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A case of recurrent hyperostotic macrodactyly

Posted on December 31, 2020 | Comments Off on A case of recurrent hyperostotic macrodactyly

by Milad Motalebi Kashani DPM1*, Melinda A. Bowlby DPM2

The Foot and Ankle Online Journal 13 (4): 6

Macrodactyly and its variation hyperostotic macrodactyly are some of the rarest deformities encountered by foot and ankle specialists. Changing the natural aesthetic shape of the foot, limiting the functionality of the lower extremity, and causing pain are some ways that this condition can affect patients’ everyday life and mental health. This study presents a case of recurrent hyperostotic macrodactyly that was managed with surgical intervention in order to debulk the soft tissue and excise excess osseous elements with successful results.

Keywords: congenital deformity, forefoot, lower extremity, foot and ankle surgery

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0006

1 – Swedish Medical Center- Cherry Hill Campus, Seattle, WA PGY-1
2 – Assistant Research Director, Swedish Medical Center-Cherry Hill Campus, Seattle, WA
* – Corresponding author- miladm14@vt.edu

Macrodactyly is a non-hereditary and congenital deformation pathology of the upper and lower extremities which can be bilateral or unilateral [1-5]. In this deformity both osseous and soft tissue components of the digit can be enlarged in size which can cause functional and esthetic problems for the patient. Two types of this condition that were described by Barsky are static, in which deformity is present at birth and increases in size proportionally to other digits, and progressive, in which a digit grows disproportionately to other digits and is most commonly encountered in the lower extremity [1].

Macrodactyly is believed to be originally described by Von Klein in 1824 in the upper extremity and later in 1925 by Feriz in the lower extremity [6-8]. This disorder is thought to be a rare condition and because of that there is no accurate estimate of the prevalence of this disorder. According to some estimates, macrodactyly accounts for 0.9% of all congenital deformities and it is more prevalent in males [9, 10]. The etiology of macrodactyly has been debated over the years, but no clear conclusion has emerged yet. Some cases of macrodactyly present in patients with other disorders such as Proteus syndrome, Neurofibromatosis type 1, Klippel–Trenaunay syndrome, lymphangioma and fibrous dysplasia [8,11,12]. However, in many of the case reports published regarding macrodactyly, this condition is observed as an isolated condition with no other associated disorders [2,3,9,13,14].

Throughout the years, different terms such as macrodystrophia lipomatosa progresia, macrodystrophia lipomatosa, megalodactyly, and localized gigantism have been used in the literature to describe macrodactyly or other variations of this deformity [7]. A unique and less discussed type of progressive macrodactyly is hyperostotic macrodactyly which usually has a later onset than typical macrodactyly and is associated with osteo-cartilaginous mass formation in peri-articular areas of the upper and lower extremity [15].

Figure 1 Preoperative medial oblique X-ray image (on the left) and clinical photo (on the right), prior to second surgery.

Figure 2 Postoperative medial oblique X-ray image, following the second surgery.

The rare presentation of hyperostotic macrodactyly and a lack of literature regarding this topic have provided practitioners with no clear guidelines regarding the management of this disorder. This case report presents a case of a mild recurrent hyperostotic macrodactyly in a patient with previous surgical interventions to address this deformity.

Case Report

A 53-year-old female with no past medical history other than asthma presented to the clinic with a painful recurrent mass on her left hallux. She related that she has had two prior surgeries. The patient reported that she was originally seen regarding this problem when she was 14 years old and was diagnosed with localized gigantism. She had a surgery at that time to fix her deformity which had satisfying results and resolved her problem for about 35 years.

The second surgery was performed four years ago when the patient was seen by another podiatrist regarding this problem. She had noticed an increase in size of her left hallux and denied any trauma to the area. She had pain both on the plantar and medial side of her left hallux and first metatarsal head with noticeable bony prominences. She changed her shoe gear, using slippers or open sandals to accommodate the prominences (Figure 1). In the second surgery, osteophytes from the left first metatarsophalangeal joint and hallux were excised and a soft tissue mass from the plantar aspect of the left hallux was removed as well (Figure 2). During the postoperative period, the patient stubbed her hallux on a heavy plastic bin which was very painful for her, however no fractures were noted, and the rest of her postoperative course was uneventful.

