International Journal of Surgery Case Reports 114 (2024) 109087

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International Journal of Surgery Case Reports

journal homepage: www.elsevier.com/locate/ijscr

Case report

Neglected bilateral triphalangeal of the thumb delta type in adult case: A

case report
Meirizal *, Hilmi Muhammad, Brahmantyo Danang Guntoro, Rahadyan Magetsari
Dept. of Orthopedic and Traumatology Sardjito General Hospital, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Yogyakarta, Indonesia

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction: In rare case, thumb has extra phalanges known as triphalangeal of the thumb (TPT). Patients with
Case report TPT can have difficulty doing work/activities that require high precision. Therefore, surgical intervention is
Congenital deformity essential. This report provides an approach for a patient with TPT.
Triphalangeal thumb
Presentation of case: A patient with TPT who underwent removal of extra phalanges and arthrodesis of inter­
Thumb deformity
phalangeal (IP) joints is presented. The left thumb deviated 25o to ulnar while the contralateral part deviated 15o
Hand reconstruction
Triphalangeal thumb management to radial. X-ray revealed both thumbs had extra delta-shaped middle phalanges. Complete excision of extra
phalanges and simple arthrodesis of IP joints with two K-wires in 10◦ to 15◦ flexion was performed. Healing
process ended without any complications and the patient had an improvement.
Discussion: Productive-age patients with TPT can have difficulty doing work and activities that require high
precision, especially in the non-opposable type of the right hand. Furthermore, the female patient is highly
emphasizing the cosmetics of her hand to increase her self-confidence. Therefore, surgical intervention is
essential for this patient. We performed complete excision of extra phalanges and simple arthrodesis of IP joints
with two K-wires in 10◦ to 20◦ flexion. The first K-wire is introduced intramedullary as a primary fixator for
longitudinal alignment, and the second wire is inserted obliquely as an anti-rotation wire. Functional outcome
was assessed after 6 months post-removal of the wire which gave a satisfying result.
Conclusion: TPT is a rare anomaly which surgical intervention can improve the appearance and the precision of
the hand.

1. Introduction maintained in isolated TPT, though its power is reduced. Sometimes,

The first finger of the palm, the thumb, generally only has two bones
inside of it. However, on rare occasions, some humans have more than
two bones. In 1559, the first case of three bones of the thumb was
described by Columbo et al. (1559). Later, this condition is known as
triphalangeal of the thumb (TPT). The epidemiology of TPT in Southeast
Asia is unknown since the condition is rare. In the Netherlands, it is
known that TPT occurs in people with a 1:16,000 ratio [1]. Nowadays, it
turns out that TPT is predominantly an inheritable condition in a
dominant manner [2]. If the TPT is inherited, both thumbs are affected.
Based on opposition movement, TPT can be classified as opposable or
non-opposable [3]. Rarely, TPT can be accompanied by a syndrome such
as Holt-Oram syndrome or Laurin-Sandrow syndrome [1,4]. When TPT
is followed by these syndromes, the thumb is usually hypoplastic and
cannot do an opposition movement. The opposition movement is
TPT can be accompanied by polydactyly. Based on the shape of the extra
phalanx, TPT can be divided into delta, trapezoid, and complete types.
Furthermore, the presentation of syndactyly can classify the TPT into
Triphalangeal Thumb-Polysyndactyly Syndrome (TPT-PS), Haas-type
Polysyndactyly, and Laurin-Sandrow Syndrome (LSS), as proposed by
experts [4].
Clinically, patients with TPT have a ridiculously long thumb,
accompanied by deviation of the thumb due to the length and shape of
the extra phalanx [1,5]. It can be thumb-like or more finger-like.
Depending on the length of the thumb and the presence of hypoplastic
muscle, TPT can be hypermobile at the metacarpophalangeal (MP) joint,
while the carpometacarpal (CMC) joint can be hypoplastic, malformed,
or absent [1]. Due to this condition, the CMC joint becomes less mobile
and diminished, particularly in the finger-like thumb. In the early years,
TPT patients may not complain of any disturbance caused by TPT. As

* Corresponding author at: Hand and Microsurgery Division, Department of Orthopedics and Traumatology, Faculty of Medicine, Public Health and Nursing,
Universitas Gadjah Mada, Dr. Sardjito Hospital, Jl. Kesehatan No. 1, Yogyakarta 55281, Indonesia.
E-mail address: meirizal426@gmail.com (Meirizal).

