What literature is there to support that people with dihydropyrimidine dehydrogenase (DPD) mutations can be severely harmed when they receive 5-FU?

Comment by InpharmD Researcher

The association between severe 5-FU-associated toxicity (grade 3 or greater) and dihydropyrimidine dehydrogenase (DPD) deficiency caused by DPYD gene mutations has been well documented in the medical literature. Commonly reported adverse events (AEs) include diarrhea, mucositis, hand/foot syndrome, myelosuppression with profound and persistent neutropenia, as well as neurotoxicity. Case-control studies reported up to 88.0% of patients with DPYD*2A mutation experiencing 5-FU-AEs. While recommendations for universal DPYD screening prior to 5-FU treatment remain inconsistent, dose adjustments based on the patient’s response and tolerability to the first treatment are considered necessary.

Background

A 2019 review discussed multiple mutations in the dihydropyrimidine dehydrogenase (DPD) gene, contributing to the 5-fluorouracil (5-FU) toxicity and inadequate dosages. 5-FU as a uracil analog exhibits its wide antitumor activities primarily through disrupting RNA and DNA synthesis. Given DPD is majorly involved in the metabolism of pyrimidines in the human body, it has the same metabolizing effect on 5-FU, a pyrimidine analog. In-vitro data demonstrated DPD catabolizes more than 80% of the 5-FU into dihydro-fluorouracil. As such, patients with DPD deficiency, either from particle loss of enzymatic activity (3-5%) or complete loss of the activity (0.2%), may have an increased risk of severe polyvisceral 5-FU-induced toxicity (grade 3 or higher). [1]

Recent studies found multiple polymorphisms associated with the DPYD gene that encodes DPD enzymes. Additionally, the genetic variants leading to DPD deficiency appeared to vary in different ethnic groups. Specifically, in the Caucasian population, IVS14+1G>A polymorphism linked to the DPYD* 2A allele was found to be the most frequent among the cancer patients, leading to severe 5-FU toxicity. While specific incidences were not reported, clinical manifestations due to the toxicity of 5-FU included fever, mucositis, stomatitis, nausea, vomiting, and diarrhea. In severe cases, it may lead to neurological abnormalities, such as cerebellar ataxia and changes in cognitive functions. Case reports also observed patients have gone into a coma or developed leukopenia, neutropenia, possibly thrombocytopenia, and anemia. In suspicion of 5-FU toxicity due to DPD deficiency, 5-FU should be stopped immediately, followed by removing traces of the drug with hemodialysis and hemoperfusion. Moreover, the subsequent dosage of 5-FU should be adjusted according to the enzymatic status of the DPD observed during the first treatment as well as the patient’s tolerability. In contrast, the high intratumoral activity of DPD may considerably decrease the cytotoxic effect of 5-FU, causing a therapeutic deficiency. Drugs decreasing DPD catabolic activities, known as DPD inhibitors, can be used along with 5-FU to act as a substrate for DPD. [1]

A 2020 consensus paper developing diagnostic and treatment recommendations for 5-FU-associated toxicities reported around 30% of severe toxicity reactions (WHO grade 3-4), particularly diarrhea, mucositis, hand/foot syndrome, myelosuppression with profound and persistent neutropenia, as well as neurotoxicity, can be explained by DPD deficiency in patients receiving 5-FU, capecitabine, and tegafur, leading to a mortality rate of 0.2-1%. Cardiotoxicity, including electrocardiogram changes, coronary artery spasm, and myocardial damage, occurs less often in a range of 1-10% of treated patients. Recommendations, primarily formulated based on European Medicines Agency (EMA) and European entities, suggest the need to test for the 4 most common genetic DPYD variants before treatment with drugs containing FU. Subsequent decisions on FU therapy and dose adjustments should then be guided by genetic testing activity scores and DPD phenotyping. [2]

