Unprecedented bilateral humeral shaft fracture after cesarean section due to epileptic seizure per se

Introduction

Although humerus fractures are frequently seen in adulthood, bilateral humeral fractures without any trauma are very scarce. Fractures of major bones are generally induced by severe external trauma or may take place secondary to severe metabolic disorders [1]. Convulsions may produce considerable muscular tension and sufficient energy capable of fracturing bones [2]. Abrupt and vigorous tonic muscular contractions of convulsive action may give rise to major bone dislocations and fractures. Convulsions induced by various etiologies were reported to precipitate skeletal problems, mostly shoulder region fractures [1].

Here, we present a 32-year-old patient with bilateral fractures of the shaft of the humerus in the early puerperal period. Although she concealed that she had a history of epilepsy before the convulsion; she confessed later that she had no epileptic seizure and had not taken any therapeutic medication for epilepsy for nearly 10 years. Bilateral humeral shaft fractures were unfortunately not diagnosed until the 5^th day subsequent to her epileptic convulsive attack on the day of the cesarean section. Informed consent has been taken from the patient.

Case presentation

A 32-year-old woman – gravida 1, parity 0, abortus 0 – was admitted to our emergency department with strong labour pains. Considering the patient’s last menstrual period, a diagnosis of early rupture of membranes at 39 weeks 5-days gestation was made in the delivery room. The gestational period was also confirmed by the embryo transfer dating as it was an IVF pregnancy. A two centimeter cervical dilatation, 40% cervical effacement, and active amniotic fluid efflux were reported after the bimanual pelvic examination was performed. Ultrasonography performed in the delivery room showed that biometry was compatible with 37 weeks 4-day gestation, the amniotic fluid index was in the normal range and the estimated fetal weight was 3325 g.

No pathological feature was noted regarding her regular follow-up according to the patient’s history and she has had no history of internal diseases including osteoporosis. She delivered a healthy girl weighing 3160 g by cesarean section. During the early post-operative care, a generalized tonic-clonic seizure lasting about 50 s occurred. At this stage, the patient was controlled with proper interventions in order to prevent her from harming herself and the patient’s airway was kept open with the appropriate treatment. After the seizure, she expressed mild bilateral shoulder pain and myalgia which she said that she had never experienced before. She had difficulty in moving her arms and suffered from generalized myalgia localized around both shoulders especially when she was breastfeeding her infant. The convulsion was followed by a post-ictal status that finished spontaneously after 50 s and was noted to have endured for nearly 80 s. The sufferer was in the post-operative care unit during the entire seizure, and no tumble from the bedside or no trauma from the outside was observed.

Subsequent to the convulsion, the patient noted the shoulder ache. The patient was examined by the consultants from the neurology, internal medicine, otorhinolaryngology, obstetrics and gynecology, pulmonary medicine and maternal-fetal medicine departments. No report about the doubt of fracture has been denoted by any of the consultants. NSAIDs as analgesics to mitigate the pain were employed. She was administered intravenous magnesium sulfate immediately because the diagnosis of eclampsia has not been definitely ruled out. She was normotensive and had no subjective symptoms regarding preeclampsia. On the neurology department’s recommendation, a contrast-enhanced cranial tomography was performed and it did not show any sign of hemorrhage or pathology.

She was discharged from the hospital upon her request as she stated that she had no complaint regarding the shoulder pain anymore. However, she was taken to the nearest emergency service on the 5^thpostoperative day after the cesarean section because of the physical inability to move and pain when she tried to hold her infant up which had increased incrementally for 2 days.

On physical exam, both shoulders showed a limitation of movement in hyperabduction without any neurovascular deficit. She was not capable of holding her elbows up because of the shoulder ache and exact range of motion could not be examined properly.

Laboratory results were as follows: hemoglobin 10.8 g/dL (12–18 g/dL), total protein 5.4 g/dL (6.4–8.3 g/dL), albumin 3.0 g/dL (3.5–5.0 g/dL), serum calcium 8.6 mg/dL (8.4–10.2 mg/dL), urea 8 mg/dL (7–20 mg/dL), creatinine 0.62 mg/dL (0.57–1.11 mg/dL), alkaline phosphatase 24 U/L (0–55 U/L), lactate dehydrogenase 420 U/L (125–220 U/L), creatinine kinase 108 U/L (38–204 U/L).

