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Case Report Plastic and Aesthetic Research

Isolated sporadic bilateral split hand

malformation: a case report and review of

the literature

3
2
1
Manjit Singh , Sukhminder Jit Singh Bajwa , Harsimarjit Kaur , Ravinder Singh 1
1 Department of Orthopaedics, Gian Sagar Medical College and Hospital, Banur 140601, Punjab, India.
2 Department of Anaesthesiology and Intensive Care, Gian Sagar Medical College and Hospital, Banur 147001, Punjab, India.
3 Department of Anatomy, Govt. Medical College, Patiala 147001, Punjab, India.
Address for correspondence: Dr. Sukhminder Jit Singh Bajwa, Department of Anaesthesiology and Intensive Care, Gian Sagar Medical
College and Hospital, Ram Nagar, Banur 140601, Punjab, India. E-mail: sukhminder_bajwa2001@yahoo.com

ABSTRACT
Typical cleft hand is a rare disorder usually inherited in an autosomal-dominant manner. We report
a case of bilateral typical cleft hand in a 6-year-old male. There was no positive family history of
such presentation, and no systemic manifestations were associated with this entity. Preanesthetic
evaluation revealed no systemic or airway challenges and parents were counseled for routine surgical
and anesthetic risks. The hands underwent successful surgical reconstruction with no peri-operative
complications. To the best of our knowledge, this is the fi rst report of an operated case of an isolated,
bilateral, and sporadic typical split hand malformation.
Key words:
Cleft hand, congenital anomalies, split hand malformation, syndactyly

INTRODUCTION anomaly yielded only two results. [5,6] The reported cases
in the literature described only unilateral afflictions. We
Surgical or medical co-morbidities of anesthetic or airway report a unique case of sporadic, isolated, bilateral SHM
difficulties in patients with congenital anomalies need with syndactyly in both feet, which was successfully
special consideration during corrective management, managed surgically and anesthetically with no untoward
especially in the pediatric population. [1-4] Split hand peri-operative incidents.
malformation (SHM), characterized by median cleft
in the hand, syndactyly, and/or a variable degree of CASE REPORT
underdevelopment of phalanges and metacarpals, is
a rare entity. [5,6] Associated co-morbidities can include A 6-year-old male presented to the Orthopedic Outpatient
sensori-neural hearing loss which can be challenging Department with congenital abnormalities in both the
to identify when eliciting history during preanesthetic hands and feet. He was the second of three children, born
evaluation or during the anesthetic induction as such out of a nonconsanguineous marriage, delivered vaginally
patients can be difficult to counsel. The literary evidence at full term with uneventful antenatal and perinatal course.
indicates the occurrence of multiple anomalies with Chief complaints of the parents pertained to the cosmetic
SHM, but a literature search for isolated cleft hand concerns due to visible deformities. There were no skin
lesions and no associated cleft lip/palate observed during
Access this article online a physical examination. The left hand was dominant, and
Quick Response Code: there was no history of sensorineural deafness. On local
Website: examination, it was observed that there was complete
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obliteration of first web space in the right hand with the
absence of the middle finger in both the hands. The right
DOI: foot showed hypoplasia of the second toe with syndactyly
10.4103/2347-9264.143558 between the third and fourth toes. The left foot had
syndactyly between the third and fourth digits [Figure 1].

114 Plast Aesthet Res || Vol 1 || Issue 3 || Dec 2014