A 2011 pharmacogenomic review suggests from the literature that there is a major association between DPD mutations and severe 5-FU-related toxicities, although the wide ranges of treatment settings and variants of DPD mutations make it difficult to highlight specific risks. The overall combined variants contribute to approximately 20% of 5-FU adverse events and may be more prevalent in combination 5-FU therapies compared to monotherapy. Consistent findings from case-control studies seem to implicate polymorphisms c.1905+1G>A, c.1679T>G, c.2846A>T as being linked to severe 5-FU related National Cancer Institute Common Terminology Criteria for Adverse Events (NCI CTCAE) grade ≥ 3 toxicities. DPD mutations specific to geographic regions may also define local risks for 5-FU toxicity. There are limited data suggesting that the type of 5-FU-based chemotherapy regimen could also potentiate toxicity with certain DPD variants. [3]

A 2000 study evaluated the clinical implications of DPD deficiency in 37 cancer patients with unexpected severe 5-FU-associated toxicity by analyzing the activity of DPD genes and the clinical presentation of patients suffering from severe toxicity following the administration of 5-FU. A decreased DPD activity was detected in the peripheral blood mononuclear cells of 59% of the enrolled subjects. The study also observed a significantly higher proportion of patients with decreased DPD activity suffered grade IV neutropenia compared with patients with normal DPD activity (55% vs. 13%, p= 0.01). Additionally, in patients with low DPD activity, the onset of toxicity occurred twice as fast compared to patients with normal DPD activity (10.0 ± 7.6 vs. 19.1 ± 15.3 days; p<0.05). Except for grade IV neutropenia, no differences in hematological, gastrointestinal, flu-like symptoms or other toxicities were observed between the two groups. A high incidence of mutations in the PDP gene was discovered in 11 of 14 patients, with the splice site mutation IVS14+1G>A being the most abundant one (43%). The findings of this study demonstrated that 57% (8 of 14) of the patients with a reduced PDP activity have a molecular basis for their deficient phenotype. Based on these data, it was suggested that a partial DPD deficiency appears to play a role in developing 5FU-associated toxicities. Taking the common use of 5-FU in therapy, the severe related toxicities in patients with a low activity DPD, and the high frequency of the IVS14+1G>A mutation in DPD-deficient patients into account, it is preferable to routinely analyze the DPD-activity or screening the IVS14+1G>A mutation prior to the initiation of treatment with 5-FU. [4]

References: [1] Sharma V, Gupta SK, Verma M. Dihydropyrimidine dehydrogenase in the metabolism of the anticancer drugs. Cancer Chemother Pharmacol. 2019;84(6):1157-1166. doi:10.1007/s00280-019-03936-w
[2] Wörmann B, Bokemeyer C, Burmeister T, et al. Dihydropyrimidine Dehydrogenase Testing prior to Treatment with 5-Fluorouracil, Capecitabine, and Tegafur: A Consensus Paper. Oncol Res Treat. 2020;43(11):628-636. doi:10.1159/000510258
[3] Amstutz U, Froehlich TK, Largiadèr CR. Dihydropyrimidine dehydrogenase gene as a major predictor of severe 5-fluorouracil toxicity. Pharmacogenomics. 2011;12(9):1321-1336. doi:10.2217/pgs.11.72
[4] van Kuilenburg AB, Haasjes J, Richel DJ, et al. Clinical implications of dihydropyrimidine dehydrogenase (DPD) deficiency in patients with severe 5-fluorouracil-associated toxicity: identification of new mutations in the DPD gene. Clin Cancer Res. 2000;6(12):4705-4712.
Literature Review

A search of the published medical literature revealed 6 studies investigating the researchable question:

What literature is there to support that people with dihydropyrimidine dehydrogenase (DPD) mutations can be severely harmed when they receive 5-FU?

Level of evidence

B - One high-quality study or multiple studies with limitations  Read more→



Please see Tables 1-6 for your response.


 

The clinical relevance of multiple DPYD polymorphisms on patients candidate for fluoropyrimidine based-chemotherapy. An Italian case-control study

Design

Retrospective, case-control study

N= 366

Objective

To explore the clinical impact of the DPYD polymorphisms and analyze the variants, c.2846A>T and c.1679T>G, in subjects who have presented fluoropyrimidine-related adverse events (FAEs), not DPYD*2A correlated, to confirm their clinical relevance and to evaluate their possible improvement of predicting toxicity when introduced in the pre-treatment screening and to evaluate the potential clinical impact of the c.2194G>A SNP, a more frequent variant of DPYD with an interesting correlation with fluoropyrimidine-related toxicity.

