagnosis can usually be made by biopsy because CMF is mostly myxoid and often has osteoclastic giant cells whereas enchondroma is generally more obviously cartilaginous and lacks giant cells. However a small biopsy may not have enough tissue for this distinction and in such instances the only diagnosis possible may be “benign cartilaginous tumour” as in this case. Other benign tumours or tumour-like lesions which may rarely occur in the toes include aneurysmal bone cyst, the closely related giant cell reparative granuloma, and true giant cell tumour all of which are histologically quite different from CMF. [5]

Chondroblastoma can be partially or largely similar to CMF microscopically but is virtually unknown in the toes. [2] Among malignant bone tumours chondrosarcoma can be histologically difficult to distinguish from CMF. For making this differentiation the x-ray is critical as the radiological appearance of chondrosarcoma is aggressive, unlike that of CMF. The toes are also an extremely uncommon site for chondrosarcoma.

Other than benign cartilage lesions, a few other tumors of the terminal digits should be considered in the differential diagnosis. A radiological diagnosis of intraosseous epidermoid cyst can be made in the presence of perilesional sclerosis with painless swelling. [5] A painful lesion with perifocal reactive sclerosis is suggestive of an osteoid osteoma and is easily distinguishable radiologically. [5]

Poorly defined lytic lesions can be associated with several pseudotumorous conditions such as osteomyelitis caused by Staphylococcus seen in diabetic patients. [5] Tuberculous and leprous dactylitis are other condition which are clinically seen as phalangeal lesions. [6] Radiologically the tuberculous lesion is central, lytic, cystic and expansive, and soft tissue extension may be seen. [5] These are easily distinguished histologically.

As our report shows, a needle biopsy is of great value in suggesting the diagnosis and can easily be carried out as an outpatient procedure. Conservative surgical treatment, such as curettage and bone grafting, appears to be ideal for CMF.

In conclusion, a wide range of entities, including CMF, needs to be considered when confronted with a lytic lesion in the toe phalanx. Radiological features may help in distinguishing these conditions. Histopathology and microbiology provide a definitive diagnosis. A thorough surgical clearance is required to avoid recurrence in CMF.


1. Fletcher CDM, Unni KK, Mertens F (eds). Pathology and Genetics of Tumours of Soft Tissue and Bone, WHO Classification of Tumours, IARC Press, Lyon: 243 – 245, 2002.
2. Wu CT, Inwards CY, O’Laughlin S, Rock MG, Beabout JW, Unni KK. Chondromyxoid fibroma of bone: a clinicopathologic review of 278 cases. Hum Pathol 29 (5): 438 – 446, 1998.
3. Schajowicz F , Gallardo H .Chondromyxoid fibroma (fibromyxoid chondroma) of bone. A clinico-pathological study of thirty-two cases.J Bone Joint Surg. 53B (2): 198-216,1971.
4. Sharma H.,Jane M J, Reid R. Chondromyxoid fibroma of the foot and ankle: 40 years’ Scottish bone tumour registry experience. Int Orthop. 30(3): 205 – 209, 2006.
5. Wang BY, Eisler J, Springfield D, Klein MJ. Intraosseous Epidermoid Inclusion Cyst in a Great Toe. A Case Report and Review of the Literature. Arch Pathol Lab Med. 27 (7): 298 – 300, 2003.
6. Olivieri .I.,Scarano E,Padula A,Giasi V,Priolo F. Dactylitis,a term for different digit diseases. Scand J Rheumatol. 35(5): 333 – 340, 2006.

Address correspondence to:Dr Anil Thomas Oommen MS(Ortho)Assistant Profressor,
Dept. of Orthopaedics, Unit 2
Christian Medical College Ida Scudder Road,Vellore 632 004.

1 Assistant Profressor, Department of Orthopaedics, Unit 2
Christian Medical College Ida Scudder Road,Vellore 632 004.
2 Profressor, and Head , Department of Orthopaedics, Unit 2
Christian Medical College Ida Scudder Road,Vellore 632 004.
3 Profressor, Department of Pathology,
Christian Medical College Ida Scudder Road,Vellore 632 004.

© The Foot & Ankle Journal, 2009