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

Posted on December 1, 2012 | Comments Off on Left Underlapping Third Toe in a Patient who Underwent Ventricular Assist Device Implantation: A Case Report and Literature Review

by Massimiliano Polastri, MSc, PT1 , 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)

PolOLtoeFig1

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

PolOLtoeFig2

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

PolOLtoeFig3

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.

Discussion

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.

References

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

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