The patient presented to our clinic two years ago due to noticing the recurrence of her deformity after the second surgery. Physical exam revealed approximately a 4 cm x 3 cm firm soft tissue mass overlying the left first tarsometatarsal joint. There was also tenderness with palpation of a prominent exostosis along the medial aspect of the left hallux Interphalangeal joint. Joint motion at first metatarsophalangeal and tarsometatarsal joints were severely limited as well, but not painful (Figure 3). Magnetic resonance imaging report indicated an ovoid, subcutaneous lipoma measuring 5.2 x 2.8 x 1.1 cm. Mild to severe arthritis of the first tarsometatarsal and metatarsophalangeal joints with ossified bodies in addition to fatty infiltration of abductor and flexor hallucis muscles was also noted.

Figure 3 Preoperative clinical photos (in the left and the middle) and dorsoplantar X-ray image (on the right), prior to the third surgery.

After discussing the possible adverse effects, benefits and alternative therapies to the surgery with the patient, the patient wished to proceed with exostectomy and removal of the soft tissue mass. During the third surgery, osseous masses from the left hallux were removed and the soft tissue mass from the dorsal and medial aspect of first metatarsal of the left foot were excised and both specimens were submitted for pathology evaluation (Figure 4). The osseous masses were clinically equivalent with osteophytes measuring 0.8×0.6×0.5cm and 1.2×0.8.0.5cm.

Figure 4 Intraoperative image of the excised fibro-fatty mass, during the third surgery.

Figure 5 Postoperative clinical photo, following the third surgery.

The soft tissue mass revealed mature adipose tissue with features of a lipoma measuring 4.5×2.5×1.3cm. The patient was kept non-weight bearing in a splint for 2 weeks and then she was allowed to weight-bear as tolerated for 2 weeks in a surgical boot. Postoperative course was uneventful, and the patient made good progress, had no complaints, and was satisfied with the results more than 18 months after the third surgery (Figure 5).

Discussion

Hyperostotic macrodactyly is a distinctive type of macrodactyly in which massive osteo cartilaginous deposits are observed around the joints [2,15]. Early in the formation process of these osteo-cartilaginous bodies around the joints, they are mostly cartilaginous and later they are substituted with osseous elements which leads to motion restriction across the affected joint [2]. As a progressive macrodactyly, it is not uncommon to observe fatty growths or lipomas and fatty infiltrations in this condition. In many reported cases this fatty hypertrophy and infiltration can be observed in both plantar and dorsal aspect of the foot [3,14]. The case presented in this article demonstrated both osseous and fatty enlargement across the first ray of the patient’s left foot.

The main goals of treatment for macrodactyly should be providing the patients with a pain free, functional foot that can fit into their shoes [4]. Unfortunately, there are very few reported cases of hyperostotic macrodactyly in the foot, however it appears that the removal and debulking of the excess fibro-fatty tissue and osseous bodies is the best method of management of this disorder which was the way the patient in this case was treated [2,9,13]. In a case report by Katz, the patient presented with lipomas across his right foot and ankle and osteo-cartilaginous growths across his fourth and fifth toes and metatarsals [9]. An surgery was performed on the patient in which the lipomas and osteo-cartilaginous bodies were excised and the fifth toe and half of the fifth metatarsal were resected. The patient had excellent results after the surgery. In another case by Matsuzaki, et al., the patient was suffering from painful and limiting osteo-cartilaginous masses around his left first metatarsal head and ankle [2]. Patient had a previous left second toe and hallux amputation surgery to address his macrodactyly. After removal of these osseous bodies from the patient’s left foot, his pain was relieved and the motion across his ankle joint increased and no recurrence was reported.

One curious aspect of the case presented in this article that requires attention is the recurrence of osteo-cartilaginous bodies almost two years after their resection in the second operation. This seems to be too early for a recurrence to happen considering that it took almost 35 years after the first surgery for her to have problems with her left foot again. A possible explanation for this can be the traumatic event to the area in the postoperative course in which the patient stubbed her big toe straight into a heavy plastic bin. Some studies have suggested that trauma can be the trigger for the osteo-cartilaginous hypertrophy observed in hyperostotic macrodactyly deformity which would explain the recurrence in this case [2,15].

