Stuttering Priapism and Its Management in a Child Undergoing Craniotomy

A 12-year-old boy was diagnosed with a retrocerebellar dermoid cyst ([ Fig. 1a ]) and reducible atlantoaxial dislocation (AAD; [ Fig. 1b ]) while being investigated for a shoulder droop. He was scheduled for a midline suboccipital craniotomy (MSOC) and excision of the cyst in a prone position. After obtaining informed consent from his parents, the child was brought to the operating room wearing a rigid Philadelphia cervical collar. Due to his fear of needles, inhalational induction was performed using oxygen, nitrous oxide (50%), and sevoflurane, with standard monitors attached. Once intravenous access was established, nitrous oxide and sevoflurane were discontinued. Anesthesia was deepened with propofol (1 mg/kg) and fentanyl (2 µg/kg), followed by paralysis with atracurium (0.5 mg/kg). The trachea was intubated using a 6.0 mm cuffed endotracheal tube, facilitated by a C-MAC videolaryngoscope and manual inline stabilization. Anesthesia was maintained with total intravenous anesthesia utilizing propofol, delivered via a target-controlled infusion pump (Paedfusor model). The effect site concentration was adjusted between 3.5 to 4.0 μg/mL to achieve a bispectral index (BIS) reading of 40 to 50. During urinary catheterization, penile erection (PE) was observed. Initially, it was attributed to a lighter plane of anesthesia; the anesthetic depth was increased by raising the effect-site concentration of propofol from 3.5 to 4.5 μg/mL, and additional analgesia was administered (two bolus doses of fentanyl 0.5 μg/kg each). This led to a reduction in BIS from 50 to 30, but did not result in detumescence (penile relaxation). A rapid literature review on intraoperative causes of priapism indicated that propofol could potentially be a contributing factor. Therefore, propofol was discontinued, and anesthesia was deepened with sevoflurane (0.9–1 minimum alveolar concentration) to achieve a BIS of 40 to 50. Attempts to facilitate detumescence included intravenous glycopyrrolate (0.2 mg), inhaled salbutamol, and ice-cold compression; however, all measures were unsuccessful. Fig. 1 ( A ) MRI-brain (T2-weighted, sagittal section) showing a lesion in the retrocerebellar region, exhibiting mass effect on the cerebellar vermis, mild effacement of the fourth ventricle, along with a focal syrinx at C1 and C3. ( B ) X-ray cervical spine lateral view showing atlantoaxial dislocation and multiple fusion anomalies of the upper cervical spine. MRI, magnetic resonance imaging. An urgent urology consultation was requested, leading to the performance of corporal blood aspiration (5 mL), which was subsequently sent for blood gas analysis. The results indicated a nonischemic type, with the blood gas report showing pH of 7.35, PCO 2 of 44 mm Hg, and PaO 2 of 66 mm Hg. Given the nonischemic findings and the partial detumescence achieved through corporal aspiration, we decided to proceed with surgery after discussing the occurrence of priapism and its potential consequences with the parents. Prolonged priapism carries the risk of corpora cavernosal ischemia, necrosis, and fibrosis, which can ultimately result in erectile dysfunction and penile disfigurement. While updating the parents, they disclosed that the child had recurrent PEs in the past, which were self-limiting and lasted for hours. Hence, we proceeded with MSOC and lesionectomy, avoiding further compression of penis in the prone position. The surgical procedure lasted 3 hours, and complete detumescence was achieved within 4 hours post-surgery. The child made an uneventful recovery. During the postoperative period, the child was investigated for sickle cell disease, hemoglobinopathies, and other hematological malignancies. All the test results were negative. Since the child had nonischemic priapism, we enquired about the past history of perineal trauma or any medication intake that could trigger the event, and all were negative. His postoperative course was uneventful. Priapism is defined as a prolonged PE lasting more than 4 hours without sexual stimulation.[ 1 ] The literature identifies three types of priapism: ischemic, nonischemic, and stuttering priapism (SP). Ischemic priapism (the low-flow type) is prevalent in children and occurs when there is uncontrolled arterial inflow coupled with obstructed venous outflow, leading to engorgement of the corpora. This condition presents a significant challenge during general anesthesia, with an incidence rate of 0.34%.[ 2 ] [ 3 ] In contrast, nonischemic priapism (high-flow type) results from uncontrolled arterial inflow while maintaining venous outflow, often due to trauma to the perineum, and it is rare. SP is an idiopathic, recurrent, ischemic, self-limited episode of PE lasting from a few minutes to 3 hours, often resolves spontaneously, and can occur with or without sexual stimulation.[ 4 ] An imbalance between vasorelaxant (responsible for erection) and vasoconstrictive (responsible for detumescence) processes is considered the primary mechanism for SP.[ 4 ] The etiology of PE is often multifactorial, involving various contributing factors. These can include psychological stimuli (such as sensory stimulation or dream-induced responses), reflex-mediated reactions (like surgical manipulation or instrumentation of the genital area), and drug-induced effects (where anesthetics suppress sympathetic vasoconstriction and alter penile vascular resistance), all of which can lead to vascular engorgement and subsequent erection.[ 3 ] Among anesthetic agents, propofol is specifically noted for its potential to cause priapism; however, the underlying mechanism remains poorly understood.[ 5 ] [ 6 ] Initially, we attributed the priapism to the use of propofol; however, the lack of detumescence even after discontinuing the medication ruled this out. After obtaining a comprehensive history from the parents, the patient was diagnosed with SP. SP can lead to ischemic priapism if not treated promptly. In the field of neurosurgery, reports of intraoperative priapism are predominantly associated with acute spinal cord injuries.[ 7 ] In our case, the child was incidentally diagnosed with reducible AAD and a retrocerebellar cyst while being evaluated for a drooping shoulder; he did not exhibit any neurological symptoms, leading us to exclude the neurological condition as a potential cause of the priapism. The management of intraoperative priapism involves different approaches, including pharmacological methods such as intravenous administration of glycopyrrolate, ketamine, dexmedetomidine, terbutaline, and inhalational salbutamol.[ 4 ] Additionally, increasing the depth of anesthesia with inhalational agents may be employed. Mechanical interventions include perineal compression and cold compression of the penis. Other techniques involve corporal blood aspiration, performing a dorsal penile nerve block, and the intracavernosal injection of pure α agonists like phenylephrine or adrenaline. Various radiological options, and surgical interventions have been described.[ 4 ] In our case, we tried mechanical and pharmacological measures and corporal aspiration, which brought partial detumescence; hence, we proceeded with surgery without the need for the other measures. Anesthesiologists play a crucial role in recognizing this entity, along with its various types and underlying causes, which facilitates early diagnosis. A comprehensive understanding of the available management strategies, both pharmacological and nonpharmacological, is essential to mitigate the morbidity associated with prolonged priapism. Publication History Article published online: 16 July 2025 © 2025. The Author(s). 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