Study Groups

Cases (n= 146)

Control (n= 220)

Inclusion Criteria

Patients candidate for fluoropyrimidine-based chemotherapy, developed severe FAE according to the National Cancer Institute Common Toxicity Criteria, Version 4.0 (CTC-NCI, V4.0) grade 3 or more

Exclusion Criteria

N/A

Methods

Cases are selected based on the occurrence of severe fluoropyrimidine-related adverse events FAEs during treatment. The control group was formed from individuals who did not show fluoropyrimidine intolerance or had very mild AEs (≤ grade 2). The cases and the control matched the most important clinical feature, such as primary tumor location, staging, and patient age. Three selected SNPs of DPYD were analyzed for all subjects, c.1679T>G and c.2846A>T, and c.2194G>A. 

Duration

From 2011 to 2016 

Outcome Measures

The presence of specific DPYD polymorphisms in the cases and controls and determine and the associations between specific DPYD polymorphisms and toxicity

Baseline Characteristics 

 

Cases (n= 146)

Control (n= 220)

       

Age, years

67 65        

Female

54% 43%        

Primary tumor location

Colon

Gastric

Rectum

Pancreas

Anus

Breast

Esophagus

Bile duct

Head and neck

Uterine cervix

Vulvar

 

32%

30%

13%

7%

5%

3%

3%

2%

2%

1%

1%

 

40%

20%

13%

7%

4%

4%

3%

5%

3%

1%

0%

       

Stages 

I-III

IV 

 

60%

40%

 

60%

40%

       

Treatment setting 

Neoadjuvant

Neoadjuvant + Adjuvant 

 Adjuvant 

Metastatic 

 

7%

4%

49%

40%

 

10%

5%

45%

40%

       

Chemotherapy regimen 

FU or Cape ± TT

FU or Cape + OXA ± TT

FU or Cape + IRI ± TT

FU or Cape + OXA + IRI ± TT

FU or Cape + DDP ± TT

FU or Cape + DDP/ OXA + TXT

FU or Cape + mitomycin C

FU or Cape + gemcitabine

FU or Cape ± others

 

10%

50%

3%

4%

11%

8%

5%

6%

3%

 

15%

50%

3%

5%

4.5%

9%

4%

5%

4.5%

       

 FU 5-fluouracil, Cape capecitabine, TT (target therapy) cetuximab or bevacizumab or trastuzumab, OXA oxaliplatin, DDP cisplatin, IRI irinotecan, TXT taxanes (paclitaxel, docetaxel)

Results

Polymorphism

Cases  (n= 146)

Control (n= 220)

p-Value

Odds ratio 95% CI  
DPYD polymorphisms 

18%

34% <0.0001  3.53 1.91 to 6.53  
c.2194G>A mutation 

60% 

40% 0.0041 2.618 1.364 to 5.026  
c.2846A>T mutation 

1.37%

0% 0.0097 17.157 0.714–412.4  
c.1679 T>G mutation 

0.55%

0% 0.1585 7.643 0.184 to 317.5  
Endpoint  All patient  c.2194G/G

c.2194 (G/A)+(A/A)

P-value  Odds ratio 95% CI
All toxicities 48%

44%

74% 0.0001 3.548 1.779 to 7.076
Haematological 25%

21%

52% < 0.0001 4.090 2.161 to 7.741
Neutropenia 25%

21%

50% < 0.0001 3.756 1.986 to 7.106
Intestinal 18%

19%

15% 0.5624 0.817 0.354 to 1.889
Others 8%

8%

9% 0.9531 1.130 0.393 to 3.253

Adverse Events

Common Adverse Events: neutropenia and gastrointestinal disorders.

Study Author Conclusions

The additional DPYD polymorphisms could enhance the prevention of fluoropyrimidine toxicity. c.2194G>A is the most frequent polymorphism and it was found to be associated with neutropenia.

InpharmD Researcher Critique

Patients on various chemotherapy strategies were included with half of the population receiving FU or Cape + OXA ± TT. The proportion of patients who specifically received fluorouracil was not defined and the differences between individual chemotherapies and FAEs were not investigated.