In conclusion, hyperostotic macrodactyly is a rare progressive form of macrodactyly in which massive and limiting periarticular osteo-cartilaginous bodies in addition to fatty tissue hypertrophy can form. Surgical intervention to remove these osseous and fatty masses in cases that they cause pain and functional disability due to blocking joints appears to be the best method to treat this condition. It is also very important to educate patients to avoid trauma to the areas affected by hyperostotic macrodactyly since trauma appears to be one of the causes of this disorder or its recurrence.

References

  1. Barsky AJ. Macrodactyly. The Journal of Bone & Joint Surgery. 1967 Oct;49(7):1255-66.
  2. Matsuzaki T, MD, Hitora T, MD, Akisue T, MD, Imaizumi Y, MD, Yamagami Y, MD, Yamamoto T, MD. Massive Heterotopic Ossification around the Ankle in a Patient with Macrodactyly of the Foot: A Case Report. Journal of Foot and Ankle Surgery, The. 2012;51(5):648-51.
  3. Zhang X, Liu Y, Xiao B, Li Y. Two cases of macrodactyly of the foot: relevance in pediatric orthopedics. Journal of Pediatric Orthopaedics B. 2016 Mar;25(2):142-7.
  4. Hop MJ, MD, van der Biezen, Jan Jaap, MD, PhD. Ray Reduction of the Foot in the Treatment of Macrodactyly and Review of the Literature. Journal of Foot and Ankle Surgery, The. 2011;50(4):434-8.
  5. Kalb JP, Suarez DA, Herrera AM. Bilateral Macrodactyly of the Halluces in an Adolescent Girl Corrected with Shortening Osteotomies of the First Metatarsal and the Phalangeal Bones: A Case Report. JBJS case connector. 2018 Jul;8(3):e58.
  6. Klein W, Germann G, Bosse A, Müller KM, Steinau HU. Clinical aspects, morphology and therapy of an unusual case of bilateral macrodactyly. Handchirurgie, Mikrochirurgie, plastische Chirurgie. 1993 Jan;25(1):12.
  7. Tatu RF, Anuşca DN, Dema ALC, Jiga LP, Hurmuz M, Tatu CS, et al. Surgical treatment in a case of giant macrodystrophia lipomatosa of the forefoot. Romanian journal of morphology and embryology. 2017;58(3):1115.
  8. Natividad D, Ellise, Patel D, Kinna. A Literature Review of Pedal Macrodactyly. Foot & ankle journal (Online). 2010 May.
  9. Katz JB. Progressive macrodactyly. The Journal of Foot and Ankle Surgery. 1999;38(2):143-6.
  10. Wang S, Han Z, Liu X. Hyperhidrotic Macrodactylism Caused by Osteoid Osteoma: A Case Report and Review of the Literature. The Journal of Foot and Ankle Surgery. 2019 May;58(3):586-90.
  11. Bulut M, Karakurt L, Belhan O, Serbest S. Ray amputation for the treatment of macrodactyly in the foot: report of three cases. Acta orthopaedica et traumatologica turcica. 2011;45(6):458- 62.
  12. Rampal V, Giuliano F. Forefoot malformations, deformities and other congenital defects in children. Orthopaedics & Traumatology: Surgery & Research. 2020 Feb;106(1):S115-23.
  13. Mullins AM. Multiple Lower Limb Osteophytosis with Macrodactyly: A Case Report. Foot & Ankle International. 1996 May;17(5):283-5.
  14. Yushan M, Alike Y, Keremu A, Abulaiti A, Ren P, Yusufu A. Precise Resection of Macrodactyly Under Assistance of Three-Dimensional Reconstruction Technology: A Case Report. The Journal of Foot and Ankle Surgery. 2020 Jan;59(1):125-7.
  15. Schuind F, Merle M, Bour C, Michon J. Hyperostotic macrodactyly. Journal of Hand Surgery. 1988 Jul;13(4):544-8.

 

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Persistent distal sciatic neuropathy following popliteal nerve block in foot and ankle surgery

Posted on August 31, 2016 | Comments Off on Persistent distal sciatic neuropathy following popliteal nerve block in foot and ankle surgery

by Spencer J. Monaco DPM1, Alissa Toth DPM2, Dane K. Wukich MD3pdflrg

The Foot and Ankle Online Journal 9 (3): 1

Popliteal nerve blocks are becoming more popular in patients undergoing foot and ankle surgery. The procedure potentially carries fewer complications and is frequently successful while allowing for earlier mobilization when compared with spinal or epidural anesthesia. Reported complications include paresthesias, pain during needle entry and blood aspiration without risk of dural injury or post procedure headache. We present two patients who underwent a popliteal nerve block for a foot and ankle surgery who developed mixed sensory and motor neuropathy that did not fully resolve within their follow up period.

