Surgical management of extensive hypertrophic scarring of the halluces secondary to a decade of untreated onychocryptosis: An illustrative case report

Introduction

Excessive scars form as a result of aberrations of physiologic wound healing and may arise following insult to the deep dermis, ¹ as can occur with onychocryptosis pathology. Common scar types include hypertrophic and keloid scars which cause pain, pruritus and contractures that significantly affect the patient’s quality of life both physically and psychologically. ¹

Hypertrophic scarring usually occurs within 4–8 weeks following wound infection, wound closure with excess tension or other traumatic skin injury. ² Keloids, however, often form as a result of a strong genetic predisposition ³ and may originate on any part of the body in predisposed individuals. ⁴ Unusual features, such as advanced age, lack of trauma preceding keloid formation or the presence of a keloid-like lesion in uncommon sites, should prompt the clinician to seek histopathological confirmation of the diagnosis. ⁵ The authors present a rare case of surgical management of keloid-like fibrous lesions (irritated hypertrophic scar tissue) on the halluces following more than a decade of untreated onychocryptosis.

Case report

A 31-year-old White male IT consultant was referred for a podiatric surgical opinion regarding the large soft tissue masses that had developed on his great toes. The patient had a history of onychocryptosis during late adolescence and developed a needle phobia following lateral nail wedge procedure (without matrixectomy) performed under local anaesthesia by a previous practitioner. He had no further medical history and no known allergies. The patient reported his hallucal toenails remained ingrown into adulthood and he developed hard lesions around the nail edges, which progressively worsened over 10 years, particularly on the left side.

The halluces were painful and pruritic at times particularly when stubbed; however, he had developed a level of tolerance to these symptoms. The patient practiced good foot hygiene and had applied povidone-iodine 10% w/v solution to the halluces periodically when they appeared inflamed. He had occasionally taken simple analgesic and anti-inflammatory medication during pain flares; however, he had not sought professional care due to his needle phobia. His shoes were worn larger than normal in order to accommodate the lesions. Despite this, the pain precluded him from physical activity and he had gradually gained weight, his body mass index (BMI) was 33.2.

In addition to his physical discomfort, he also experienced psychosocial effects. His feet were shod with shoes and/or socks at all times, he avoided public transport and other crowded places out of fear of being stepped on, he avoided the beach and other settings where there was a possibility of having to remove his shoes and he had not revealed his bare feet to his partner of 2 years.

Physical examination revealed large hallucal periungual soft tissue masses L >> R which were suspected to be due to fibrosed granulation tissue (Figure 1). The left hallucal soft tissue mass had proliferated significantly and overlapped the adjacent left second and third toes. The nail tissue beneath the masses was macerated and malodourous; however, there was no significant rubor or cellulitis present. There were no hypertrophic scars on other parts of his body from previous cuts and superficial injuries and to his knowledge there was no familial history of a keloid scarring predisposition.

OPEN IN VIEWER

Figure 1.

Large periungual soft tissue masses L >> R secondary to more than a decade of onychocryptosis. (a) Left dorsal aspect, (b) left frontal aspect and (c) right dorsal aspect.

The patient expressed preference for the procedure(s) which had the most likelihood of resolution with the least risk of revision surgery. As a result, complete excision of the soft tissue masses, nail matrixectomy and distal tuft resection to ensure closure without wound tension was proposed (modified Zadik–Syme procedure). The risks and complications typical of this type of surgery and in particular that his great toes would be shorter in appearance and without toenails, was discussed. Pedal neurovascular assessment revealed no apparent deficiency (NAD) and the patient was deemed fit for surgery. Informed consent was obtained and he was scheduled for bilateral procedures under general anaesthetic as ambulatory day case podiatric surgery.

Surgical technique

The feet and lower limbs were prepped and draped in typical fashion. Local anaesthetic mayo blocks were administered bilaterally while the patient was under general anaesthesia with a total of 20 mL 0.75% ropivacaine hydrochloride combined with 8 mg/2 mL dexamethasone sodium phosphate. Calf tourniquets were applied at a pressure of 250 mmHg following exsanguination with a sterile Esmarch bandage. The periungual soft tissue masses were excised, and modified Zadik–Syme total nail matrixectomies were performed followed by plastic remodelling of the surrounding skin for even wound edges (Figure 2). The distal tuft of the distal phalanges was resected to allow tissue approximation without tension and care was taken not to excessively shorten the distal phalanges. The wound edges were carefully approximated with 3-0 Monocryl (poliglecaprone 25) using simple interrupted sutures (Figure 3). The halluces were dressed with Jelonet, Betadine antiseptic ointment, wet to dry gauze and modified Jones compression dressings to both feet.

OPEN IN VIEWER

Figure 2.

The (a) left and (b) right halluces following excision of the fibrous masses, the nail matrices and resection of the distal tuft of the terminal phalanges.