References:
[1] Iachetta F, Bonelli C, Romagnani A, et al. The clinical relevance of multiple DPYD polymorphisms on patients candidate for fluoropyrimidine based-chemotherapy. An Italian case-control study. Br J Cancer. 2019;120(8):834-839. doi:10.1038/s41416-019-0423-8

 

DPYD Variants as Predictors of 5-fluorouracil Toxicity in Adjuvant Colon Cancer Treatment

Design

Case-control study

N= 2594 

Objective

To test the individual associations of adverse events (AE) with grade 3 or greater 5-fluorouracil (5-FU) toxicity from the genotyped DPYD*2A, D949V, and 1560S

Study Groups

Grade ≥3 5-FU without AE (n= 1735)

Grade ≥3 5-FU with AE (n= 859)

Patients with AE data (n= 2594)

Inclusion Criteria

Enrolled in a separate randomized phase III trial to be treated with FOLFOX or FOLFIRI, alone or combined with cetuximab; histologically confirmed adenocarcinoma of the colon stage III; at least 1 pathologically confirmed positive lymph node; absolute neutrophil count ≥ 1500/mm3

Exclusion Criteria

Uncontrolled high blood pressure; unstable angina, New York Heart Association class III or IV heart disease; symptomatic pulmonary fibrosis; allergy to platinum compounds; distant metastatic disease

Methods

Patients with stage III colon cancer enrolled in a separate study were also enrolled in this study to be genotyped for 25 DPYD variants that can decrease DPD enzyme activity. These variations were compared with the rate of AEs in 5-FU treatment.

Duration

Include the duration of the trial as a whole, as well as the duration of the interventions.

Outcome Measures

Primary: rate of at least one grade ≥3 AE related to 5-FU treatment among DPD genotypes

Baseline Characteristics

 

Grade ≥3 5-FU without AE (n= 1735)

Grade ≥3 5-FU with AE (n= 859)

p-Value

Age, years

57.0 62.0 < 0.001

Female

743 466 < 0.001

Race

Missing

White

Black

Asian

Other

 

25

1476

131

89

14

 

8

772

39

32

8

< 0.006

T stage

T1 or T2

T3

T4

 

272

1278

184

 

125

639

95

0.36

N stage

N1

N2

 

1001

734

 

531

328

0.16 

Results

Endpoint

Patients with AE data (n= 2594)

Odds ratio (95% confidence interval [CI])

p-Value

Grade 3 or greater 5-FU AE

DPYD*2A

I560S

D949V

 

22/25 (88.0%)

2/4 (50.0%)

22/27 (81.5%)

 

15.21 (4.54 to 50.96)

N/A

9.10 (3.43 to 24.10)

 

< 0.001

0.48

< 0.001

Adverse Events

DPYD*2A: significant associations include nausea/vomiting and neutropenia

D949V: significant associations include dehydration, diarrhea, leukopenia, neutropenia, and thrombocytopenia

Study Author Conclusions

In the largest study to date, statistically significant associations were found between DPYD variants (DPYD*2A and D949V) and increased incidence of grade 3 or greater 5FU-AEs in patients treated with adjuvant 5-FU-based combination chemotherapy.

InpharmD Researcher Critique

The cohort of patients were limited to stage III colon cancer patients which limits generalizability to other cancer patients. The small number of identified patients with I560S mutations precluded further investigations for those patients.



References:
[1] Lee AM, Shi Q, Pavey E, et al. DPYD variants as predictors of 5-fluorouracil toxicity in adjuvant colon cancer treatment (NCCTG N0147). J Natl Cancer Inst. 2014;106(12):dju298. Published 2014 Nov 7. doi:10.1093/jnci/dju298

 

DPYD*2A Mutation: The Most Common Mutation Associated with DPD Deficiency

Design

Case report

Case presentation

A 75-year-old Caucasian male with metastatic pancreatic adenocarcinoma had a remarkable past medical history, including peripheral vascular disease requiring material bypass surgery of the right lower extremity five years ago. His medications included oxycontin, warfarin, alprazolam, and zolpidem. He also had a history of smoking one pack of cigarettes daily for over 50 years. The initial physical examination was notable for an Eastern Cooperative Oncology Group (ECOG) performance status of 1, right upper quadrant tenderness, and hepatomegaly. The initial laboratory evaluation was within normal limits for a cancer patient. 