https://doi.org/10.1016/j.ijscr.2023.109087
Received 23 October 2023; Received in revised form 17 November 2023; Accepted 20 November 2023
Available online 24 November 2023
2210-2612/© 2023 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY license
(http://creativecommons.org/licenses/by/4.0/).
Meirizal et al.
time goes on, patients may encounter difficulties in daily activities that
require oppositional movement and precise movement, such as writing,
picking up a small object, etc. On other occasions, the patient may not
experience these difficulties but feel unconfident with the appearance of
their thumb. Generally, parents are the ones who seek a correctional
surgical procedure.
Since its findings, however, there has been no guideline regarding
how to approach a patient with TPT of any age. Hence, this report
provides an approach for a patient with TPT. Also, the work has been
reported in line with the SCARE criteria [6].
2. Case report
A 23-year-old female presented to our hospital with bilateral
congenital malformed thumbs. The patient had understood her condi­
tion since she was a child and refused to undergo surgical correction. In
adulthood, the patient encountered two main problems. First, she usu­
ally felt unconfident due to the cosmetic appearance of her thumbs.
Second, those thumbs disturbed her activities when precise movements
were needed, such as sewing. This activity, sewing, is essential for her
job as a tailor. As time went on, the patient felt more uncomfortable with
her thumbs since they made sewing difficult. Finally, the patient and her
parents sought medical advice for her thumbs.
Physical examination showed that the left thumb deviated 25◦ to the
ulnar side, while the contralateral thumb deviated 15◦ to the radial side
without any swelling in either thumb (Fig. 1). There was no sign of pain
or inflammation in either thumb. Both thumbs also did not suffer from
limited movement in abduction and flexion. Other than the malforma­
tion, there was no medical condition such as anemia or a history of
trauma. The Disability of Arm, Shoulder, and Hand (DASH) score was
evaluated to assess the specific disability of the extremity, and the result
was 17.5. The Sollerman Hand Function (SHF) and Michigan Hand
Outcome were also evaluated, giving scores of 56 and 60 for the left and
right hand on the SHF test, and 65.6 and 66.6 for the left and right hand
on the Michigan Hand Outcome.
Under the plain x-ray of the thumb (Fig. 2), it was revealed that both
thumbs had an extra delta-shaped bone between two normal bones at
the distal interphalangeal (DIP) joint. Hence, the patient was diagnosed
with bilateral delta-type TPT.
The patient underwent operation for the correction of both thumbs.
In general anesthesia, an H-shaped incision was made on the dorsal side.
To expose the accessory phalanx, the extensor tendon was split longi­
tudinally from the PIP joint through the DIP joint. Then, the accessory
phalanx was removed, followed by ligament and tendon reconstruction.
The distal phalanx and the proximal phalanx were placed close
together with denudation of the articular surface. The first K-wire was
introduced retrogradely to the distal phalanx intramedullary with the
joint flexed to gain an optimal view, then with an antegrade fashion,
advanced the K-wire to the proximal phalanx. After the IP joint was in
adequate contact, the IP joint was bent in a 10◦ –15◦ flexion position. The
first K-wire was the primary fixator for longitudinal alignment. Then,
the second K-wire was introduced obliquely over the IP joint. The second
K-wire acted as an antirotation wire to stabilize the joint (Fig. 3). After 3
Fig. 1. Clinical picture of both thumbs at pre-operative condition.
2
International Journal of Surgery Case Reports 114 (2024) 109087
Fig. 2. X-ray of both thumbs at pre-operative condition.
days of observation, there were no complications.
Six months later, during the follow-up, the K-wire was removed
(Fig. 4). There were no chief complaints or adverse events before or after
the K-wire removal. To examine the function of the hand after the
correctional surgery, the SHF test was executed. The patient also per­
formed tasks with her hands, such as inserting a thread into a pinhole
precisely, and returned to work earlier. Both of her hands had a satis­
fying outcome, with an overall score of 80 and 79 in the dominant and
non-dominant hands, respectively. The Michigan Hand Outcome score
was 97.2 and 98.9, and the DASH Score was 7.
3. Discussion
TPT is a rare congenital anomaly with an incidence of 1 in 25,000
live births. It is not common to find TPT cases in young adults because,
usually, parents of TPT children seek correction surgery early in life. An
adult with TPT may find difficulties whenever they need to do precise
work. Most surgical intervention has been done in TPT with other
conditions such as polydactyly, syndactyly, web contracture, and thenar
muscle atrophy [4]. In contrast, this patient does not have any other
medical condition other than TPT. Regardless, surgical intervention is
essential for this patient to achieve a more precise movement and regain
her confidence by improving the appearance of patient’s thumbs
(Fig. 5).
Since 1976, TPT correction surgery has consisted of 3 interventions:
removal of the accessory phalanx, arthrodesis of the interphalangeal
joint, and reduction osteotomies [8]. The removal of the accessory
phalanx corrects the angulation and length of the thumb; however, this
procedure may reduce the stability of the thumb. Furthermore, the
removal of additional phalanxes requires a reconstruction of the
collateral ligament. Moreover, according to a report [9], the power of
the thumb with TPT diminished significantly when compared with the
unaffected thumb, although reconstruction surgery had been done. They
assumed the surgical intervention might spare the anatomical correction
without any significant improvement in the thumb’s function. In some
experts study [10], most TPT patients had hypoplastic thenar, and this is
the sole cause of diminished function of the thumb. Therefore, it is
recommended for TPT patients under 2 years old to allow early cerebral
integration of the reconstructed thumb. In the case of teenage or adult
TPT, the stability of the joint can be partially achieved with the help of
K-wire insertion.
In this case report, a simple accessory phalanx removal and
arthrodesis were performed without reductive osteotomies. To achieve
the desired stability of the joint, two non-parallel K-wires were inserted
Meirizal et al. International Journal of Surgery Case Reports 114 (2024) 109087

