The focus of the present letter to editor is on keloid formation and its management
following treatment of congenital form of Boutonniere finger. Keloids and hypertrophic
scars represent fibroblast-mediated hyperactive response to dermal injuries that extend
beyond the wound margins, causing functional and aesthetic impairments.[1] Keloids most commonly develop on chest, ear lobe, shoulders, and upper arm.[2]
[3] Lesions which extend beyond the wound borders are called keloids.[4] They usually appear in areas such as ear, chest, and deltoid regions.[2] They are usually the result of fibroblast abnormal proliferation and accumulation
of excessive extracellular matrix during wound healing.[3] Herein, we describe an atypical keloid on the finger of a 3-year old patient after
treatment of congenital deformity.
Our patient was a 3-year old female with keloid formation following surgical treatment
of congenital deformity. She initially presented with hyperextension in the distal
interphalangeal (DIP) joint, a flexion contracture in the proximal interphalangeal
(PIP) joint, and a decrease in range of motion in the metacarpophalangeal joint (MCP)
in the 3rd digit of her right hand. This was a congenital form of Boutonniere deformity
([Fig. 1]).
Fig. 1 Congenital Boutonniere deformity of third digit prior to operation
She underwent an operation in her second year of life. Open capsulotomy and reduction
of joint displacement of both DIP and PIP were performed. Following releasing the
contracture, central slip of extensor tendon was not found, which was reconstructed.
The joint was kept extended with a k-wire insertion. After 6 weeks, the pin was removed,
and hand physiotherapy was started.
We noted a keloid forming on the dorsal surface of the middle and distal phalanges
of the right middle finger during follow-up assessment in clinic after 3 months ([Fig. 2]). Since the lesion started to grow, three courses of intralesional triamcinolone
were injected every 4 weeks. However, the lesion continued to grow vertically up to
4.5 cm in height and had an odd finger-like appearance ([Fig. 3]).
Fig. 2 The keloid which appeared following treatment of congenital Boutonniere deformity.
Fig. 3 The keloid increased in size within the next 5 months, following successful treatment
of congenital Boutonniere deformity.
The patient had no history of any keloid formation before. The history and physical
examination were otherwise unremarkable.
Due to the functional impairments and enlarged size, the keloid was resected and sent
to the pathology department for histopathological examination. The specimen was reported
to be fibroconnective tissue with acidophilic bands of hyalinized collagen. In addition,
fibroblasts associated with thin-wall blood vessels in the background were reported.
The thickened hyalinized collagen bundles that were reported histologically are indicative
hallmarks of keloid formation. The child was followed up for 8 months after excision
of keloid. The excision was successful in terms of function and structure ([Fig. 4]). However, some extension contracture remained on distal interphalangeal joint,
due to the effect of keloid. Hence, physical therapy is continued for her.
Fig. 4 The third digit appearance after excision of the keloid during the follow-up visit
in 8 months.
Keloids are rare on digits and only 23 cases have been reported in 10 studies, with
most of them appearing following syndactyly treatment.[5] There have been no reports on keloid formation after congenital form of Boutonniere
deformity. There is no consistent standard treatment available for keloids formed
in the toes and fingers. In 2014, Yamawaki et al stated that a keloid formation after
syndactyly release was treated with surgical excision and intralesional steroid injection.[5] Some studies used skin grafts or regional flaps to cover the defect after keloid
resection. Direct closure was performed in 6 cases.[5] We treated this keloid with multiple intralesional triamcinolone injection and surgical
resection successfully.