He was treated with intravenous (IV) bolus fluorouracil (5-FU) once weekly for three weeks, followed by one week with no therapy. He tolerated the first two treatments without significant toxicity. In the third week, he presented with generalized weakness, altered mental status, ECOG performance status of 3, grade 3 anorexia, pancytopenia (white blood count [WBC] 2.2x10³, hemoglobin 8.6 g/dL, platelets 14x10³ [grade 4]), increased serum creatinine (2.4 mg/dL), and INR 3.4 (grade 3). He was admitted to the oncology unit for the transfusion of red blood cells and platelets with an extensive workup to rule out an underlying infection, disseminated intravascular coagulation, and gastrointestinal (GI) bleeding. Given his presentation with altered mental status and pancytopenia following 5-FU treatment, dihydropyrimidine dehydrogenase (DPD) deficiency was a major diagnostic concern. 5-FU was stopped, and blood was collected to isolate peripheral blood mononuclear cells (PBMC) to determine the activity of DPD, which failed due to an insufficient amount of PBMC. His mental status was not resolved, and he passed away on day four in the hospital. 

The post-mortem examination was remarkable for moderately to poorly differentiated pancreatic adenocarcinoma with extensive metastatic spread to the liver, diaphragm, and regional lymph nodes. GI examination revealed a perforated duodenal ulcer and blood culture positive for Candida albicans. Central nervous system examination revealed lacunar infarction, hypertensive small vessel disease, and remote focal hemorrhage of the basis pontis. Because of pancytopenia, DPD enzyme activity could not be assessed in the patient. Genotype analysis of the DPYD gene revealed the presence of the heterozygous mutation, IVS14+1 G>A, DPYD*2A, now recognized as the most common cause of DPD deficiency. 

Study Author Conclusions

This case demonstrates the importance of understanding the interplay between pharmacology and clinical medicine. In patients with severe DPD enzyme deficiency, even a small dose or very short administration of 5-FU could lead to a marked surge in plasma 5-FU concentrations, leading to increased 5-FU anabolism in susceptible tissue (GI tract, hair, and bone marrow). There are more than 32 sequence variations in the DPYD gene, and only DPYD*2A and DPYD*13 are associated with DPD deficiency. Options for DPD-deficient patients are limited and usually require discontinuation of 5-FU. 

In conclusion, DPD deficiency is a relatively common enzyme disorder to be aware of when considering 5-FU chemotherapy administration. Findings to look for include mucositis, granulocytopenia, and neuropathy. If undiagnosed, DPD deficiency can lead to death. Treatment consists of stopping 5-FU and is otherwise largely supportive. To date, screening patients for the presence of a DPD deficiency before the treatment with 5-FU is not yet routinely performed. Authors suggest that patients should be informed about the risks of the therapy, and DPD testing should be performed upon manifestations of severe untoward toxicity.

References:
[1] Saif MW, Ezzeldin H, Vance K, Sellers S, Diasio RB. DPYD*2A mutation: the most common mutation associated with DPD deficiency. Cancer Chemother Pharmacol. 2007;60(4):503-507. doi:10.1007/s00280-006-0392-5

 

Multiple Organ Failure due to 5-Fluorouracil Chemotherapy in a Patient with a Rare Dihydropyrimidine Dehydrogenase Gene Variant

Design

Case report

Case presentation

A 35-year-old male was diagnosed with advanced caecum cancer that had penetrated the pelvis wall and metastasized to lymph nodes in the abdomen. Later that year, surgery was performed, and over a week later, the patient was started on a chemotherapy regimen of rapid bolus intravenous 950 mg 5-fluorouracil (5-FU) and 45 mg folinic acid. On the first and second day, the patient experienced emesis following administration of the regimen. However, on the third day, he became somnolent and unresponsive to verbal contact. 