Key words: popliteal nerve block, foot and ankle surgery, sciatic nerve

ISSN 1941-6806
doi: 10.3827/faoj.2016.0903.0001

1,2 – Resident Physician, University of Pittsburgh Medical Center, Podiatric Residency Program, Pittsburgh PA
3 – Professor of Orthopaedic Surgery, Division of Foot and Ankle Surgery, Pittsburgh PA
* – Corresponding author: monacosj2@upmc.edu

Operative and postoperative analgesia has been provided in varying forms which include general anesthesia, spinal or epidural anesthesia, local anesthesia with IV sedation, and peripheral nerve block [1]. Popliteal nerve blocks are becoming more popular in patients undergoing foot and ankle surgery, allowing for earlier mobilization compared with spinal or epidural anesthesia. As a matter of fact, they are being increasingly performed by foot and ankle surgeons rather than by an anesthesia service [5]. The popliteal nerve block was first described by Gaston Labat in 1922 and can be administered from a posterior or lateral approach, with or without the assistance of ultrasound or nerve stimulation. It is believed that the anesthetic interferes with the sodium and potassium channels thus interfering with the action potential [1].

Borgeat et al retrospectively evaluated 1001 patients and reported on complications such as paresthesias, pain during anesthetic administration and blood aspiration [2]. They concluded the procedure is frequently successful and causes few complications.

In 2014, a study reviewing 143 popliteal blocks performed by podiatric surgical residents showed no postoperative complications but an overall success rate of only 76.2% [5]. The purpose of this paper is to present two patients who developed persistent mixed sensory and motor neuropathic syndromes from a popliteal nerve block following a foot and ankle surgical procedure that were still present at final follow up.

Case 1

A 46 year old female presented to our foot and ankle clinic in regards to a right foot drop. She underwent a peroneal tendon repair 8 months prior at an outside facility. She was able to walk with a limp before her surgery however, is now unable to put her foot flat on the ground. During her procedure a calf tourniquet was used for 30 minutes at a setting of 350 mmHg. She received a popliteal nerve block without the use of ultrasound or nerve stimulation. The patient reported the block did not work and she was able to feel her leg and foot before surgery.

1

Figure 1 Clinical photograph of 25 degrees plantarflexion.

Upon presentation to our clinic, she complained of paresthesias including tingling in her entire foot and numbness in the S1 nerve distribution. She tried multiple custom made ankle and foot orthotics with no relief.  She has past medical history of psoriatic arthritis. Past surgical history includes right finger soft tissue mass excision and hysterectomy. Medications include meloxicam and gabapentin.

Physical examination revealed an alert and oriented female with a BMI of 25. Overall her pain was 6 out of 10. She had palpable pedal pulses. Light touch and vibratory sensation were intact. Achilles and patellar deep tendon reflexes were also intact. Her ankle was fixed at 25 degrees of plantarflexion which was non-reducible and did not improve with knee flexion (Figures 1 and 2). Manual muscle testing demonstrated 3/5 inversion and eversion, 4/5 digital plantarflexion and dorsiflexion and 3/5 ankle dorsiflexion.  Mid-calf circumference was six centimeters less than the non-affected side. Electromyography (EMG) and nerve conduction velocity (NCV) studies showed acute axonal degeneration in muscles innervated by the tibial, superficial peroneal, lateral plantar and deep peroneal nerves consistent with a distal sciatic neuropathy.  A 3T MRI scan was completed which showed signal intensity of the posterior tibial muscle and soleus muscles indicating atrophy. She underwent a Z lengthening of the triceps surae and posterior ankle joint capsule release to correct the equinus deformity (Figure 3). At 4-month follow up, the patient’s foot remained at 90 degrees relative to the leg, however, had continued neuropathic symptoms. She was referred to peripheral nerve surgery for possible neurolysis and nerve grafting.

2

Figure 2 Clinical photograph illustrating equinus deformity during weightbearing.

3

Figure 3 Intraoperative photograph of Z lengthening with posterior ankle joint capsule release.

