5 Interesting Medication Case Reports You Should Know

This article is part 1 of a 6-part series on interesting and unusual medication-related case reports.

Case reports are defined as the scientific documentation of an individual patient. These reports are often written to document an unusual clinical presentation, treatment approach, side effect, or response to treatment. Within the report can be found an array of medical information including patient signs and symptoms, diagnosis, treatment, and follow-up. Most experts see case reports as the first line of evidence in healthcare, which can sometimes lead to future higher-level studies.

Case reports can be a great learning opportunity for both pharmacists and pharmacy students to understand a case progression and the unconventional response and effects of medications.

This article highlights 5 published case reports that document medications treating conditions for which they are not FDA-approved:

1. Risperidone reversal of bed-wetting.¹

Risperidone is an atypical antipsychotic indicated for the treatment of schizophrenia, bipolar disorder, and irritability associated with autistic disorder. Some reports in the literature have shown risperidone to induce enuresis (bed-wetting) as a side effect, while others have shown it to lead to transient urinary incontinence.

A 2010 case report tells the story of a 12-year-old girl who was on the spectrum and nocturnal enuresis that was almost nightly in frequency. During the 3 years prior to presentation, she exhibited a symptom of inappropriate smiling; no other mood or behavioral symptoms were observed. The patient was subsequently started on a trial of risperidone 0.5 mg nightly for the inappropriate smiling.

On day 10 of therapy, the parents noted that there had only been one instance of bed-wetting and by day 20, the enuresis was in complete remission and the inappropriate smiling also stopped. During a 10-day lapse in medication adherence, both symptoms returned. When the risperidone was subsequently restarted, both symptoms resolved through a 3-month follow-up.

As risperidone does not have any anticholinergic or pro-adrenergic effects, the authors were unable to explain its efficacy in treating this patient’s enuresis. They speculate the medication may have had an impact on non-rapid eye movement sleep during which enuretic episodes may have occurred.

2. Treatment of autism with low-dose phenytoin.²

Autism is a developmental disorder characterized by impaired social function and communication. To date, only 2 medications, risperidone and aripiprazole, have been FDA-approved for the treatment of autism, and these are specifically indicated for the associated behavioral disturbance symptoms.

In 2015, a case report was published looking at the effect of the anti-epileptic phenytoin (Dilantin) to enhance social functioning in a patient with autism spectrum disorder. The patient was a 19-year-old man with a childhood diagnosis of autism and comorbid attention deficit hyperactivity disorder (ADHD). He suffered from persistent disruptive and insensitive behavioral, difficulties in establishing and maintaining lasting relationships, social isolation, and intolerance to noise, lights, and crowds.

Following the addition of a very low 2 mg sublingual dose of phenytoin, the patient experienced improvements in multiple areas social functioning. Specifically, for the first time, he was able to sustain eye contact, demonstrate more spontaneous facial expression, limit distractions, and use gestures during conversation. He continued to take one compounded 2 mg phenytoin capsule daily with improvements in behavior and no aggressive outbursts. During a 4-week period when he stopped taking the medication, his symptoms reverted. He was subsequently reinitiated on phenytoin increased to a 5 mg dose, and had symptom reduction over an 18 month follow-up period.

According to the author, the pharmacological action of antiepileptic medication for the treatment of autism is not completely surprising because epilepsy and autism are thought to possibly have a common neurodevelopment origin. The exact link between the 2 has yet to be determined.

3. Trigeminal neuralgia relieved with oral sumatriptan.³

Trigeminal neuralgia is a chronic pain condition involving pain in the lower face and jaw. It is estimated that approximately 150,000 people are diagnosed every year with the condition. Treatment options include surgery and medications, including carbamazepine, baclofen, phenytoin, gabapentin, and/or opioids. In the literature, trigeminal neuralgia has been described as the most excruciating pain known to man.

A 2009 case report was published after a treatment-resistant woman with trigeminal neuralgia received sumatriptan, a serotonin agonist FDA-approved for migraine headaches. The case details a 51-year old woman with a 4-year history of recurrent, right-sided facial pain, in all 3 branches of the trigeminal nerve. The episodes were reported to occur on average 2 to 3 times per week. The patient cycled through a number of treatment options without adequate relief of symptoms, including gabapentin, carbamazepine, dothiepin, pregalabin, numesulide, oxycodone, decompression of the right trigimeal nerve, and radiofrequency ablation.