The patient was transferred to the ICU, experiencing dyspnea and tachycardia with a heart rate of 120 bpm and blood pressure of 90/60 mmHg. Other abnormal labs included: potassium 6.89 mmol/L, ammonia 689 mcg/dL, creatinine 6.4 mg/dL, urea 275 mg/dL, troponin 0.513 ng/mL with creatinine kinase within the reference range, and platelet count of 826,000/μL. An x-ray showed central venous congestion, and an electrocardiogram showed severe left ventricular insufficiency. In addition, the patient also experienced a seizure and cerebellar ischemia. The patient was given aspirin 100 mg for secondary antithrombotic prophylaxis. With clinical improvements, he was then moved to the general floor. 

A genetic analysis was performed to identify if the patient had an underlying genetic defect of dihydropyrimidine dehydrogenase (DPD). Direct sequencing of all 23 exons of DPYD revealed 3 heterozygotic nucleotide substitutions, which led to amino acid changes: c.85C>T in exon 2 (Cys29Arg), c.1627A>G in exon 13 (Ile543Val), and the rare c.1601G>A (Ser534Asn). Also, two heterozygotic single nucleotide polymorphisms in intron 13 (IVS13+39C>T and IVS13+40G>A) were found. 

Study Author Conclusions

Although the known exon-14 skipping mutation was not shown, there were other mutations found that could be of concern. The first two mutations, c.85C>T in exon 2 (Cys29Arg) and c.1627A>G in exon 13 (Ile543Val), are considered non-pathologic, but data surrounding the rare mutation, c.1601G>A (Ser534Asn), are limited and variable. One previous study has shown that both c.1601G>A substitution and intronic IVS13+40G> have been reported with reduced enzymatic activity.  

Recommendations to screen for the exon 14-skipping mutation before onset of 5-FU chemotherapy were removed because of little association with severe adverse events. 5-FU toxicity can also be caused by rare mutations and have fatal consequences. It is also believed that tolerability of 5-FU can be affected by dosing regimen. In Europe, a weekly 24-hr infusion is preferred over the standard Mayo Clinic regimen used in this case. 

This case study provides evidence of a possible significance between the c.1601G>A mutation and the occurrence of severe 5-FU-related adverse events. Also, it provides evidence to support the clinical value of analyzing the DPYD gene prior to improving drug safety in patients taking 5-FU. 

References:
[1] Lazar A, Mau-Holzmann UA, Kolb H, Reichenmiller HE, Riess O, Schömig E. Multiple organ failure due to 5-fluorouracil chemotherapy in a patient with a rare dihydropyrimidine dehydrogenase gene variant. Onkologie. 2004;27(6):559-562. doi:10.1159/000081338

 

Dihydropyrimidine dehydrogenase deficiency as a cause of fatal 5-Fluorouracil toxicity

Design

Case report  

Case presentation

A 54-year-old male with a past medical history of smoking, hypothyroidism, and newly diagnosed T2N2bM0 (clinically staged) human papillomavirus positive (HPV+) squamous cell carcinoma (SCC) of the left base of tongue with metastases to the cervical lymph nodes enrolled in a clinical trial utilizing dose reduced/deintensified chemoradiotherapy for HPV+ oropharyngeal SCC. Prior to hospitalization, patient achieved complete resolution of his primary tongue tumor as well as a decrease in the size of the cervical lymph node after receiving carboplatin/abraxane induction chemotherapy (cycle 1). Unfortunately, upon receiving inpatient chemoradiotherapy with 5-FU, paclitaxel, and hydroxyurea (part of cycle 1), the patient developed fever in the setting of pancytopenia, followed by intractable diarrhea and vomiting. In suspicion of hydroxyurea-induced pancytopenia, dose of hydroxyurea was reduced and then held. However, patient continued to deteriorate, with subsequently developed septic shock, progressive desquamative skin lesions, acute kidney failure, and sustained pancytopenia. 

Shortly after being transferred to the intensive care unit, patient was transitioned to comfort care and passed away 12 days after inpatient admission. Consent was granted for a complete unrestricted autopsy by the corresponding family member. Autopsy findings revealed toxic epidermal necrolysis, diminished trilineage hematopoiesis in bone marrow, acute renal tubular necrosis, severely damaged gastrointestinal mucosa, and positive blood and bilateral lung cultures for Stenotrophomonas maltophilia. DNA analysis further revealed a point mutation one base downstream of exon 14 (DPYD*2A/c.1905+1G>A) at approximately 100% variant allele frequency. 