As urgent osseous pathology was suspected as a result of the physical examination, bilateral shoulder anterior-posterior (AP) roentgenograms (Figure 1) were taken and the diagnosis of bilateral humeral shaft fracture was made on the 5^th day after the convulsion. Thereafter; immobilization, bed rest and analgesics were recommended and a shoulder magnetic resonance imaging (MRI) was planned. The shoulder MRI showed glenohumeral dislocation with bilateral displaced multiple proximal humerus fractures. On the 20^th day after the convulsion, she underwent orthopedic surgery bilaterally regarding the proximal humerus fractures under general anesthesia. After the hemiarthroplasty procedure including cementation and tenotomy of the right biceps applied for the right shoulder, biceps tenodesis was performed for the left one. Her appropriate anti-epileptic medical treatment was re-arranged according to the neurology department’s recommendations. No other serious problem occurred after the discharge of the patient.

Figure 1:

Anterior-posterior radiograph of chest including humeral shaft fracture site (fracture site is denoted with the arrow).

Discussion

Bilateral spontaneous shoulder pounding, incorporating bilateral fractures of the proximal humerus are rarely encountered. As we know, many of proximal humerus fractures emerge as a result of the grand mal epileptic convulsions which are electrically provoked or the metabolic disorders like hypocalcemia or uremia [3]. Many underlying variables may contribute to the augmented fracture hazard in the epileptic patients. Strong muscle contractions due to a great energy burst produced by the tetanic convulsions may damage the bone; however, indirect processes may likewise augment the fracture hazard. Osseous diseases are implicated as the most important precipitating risk factors in several types of research in the literature, and fracture incidence is reported to be higher in patients with chronic epilepsy compared to the other chronic diseases [1]. If taken for a long time, the anti-convulsant medication may bring on osteomalacia but in our patient there was no history of osteomalacia and anti-convulsant medication for nearly 10 years.

We report a 32-year-old non-osteoporotic women who had not used any anti-epileptic medication for nearly 10 years. Neufeld et al. reported that nearly 35% of patients with epilepsy experience extra damage in their lifespan as a consequence of convulsive seizures [4]. The significance of secondary damage assessment in patients admitted to the emergency service is affirmed as a result of these findings of this study. Of the patients who experience seizure, fractures are reported to be less frequent complications; but, these typical bilateral fractures have been established to occur with a prevalence of 0.25–2.4% [2]. Epileptic patients suffer a fracture event 33% more in their lifespan than those without epilepsy [2]. Some seizure-precipitated fractures, such as compression fractures of the humerus or the shaft and column of the femur, are represented as the fractures induced by external severe trauma. In the non-traumatic group, the proximal humerus was reported to be the most common and typical area of fractures [2, 5].

If there is no evidence of the patient’s epileptic seizure or the seizure has not been observed, such a fracture may lead us to suspect an assault especially if the patient has no memory of how the fracture occurred [1]. In the literature, there are few cases of acute humeral fractures due to the convulsive seizures. Seizure-associated fractures that commonly involve the proximal humerus may occasionally represent diagnostic handicaps; however, they are generally distinguished because of the severe pain or disability and convulsion history [1]. Physicians ought to be aware of the contingency of seeing fractures in the epileptic women after convulsions. The requirement for the emergent radiographic survey should be considered when there is severe pain or immobility in any section of the body. In order to prevent unrecognized damage after convulsive events, a complete assessment must be implemented prior to the diagnosis of ligament strain or muscle wrench as the etiology of the injury. Diagnosis of the damage may be postponed or caught serendipitously on the radiographic imaging for unrelated reasons [3]. Simultaneous bilateral proximal humerus fractures are scarce, but should be diagnosed and handled as soon as possible as they have a potential association with immense morbidity.

In conclusion, in order to recognize the fractures as a result of the convulsions one should use radiographs. In addition, for any epileptic patient suffering from shoulder pain after a convulsion, an AP shoulder radiograph should be acquired. Acute and spontaneous bilateral fractures of the humerus without any trauma are not such rarities, so a high degree of suspicion should be kept in mind while treating a patient in the post-ictal phase; as, if treated promptly, treatment leads to complete functional recovery. Nevertheless, a further radiological evaluation must always be sought to rule out pre-existing pathology for further treatment planning.