Fig. 3. Representative operative procedure of the left hand with TPT [7].

4. Conclusion

TPT in adults is a rare condition since most TPT patients have already
undergone a correction surgery in early life as per their parents’ consent.
Nevertheless, it is not impossible to find TPT cases in older patients.
Surgical intervention is essential not only to give an acceptable
appearance but also to improve the precision of the hand to perform a
task that requires high precision. In adult TPT patients, removal of
accessory bone followed by arthrodesis is another modality that offers a
favorable outcome.

Fig. 4. Clinical picture of both thumbs at six months post-operative condition. Declaration of informed consent

Written consent for the publication of this case report and its
accompanying images has been acquired from the patient (or their
guardian or next of kin). There is no information in submitted manu­
script that can be used to identify the patient.

Ethical approval

Ethical clearance is not required for this case report, according to our
institution’s research ethics committee. The committee has verified that
the report adheres to standard clinical practices and does not involve
experimental interventions or the need for additional data collection.

Funding

The author(s) received NO financial support for the preparation,

research, authorship, and/or publication of this manuscript.

Author contribution
Fig. 5. X-ray of both thumbs at six months post-operative condition.
Conceptualization: M.M., H.M., B.D.G., R.M.
for six months. The first K-wire has a role as a primary fixator for lon­ Data Curation: M.M., H.M., B.D.G., R.M.
gitudinal alignment, and the second wire functions as an anti-rotation Formal Analysis: M.M., H.M., B.D.G., R.M.
wire. In comparison with Woods’ procedure that has been mentioned Funding Acquisition: Not applicable.
before, this approach is much simpler, and the outcome is favorable for Investigation: M.M., H.M., B.D.G.
the patient, as proven by the SHF and DASH assessments, where both Project Administration: M.M., H.M., B.D.G.
give a good result. Resources: M.M., H.M., B.D.G., R.M.
Nowadays, an exploration of genetic mutation has become popular, Software: Not applicable.
especially in congenital anomalies. Some genetic mutations have been Supervision: M.M., H.M., R.M.
described in the literature [2,11]. It may give a better understanding of Validation: M.M., H.M., B.D.G., R.M.
the pathology of TPT itself and help further research to prevent the Visualization: M.M., H.M., B.D.G., R.M.
development of congenital TPT. In this report, genetic testing is not done Writing-original draft preparation: M.M., H.M., B.D.G., R.M.
because the findings of genetic mutations do not affect the treatment. Writing-review and editing: M.M., H.M., B.D.G., R.M.
TPT in adults is a rare condition since most TPT patients have already
undergone a correction surgery in early life as per their parents’ consent. Guarantor
Nevertheless, it is not impossible to find TPT cases in older patients.
Surgical intervention is essential to improve appearance and the preci­ Meirizal.
sion of the hand to perform a high precision task. In adult TPT patients,
removal of accessory bone followed by arthrodesis is another modality Research registration number
that offers a favorable outcome.
This case report is not a first-in-man case.

3
Meirizal et al.
Conflict of interest statement
The author(s) do NOT have any potential conflicts of interest with
respect to this manuscript.
Acknowledgements
None.
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