Study Author Conclusions

Dihydropyrimidine dehydrogenase deficiency is known to be a leading cause of severe fluoropyrimidine toxicity, culminating in fatality, as was observed in this case. Individuals’ functionality of the DPYD polymorphism may determine the extent of toxicity. Three clinically recognized common non-functional DPYD variants included DPYD*2A/c.1905+1G>A (as seen in this case), *13/c.1679T>G, and rs67376798/c.2846A>T. Homozygous or compound heterozygous nonfunctional alleles can cause severe or even fatal toxicity.

Although not part of the routine clinical practice, prospective screening for DPYD*2A polymorphism leading to genotype-guided dosing has been successful at significantly reducing toxicity, while at the same time being cost-efficient, posing a valid argument of upfront screening which would positively impact both patient safety and treatment efficacy. 

References:
[1] Fidai SS, Sharma AE, Johnson DN, Segal JP, Lastra RR. Dihydropyrimidine dehydrogenase deficiency as a cause of fatal 5-Fluorouracil toxicity. Autops Case Rep. 2018;8(4):e2018049. Published 2018 Nov 30. doi:10.4322/acr.2018.049

 

Unpredicted Severe Toxicity after 5-Fluorouracil Treatment due to Dihydropyrimidine Dehydrogenase Deficiency

Design

Case report 

Case presentation

A 37-year-old female patient underwent curative resection for a stage II (pT3N0M0) poorly differentiated gastric adenocarcinoma, followed by adjuvant chemotherapy consisting of intravenous (IV) 5-Fluorouracil (5-FU) (600 mg/m2 on days 1 and 8) and epirubicin (50 mg/m2 on day 1) in 3-week cycles. As the patient developed erythematous rashes on her hands and oral mucositis on day 5, dose of 5-FU administration on day 8 was skipped. On day 14, the patient experienced fever, stomatitis, diarrhea, leucopenia (0.290×109/L), neutropenia (0.012 ×109/L), and thrombocytopenia (34×109/L). After clinical recovery with granulocyte-colony stimulating factor and broad-spectrum antibiotics, the patient then resumed her treatment with oral doxifluridine on day 35 (200 mg TID, a prodrug of 5-FU), but 3 days later, she developed oral mucositis, and thus further medication was canceled. 

Two weeks after this initial oral doxifluridine treatment, the patient presented at the emergency room due to severe oral pain and diarrhea. Physical exam further revealed grade 3 oral mucositis and erythematous rashes on her hands and feet, and laboratory results showed leucopenia (1.6×109/L), neutropenia (0.421×109/L), and thrombocytopenia (41×109/L). Real-time quantitative polymerase chain reaction (PCR) then demonstrated a dihydropyrimidine dehydrogenase (DPD) mRNA level of 2.5 (control group levels 30-80, cut-off value 3.0), indicating DPD deficiency. Patient eventually improved on 2 weeks of supportive care and was doing well without any evidence of disease recurrence or sequelae of 5-FU toxicity at 1-year follow-up. 

Study Author Conclusions

To date, a number of patients with DPD deficiency have been reported to suffer from severe toxicities after 5-FU treatment. Thus, it was suggested that patients with a DPD activity <70% of that observed in the normal population may be prone to develop severe 5-FU-associated side effects. Additionally, patients with severe DPD deficiency may experience a higher incidence of toxicity compared to those with moderate deficiency. Moreover, a recent population study suggested that 31-34% of patients treated with 5-FU exhibited dose-limiting toxicity, and 40-50% of patients exhibiting grade 3 or 4 toxicity to 5-FU were shown to be partially or profoundly DPD deficient. 

Consequently, when considering the routine use of 5-FU for the treatment of cancer, and even though DPD deficiency is a rare metabolic defect, study authors recommend performing DPD activity analysis or screening for DPD mutations in confined patients who experience unpredicted severe toxicity after initial 5-FU administration.

References:
[1] Baek JH, Kim JG, Kim SN, et al. Unpredicted severe toxicity after 5-fluorouracil treatment due to dihydropyrimidine dehydrogenase deficiency. Korean J Intern Med. 2006;21(1):43-45. doi:10.3904/kjim.2006.21.1.43