The patient was subsequently given a trial of oral sumatriptan 50 mg, to be taken at the onset of facial pain, along with a maintenance dose of propranolol 160 mg daily. The patient rapidly responded to therapy, with a reduction in the frequency of her facial pain from 2 to 3 episodes per week to 1 every 2 weeks.

While the mechanism for sumatriptan’s effect in this patient is not fully understood, it is thought the medication’s effects on constricting blood vessels in the brain may have relieved whatever compressive effects were occurring at the root of the affected trigeminal nerve. The authors state that this case highlights a possible treatment option for trigeminal neuralgia resistant to standard treatment options, but acknowledge that further research is required to validate their findings.

4. Duloxetine for treatment-resistant tennis elbow.⁴

Tennis elbow is a common musculoskeletal disorder characterized by pain in the elbow due to overuse. Management includes pharmacological options (NSAIDs, steroid injections), nonpharmacological options (icing the elbow, elbow support, physical therapy), and surgery (particularly for resistant cases).

In 2008, researchers published a case report of 2 patients who did not respond to medical and surgical treatment interventions and were then treated with duloxetine. Both patients were healthy women, aged 32 and 27 years old. The pair presented with severe pain due to tennis elbow of about 18 months in duration. They continued to experience these painful symptoms despite treatment with analgesics, local steroid injections, physiotherapy, cryotherapy, ultrasound, and surgical release.

Both patients were prescribed duloxetine 60 mg per day. After 4 to 6 weeks, they reported substantial improvement in pain symptoms, and during a 6-month follow-up they were completely pain free.

Duloxetine is a serotonin and norepinephrine reuptake inhibitor FDA-approved for the treatment of major depressive disorder along with fibromyalgia, chronic musculoskeletal pain, and diabetic peripheral neuropathy. The mechanism behind treating pain due to tennis elbow is thought to be similar to its role in treating chronic musculoskeletal pain and fibromyalgia; however, researchers were unable to explain the maintained state of recovery in these 2 patients.

5. Naltrexone to treat sexual fetishism.^5,6

Fetishism is a sexual disorder characterized by intense sexually arousing fantasies, sexual urges, or behaviors involving nonliving objects over a period of at least 6 months. The fantasies or behaviors, which appear almost exclusively in males, often result in clinically significant distress or impairment in social functioning. Examples of fetishes include feet, hair, clothing, rubber items, and bodily fluids.

This 2014 case report tells the story of a 40-year-old married, heterosexual male who reported intense urges to roam around during night hours to obtain undergarments of women for sexual gratification since 25 years old. He was caught stealing undergarments multiple times by neighbors and the local police. He reported numerous unsuccessful attempts to control this behavior. Additionally, he suffered from comorbid alcohol and cannabis dependence.

After a failed trial of fluoxetine, the patient was started on oral naltrexone 50 mg per day to decrease cravings for alcohol dependence. He continued the medication for 11 months and reported no urge to carry out fetish behavior, and additionally had been abstinent from alcohol and cannabis.

Within the literature, naltrexone has been shown to successfully treat a number of disorders such as alcoholism, paraphilias, and gambling. Additionally, in a case report published the following year, naltrexone was found to be effective in an individual suffering from compulsive pornography use. The likely mechanism of naltrexone for these conditions is through a reduction in dopamine release.

References:

• Mendhekar DN, Andrade C. Risperidone-related reversal of primary enuresis: an unusual case report. World J Biol Psychiatry. 2010;11(2 Pt 2):514-515.
• Bird P. The treatment of autism with low-dose phenytoin: a case report. J Med Case Reports. 2015;9:8.
• Moran JA, Neligan A. Treatment resistant trigeminal neuralgia relieved with oral sumatriptan: a case report. J Med Case Reports. 2009;3:7229.
• Wani ZA, Dhar SA, Butt MF, et al. Duloxetine in treatment of refractory chronic tennis elbow: Two case reports. J Med Case Reports. 2008; 2:305.
• Firoz K, Nidheesh Sankar V, Rajmohan V, et al. Treatment of fetishism with naltrexone: a case report. Asian J Psychiatr. 2014;8:67-68.
• Kraus SW, Meshberg-Cohen S, Martino S, et al. Treatment of Compulsive Pornography Use With Naltrexone: A Case Report. Am J Psychiatry. 2015;172(12):1260-1261.