OPEN IN VIEWER

Figure 3.

The (a) left and (b) right halluces following wound approximation with simple interrupted sutures.

Following resection, the soft tissue masses were found to have emanated from the nail sulci as suspected. The masses had not invaded the deeper subcutaneous tissues or the underlying phalanges. There was no abscess or purulence evident. The nail plates had been partially enveloped by the lesions, which resulted in maceration of the nail margins; however, no further nail irregularity or subungual pathology was observed. For these reasons, only the excised soft tissue masses were sent for histopathology and multiple sections were examined. Further information may have been derived by examination of specimens of local phalangeal, nail and/or matrix tissue; however, this was not deemed clinically necessary at the time.

The histology results showed proliferative bundles of mature fibrous/fibroblastic tissue with scattering of chronic inflammatory cells, including many plasma cells and lymphocytes. The overlying epidermis was histologically unremarkable. There was no evidence of malignancy. The features were those of a benign fibrous lesion, consistent with irritated hypertrophic scar due to a clinical history of onychocryptosis (Figure 4).

OPEN IN VIEWER

Figure 4.

(a) Haematoxylin and eosin–stained section of the hypertrophic scar tissue show polypoid/lobulated expansion throughout the dermis. The overlying epidermis is histologically unremarkable (×12.5) and (b) dense thin collagen fibres are seen with proliferative bundles of mature fibrous/fibroblastic tissue with a scattering of perivascular chronic inflammatory cells (×400).

Following the first dressing change at 7 days, the great toes were redressed weekly with Betadine, Cutiplast and Coplus light digital compression for 2 weeks at which point the suture material was removed. Coplus compression was applied for further 3 weeks and then a Silipos toe cap was worn for 6 weeks to assist with scar tissue maturation. At 12 weeks post-op, there was no sign of scar tissue hypertrophy and the toes displayed close to normal morphology, albeit with anonychia (Figure 5). The patient was satisfied with the aesthetic result and more importantly he had gained confidence to return to crowded social settings, wear appropriately fitted footwear and increase his physical activity. At 5.5-year follow-up, the patient reported he was completely satisfied with the result (Figure 6). He was particularly thankful for being able to kick a football comfortably and wear flip-flips during hot Australian summers without being self-conscious.

OPEN IN VIEWER

Figure 5.

The (a) left and (b) right wounds exhibited progressive maturation at 12 weeks post-operatively.

OPEN IN VIEWER

Figure 6.

Long-term follow-up (5.5 years) shows no recurrence and relatively normal toe morphology: (a) left and (b) right.

Discussion

It is not unusual for individuals to suffer with hypergranulating onychocryptosis for a number of years prior seeking professional treatment. Reluctance may be due to needle phobia, procedural anxiety or a hope that the condition may regress and be self-limiting. If left untreated, the condition can lead to disfigurement of the halluces due to the development of fibrous lesions, skin bridges ⁶ and keloids. ⁷ Psychosocial effects can lead to further avoidance in seeking treatment, as demonstrated in this case.

There are a variety of other focal masses that can occur in the digits which enter the differential diagnosis. These can include dermatofibroma, ⁸ dermatofibrosarcoma, ⁹ desmoid tumour ¹⁰ and giant cell tumour. ¹¹ Periungual lesions such as verrucae ¹² and fibromata ¹³ are commonly seen while tumours arising from the nail matrix such as onychoblastoma (nail unit hamartoma) ¹⁴ and onychomatricoma ¹⁵ are rare. Subungual tumours can include glomus tumour, subungual exostosis, soft tissue chondroma, keratoacanthoma, hemangioma and lobular capillary hemangioma. ¹⁶ Benign cystic lesions such as epidermal and mucoid cysts also occur in the digits. Malignant tumours including squamous cell carcinoma and malignant melanoma must also be considered. Although some lesions may display distinct clinical characteristics, histopathologic analysis is critical for definitive diagnosis.

Hypertrophy of the digits in the form of digital clubbing can be due to physical manifestations of serious diseases such as infection, malignancy and other inflammatory conditions. ¹⁷ Digital clubbing, as seen in Rubinstein–Taybi Syndrome, has been reported to lead to nail abnormalities and chronic onychocryptosis. ¹⁸ Other reports of onychocryptosis associated with a digital tumour include the presence of cutaneous leiomyosarcoma. This is a very rare malignant tumour, which can present as a periungual non-healing pyogenic granuloma. ¹⁹

In 2012, Lee et al. ⁷ reported a rare case of confirmed keloid scar tissue which occurred on the left great toe of a 22-year-old female secondary to chronic paronychia and onychocryptosis. The patient had a history of keloid on her left earlobe after ear piercing, which improved after excision and intralesional steroid injection. The keloid lesions (medial and lateral sulci) on her great toe developed after 7 years and multiple failed wedge resections in the primary care setting. The keloids underwent tangential excision by a plastic surgeon and a multimodal post-operative wound care regimen was implemented which included topical anti-inflammatory and antibiotics, ibuprofen (3/52), tanilast (6/12), intralesional triamcinolone injection and silicone gel application. At the 14-month review, normal appearance of the great toe was maintained.