Case 2

A 17-year-old male sustained a 5th metatarsal zone 2 injury of his right foot and was treated with percutaneous intramedullary screw fixation. He received a preoperative regional nerve block by the anesthesia service. Ultrasound or nerve stimulation was also not used.  During his procedure a calf tourniquet was used for 45 minutes at 250 mmHg. During his postoperative course, he developed ipsilateral calf and intrinsic foot muscle atrophy along with pain he described as “pins and needles.” He had an unremarkable past medical history. He had no other past surgical history.

The patient’s BMI was 26.9. Physical examination revealed impaired sensation in the peroneal and tibial nerve distributions at the pedal level. Strength testing revealed 4/5 strength of the tibialis anterior and gastrocnemius muscles. Extensor hallucis longus was 4/5 with full strength to hamstrings, quadriceps, and adductors. EMG/NCV studies showed chronic right sciatic neuropathy distal to the biceps femoris and semimembranosus muscles at 12 months following surgery as well as severe axon loss to intrinsic foot muscles. He was referred to physical medicine and rehabilitation. He was recommended custom orthotics and exercises as well as a home transcutaneous electrical nerve stimulation unit. He was also given B12 vitamin complex and fish oil. His symptoms improved with the exception of intrinsic muscle function and tone, which was persistent at 2 year follow up.

Discussion

Motor and/or sensory neuropathy from a popliteal nerve block is uncommon for patients undergoing foot and ankle surgery with reported incidence of between 1.26% and 5% [1-2]. In a recent retrospective study of 1014 patients who had a popliteal block for foot and/or ankle surgery, the overall success rate was 97.3%. 135 patients reported varying manifestations of neuropathic complications.  Eight of these patients retrospectively reviewed developed exclusively motor deficits, 118 exclusively sensory deficits and the remaining nine patients reported mixed sensory and motor deficits.

At final follow up, 14 patients had residual neuropathic symptoms. No statistical significance was found between tobacco use, diabetes, tourniquet location or time, block procedure techniques, single or continuous blocks, or ultrasound or nerve stimulation [1].

A retrospective study of popliteal nerve blocks for hallux valgus surgery showed an incidence of 1.91% for 157 consecutive hallux valgus surgeries. 44% of the blocks were performed with ultrasound in conjunction with nerve stimulation [4].

In 2012, Gartke et al prospectively studied the effects of continuous rather than single shot popliteal blocks in foot and ankle surgery [3]. The study showed a 41% incidence at 2 weeks that decreased to 24% at 8 months. In this study, only 4% of the patients manifested symptoms to warrant referral to a neurologist or pain specialist.  

Although regional nerve blocks prior to foot and ankle surgery are generally effective and obviate the negative side effects of opioids or other sedation, careful patient counseling should be planned prior to the procedure. Continuous popliteal nerve blocks may have a higher incidence of transient postprocedural neuropathy versus single shot blocks. Although the majority of neuropathies are isolated sensory deficits that resolve in a period of months, we present two cases of mixed sensorimotor deficits that persisted beyond final follow up. Interesting, both patients that developed distal sciatic neuropathy did not have guidance from either an ultrasound or nerve stimulator during the nerve block. Moving forward, all patients at our institution undergoing a popliteal nerve block have either ultrasound guidance and/or nerve stimulation which is performed by the anesthesia service.

References

  1. Anderson JG, Bohay DR, Maskill JD, et al. Complications After Popliteal Block for Foot and Ankle Surgery. Foot Ankle Int. 2015;36(10):1138-43.
  2. Borgeat A, Blumenthal S, Lambert M, Theodorou P, Vienne P. The feasibility and complications of the continuous popliteal nerve block: a 1001-case survey. Anesth Analg. 2006;103(1):229-33.
  3. Gartke K, Portner O, Taljaard M. Neuropathic symptoms following continuous popliteal block after foot and ankle surgery. Foot Ankle Int. 2012;33(4):267-74.
  4. Hajek V, Dussart C, Klack F, et al. Neuropathic complications after 157 procedures of continuous popliteal nerve block for hallux valgus surgery. A retrospective study. Orthop Traumatol Surg Res. 2012;98(3):327-33.
  5. Hegewald K, McCann K, Elizaga A, Hutchinson BL. Popliteal blocks for foot and ankle surgery: success rate and contributing factors. J Foot Ankle Surg. 2014;53(2):176-8.

 

 

 

 

 

 

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