Working Diagnosis:
1. Sequela of anterior shoulder dislocation:
Anterior-inferior labral tear
Posterior labral tear
Hill-Sachs lesion
2. Possible shoulder instability secondary to absence of the long head of the biceps tendon
Treatment:
25 days post-injury, patient underwent:
Arthroscopic removal of chondral loose body and chondroplasty of humeral head
Anterior labral repair/capsulorrhaphy
Posterior labral repair/capsulorrhaphy
Required total of 7 sutures
Arthroscopic confirmation of absent long head of biceps tendon
Outcome:
The patient did well following surgery. He did extensive home and office-guided physical therapy for 3 months, at which time he was released to a slow, gradual return to play protocol for baseball season. At 6 months post-op, patient had started pitching on his high school baseball team with the affected shoulder and overall was doing very well. The patient and his mother state that he was doing so well, it was almost as if his injury and repair had never occurred. At the time of this case submission, patient had yet to determine if he would be returning to football.
Author's Comments:
This patient has a few interesting points for discussion.
The patient's mother was astute enough to remember that she had taken divalproex sodium with high doses of folic acid during her pregnancy with him. Divalproex sodium, valproic acid, and sodium valproate are all valproate products. These are FDA approved for use in seizure disorders, bipolar disorder, and migraine prophylaxis. In utero exposure to these medications can cause fetal valproate syndrome (FVS). Broadly, FVS can be characterized by neural tube defects, cleft lip and palate,, cardiovascular abnormalities genitourinary defects, developmental delay, endocrine disorders, limb defects, and autism (Ornoy, 2009).
The patient never had a formal diagnosis for his thumb malformation, but radial ray anomaly is fitting. Radial ray anomalies generally referred to a spectrum of congenital anomalies that involve the radius, radial carpal bones, or thumb. It can be seen in Holt-Oram syndrome, some trisomies, diabetic embryopathy, Fanconi anemia, VACTER-L syndrome, and teratogenic drug exposure (Olson, 2022). A figure from a study by Sevilla-Montoya et al. (2017) displayed a patient with Holt-Oram syndrome who had very similar hand morphology as our patient Case Photo #9 . Despite the more specific definition of the patient's thumb anomaly, a PubMed literature search did not correlate absent long head of biceps tendon with either radial ray anomalies or fetal valproate syndrome.
This also brings up the important point of medication review and management in women who were pregnant, trying to conceive, or of childbearing age and not on birth control. FVS from valproate drug is only one example of the effects of teratogenic drugs in utero. Medication review is a basic exercise that should be done at the time the patient meets with the first provider who diagnosis pregnancy, as teratogens usually have the greatest impact on the fetus during the first few months of development. Of note, the mother was still on divalproex sodium at the beginning of her next pregnancy. She was, however, seeing a different obstetric provider who took her off the divalproex immediately. Despite this, the baby was stillborn with a sacral dimple, which was benign when evaluated with ultrasound.
The other point of interest is the absent long head of the biceps tendon. It caused these authors to pose the questions: How much does the long head of the biceps tendon contribute to the stability of the shoulder and would he have avoided his shoulder injury had the tendon been present? A literature review by Kumar et al. (2017) reviewed 35 cases of absent long head of biceps tendon after applying exclusion criteria. They ultimately concluded that there were not enough cases to state whether or not the congenital finding contributed to shoulder pain, impingement, or instability. They also mention that it could just be an incidental finding. One point of interest in the review was that 15 of 25 cases where MRI results were available had actually missed the absent long head of the biceps tendon. This was found arthroscopically and confirmed retroactively on MRI. Given these numbers, finding asymptomatic patients with this incidental finding and setting up a study to look at signs, symptoms, stability, and biomechanics of the shoulders would be a challenge. A more feasible way to attempt to find more patients with this incidental finding would be to use musculoskeletal ultrasound, as the biceps tendon is typically an easy structure to visualize under ultrasound.
Editor's Comments:
The glenohumeral joint is stabilized by both dynamic and static structures. The dynamic structures include the rotator cuff, long head of biceps brachii, and deltoid. The static structures include the glenohumeral joint capsule, labrum, glenohumeral ligaments, and bony anatomy. The decision for operative vs non operative management of shoulder dislocations is based on associated pathology and risk for re-dislocation. Males under the age of 20 who play contact sports and have greater than 25% bone loss are at highest risk for re-dislocation.
References:
1. Ornoy, A. Valproic acid in pregnancy: how much are we endargering the embryo and fetus? Reproductive Toxicology. 2009;28(1):1-10.
2. Olson et al. Radial Dysplasia. Stat Pearls. 2022.
3. Sevilla-Montova et al. Heterogeneous diagnoses underlying radial ray anomalies. The Indian Journal of Pediatrics. 2017; 84: 2000-2005.
4. Kumar et al. Congential absence of the long head of biceps tendon & its clinica implications: a systemic review of the literature. Muscles, Ligaments, and Tendons Journal. 2017; 7(3): 562-569.
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