The scar tissue observed in our case demonstrated clinical keloid-like characteristics. The lesions extended beyond the original confines of the wound and displayed persistent enlargement ²⁰ without a quiescent or regressive phase. However, the authors did not expect the lesions to be keloids as they did not invade the surrounding skin, there was no hyperpigmentation or colour change over time, no ulceration, no obvious epithelial thinning and they projected only from the areas of skin affected by the cryptotic nail edges. The clinical features were indicative of hypertrophic fibrous lesions as they occur when the inflammatory response to injury is prolonged. ²¹ It is recommended that clinical suspicion alone should not be used to differentiate between hypertrophic scars and keloids as incorrect identification may result in inappropriate management. ¹ As a result, the excised masses were sent for histopathologic testing.

Distinguishing hypertrophic scar from keloid histopathologically is sometimes difficult as the presence of the hallmark sign of keloid scar tissue, keloidal collagen, has only been found to have a 55% diagnostic value. Similarly, alpha-smooth muscle actin, a differentiating marker of hypertrophic scarring, is variably expressed in both forms of scar tissue. ²² Fortunately, the use of the scanning electron microscope can help to demonstrate distinct morphological differences. Normal skin contains distinct collagen bundles, the majority of which run parallel to the epithelial surface. Collagen bundles in hypertrophic scars, as seen in this case, exhibit a nodular structure, they are flatter and less clearly demarcated, loosely arrayed in a wavy pattern, fragmented and shortened, but the majority of bundles lie parallel to the epithelial surface. Such nodular structures are always present in hypertrophic scars and rarely in keloids. Keloid collagen fibrils are larger, more irregular and packed closely together. Discrete collagen bundles are virtually non-existent and the fibres lie haphazardly connected, loose sheets that appear randomly oriented to the epithelial surface. ²³ In a study of the histopathological differential diagnosis of keloid and hypertrophic scars, ²² it was found that in scars with no detectable keloidal collagen, the presence of the following features favours the diagnosis of keloid: non-flattened epidermis, non-fibrotic papillary dermis, a tongue-like advancing edge, horizontal cellular fibrous band in the upper reticular dermis and prominent fascia-like band.

The histopathologic analysis of the fibrous masses in our case showed proliferative bundles of mature fibrous/fibroblastic tissue with a scattering of chronic inflammatory cell infiltrate, including many plasma cells and lymphocytes. The overlying epidermis was histologically unremarkable and there was no evidence of malignancy. The histological features were those of a benign fibrous lesion, consistent with irritated hypertrophic scar tissue secondary to chronic onychocryptosis. These findings are distinguishable from keloid because important features of keloid such as hyalinization (thick, glassy collagenous fibres that are deeply acidophilic and arranged haphazardly) and epidermal atrophy were missing. ²⁰

In light of the histopathology results, the multimodal approach to scar management often adopted for keloids was not indicated. In order to maximize the aesthetic result however, methodical intraoperative approaches and adjuvant conservative therapies were implemented for 3 months post-operatively. These methods included careful surgical technique and preparation of clean wound edges, even approximation of wounds with minimal tension, perioperative antibiotics and antiseptic dressings to reduce the risk of infection and subsequent delayed healing, early compression therapy ²⁴ to reduce excessive oedema and maintain normal digital morphology and the use of silicone gel ²⁴ toe caps once the wounds had epithelialized. Although the mechanisms by which these methods assist have not been definitively established, it is preferable to prevent scarring by minimizing risk factors as much as possible. ²⁵ Ultimately, the goal of treating scars is to restore functionality, provide relief of symptoms, enhance cosmetics and prevent recurrence, ²⁶ all of which were achieved in this rare case.

Conclusion

The authors present a case of keloid-like fibrous lesions of the halluces facilitated by a 10-year history of chronic onychocryptosis. Surgical management involved bilateral hallucal modified Zadik–Syme procedures. Close post-operative monitoring and multimodal scar tissue prevention were implemented. The lesions were confirmed as benign and differentiated from keloid scarring based on clinical and histopathology results. Long-term follow-up (5.5 years) revealed no sign of recurrence and the halluces were only marginally shorter providing good cosmesis and function. The patient indicated complete satisfaction and a return to regular footwear and social activities. Chronic onychocryptosis can trigger and facilitate proliferation of hypertrophic scar tissue widely exceeding the normal tissue margins; excision with total matrixectomy can provide excellent and reliable long-term outcomes.