Abductor pollicis longus (APL) muscle is known to exhibit different variations with respect to its attachments. Various studies have reported the splitting of the APL muscle. Comparative anatomical findings of split insertion of APL is commonly found in chimpanzees, gorillas and gibbons. In the present study, we describe an anomalous APL muscle, which originated from the posterior surface of the shaft of the radius and ulna and traversed a course deep to the extensor retinaculum. Interestingly, immediately after emerging form the deeper aspect of extensor retinaculum, the thin tendon of the APL muscle continued again as a muscular belly in relation to the dorsolateral part of the 1st metacarpal bone, to end as a tendon with its attachment to the base of the proximal phalanx. Such an unusual variation of APL with its attachment into proximal phalanx is a rare finding and may be of importance in altering the mechanics of the thumb during abduction. The clinical significance of such an anatomical variation of APL may be important during reconstructive surgeries involving thumb and also of academic interest.