Anaphylaxis due to cashew nut allergy associated with acute pancreatitis without abdominal pain: a case report

Abstract

Objective

Although gastrointestinal symptoms are common immediate reactions to food allergies (FA), the relationship between acute pancreatitis (AP) and FA remains unclear.

Patient

A 3-year-old boy developed vomiting, dyspnea, and urticaria 30 min after eating a cashew nut (CN) and was diagnosed with anaphylaxis.

Results

The patient was diagnosed with AP based on markedly elevated serum pancreatic amylase activity (1,847 IU/L) and mild pancreatic enlargement on abdominal ultrasonography, despite the absence of abdominal pain. All symptoms resolved immediately after intramuscular adrenaline injection, and serum pancreatic amylase activity normalized after 3 days. CN allergy was diagnosed based on blood tests showing elevated serum levels of CN- and Ana o 3-specific immunoglobulin E.

Conclusions

This is the first reported case of AP associated with anaphylaxis due to a CN allergy. AP should be considered in patients with FA who present with gastrointestinal symptoms, even if vomiting is the only symptom and abdominal pain is absent. AP associated with anaphylaxis may resolve rapidly with appropriate treatment.

Introduction

Food allergy (FA) presents with several immediate symptoms, including gastrointestinal symptoms, and is reported in 18.6% of Japanese cases with FA¹⁾. However, the association between acute pancreatitis (AP) and FA remains unclear, and no reports of AP complicated by a cashew nut (CN) allergy exist. Here, we report a rare case of CN-induced anaphylaxis complicated by AP in a pediatric patient. The parents provided informed consent for the publication of this study and accompanying data.

Case Presentation

A 3-year-old boy with no specific medical history presented to the emergency department (day 1) with dyspnea, multiple episodes of vomiting, and urticaria 30 min after eating a piece of CN. The patient had no abdominal pain. When he came to the hospital, >4 h had passed since the onset of symptoms. His oxygen saturation level was 97% on ambient air; however, he reported mild dyspnea, nausea, and persistent generalized urticaria. He was diagnosed with anaphylaxis, presenting with moderate respiratory, gastrointestinal, and cutaneous symptoms¹⁾, and received an intramuscular injection of adrenaline, followed by intravenous prednisolone and diphenhydramine. His symptoms resolved immediately after an adrenaline injection, which was administered 5 h after onset. Blood tests demonstrated elevated activities of pancreatic enzymes (total amylase, 2,156 IU/L; pancreatic amylase, 1,847 IU/L), and abdominal ultrasonography (Figure 1) revealed mild pancreatic enlargement (pancreatic head diameter, 23 mm; pancreatic tail diameter, 22 mm)^{2, 3)}. Based on these findings, the patient was diagnosed with complicated AP^{4, 5)}. The diagnosis of CN allergy was based on the ImmunoCap^® assay results demonstrating that his blood serum levels of CN- and Ana o 3-specific immunoglobulin E were 61.7 UA/mL and 76.7 UA/mL, respectively. Ana o 3 is one of the allergen in CN, and sensitization to it is suggested to be a high-risk factor for severe anaphylaxis⁶⁾. He was hospitalized and provided with fasting instructions and intravenous fluids for half a day. He was discharged the next day (day 2) with no recurrence of skin symptoms or vomiting, and could eat without any abdominal pain. His serum total amylase and pancreatic amylase activities normalized on day 4 (65 and 17 IU/L, respectively). One month after discharge, the patient underwent magnetic resonance imaging/magnetic resonance cholangiopancreatography, and the imaging findings were normal, with no morphological abnormalities, including pancreaticobiliary maljunction.

Figure 1.

Abdominal ultrasound performed on the day of admission revealed mild pancreatic enlargement (pancreatic head diameter: 23 mm, pancreatic tail diameter: 22 mm). The upper normal limits for the diameter of the pancreatic head and tail in children under 7 years of age are 19 mm and 16 mm, respectively²⁾.

Discussion

To the best of our knowledge, cases of AP complicated by milk, egg, buckwheat, peanut, kiwi, cod, banana, and other FAs have been reported (Table 1)^{7,8,9,10,11,12,13,14,15,16,17)}; however, no cases of CN allergy complicated by AP have been reported.

Table 1. Previously reported cases of food allergy complicated by acute pancreatitis.

Reference	Subject number	Age (years)	Food	GI symptoms	FA symptoms other than GI symptoms	Time between ingestion of food and onset	AMY (IU/L)	LIP (IU/L)	US findings	CT findings	Time between AMY normalization and onset	Time between GI symptom resolution and onset
Suzuki7)		28	Milk	Epigastric pain and vomiting	None	Immediately	113 (1 week after onset)	202 (1 week after onset)	Normal	Edematous intestinal wall	2 weeks	2 weeks
Inada8)		1	Milk	Vomiting	Urticaria	1 hour	427	4,840	Normal	Normal	36 hours	12 hours
Lorenzo9)		23	Milk	Abdominal pain, vomiting, and diarrhea	Facial erythema and generalized pruritus	10–15 minutes	2,061	921	Enlarged pancreas	NA	NA	36 hours
Tse10)		25	Egg	Abdominal pain, vomiting, and diarrhea	None	Within 60 minutes	NA	2,400	Normal	Normal	NA	>1 day
Ogura11)	1	3	Egg	Abdominal pain and vomiting	Urticaria	45 minutes	1,374 (P-AMY)	776	NA	Swollen pancreas	4 days	3 days
Ogura11)	2	9	Buckwheat	Abdominal pain and vomiting	Laryngeal pain, cough, urticaria, and hypotension	160 minutes	667 (P-AMY)	465	NA	Swollen pancreas	2 days	2 days
Sasaki12)		12	Peanut	Abdominal pain	Throat discomfort	Immediately	632	451	Normal	Normal	1 day	NA
Gastaminza13)		48	Kiwi	Epigastric pain, vomiting, and diarrhea	Facial erythema	15 minutes	835	NA	NA	Normal	48 hours	24 hours
Pellegrino14)		8	Codfish	Abdominal pain	Rhinitis, asthma, stridor, urticaria, and pallor	5 minutes	2,559	NA	Swollen pancreas	NA	72 hours	NA
Iwata15)		7	Cod roe	Abdominal pain	Facial erythema, wheezing, pruritus, and respiratory distress	3 hours	2,410	2,080	Enlarged pancreas	NA	Within 4 days	Within a few hours
Inamura16)		47	Banana	Epigastric pain, vomiting, and diarrhea	None	NA	945	1,893	Normal	Normal	2 days	24 hours
Matteo17)	1	40	Milk, beef	Abdominal pain	Migraine and pruritus	NA	2,038	NA	Normal	Normal	NA	3 hours
Matteo17)	2	17	Beef, egg, fish, potato	Abdominal pain	Headache, myalgia, and fever	NA	2,150	NA	Enlarged pancreas	Enlarged pancreas	NA	2 hours
Toki (present case)		3	Cashew nut	Vomiting	Dyspnea and urticaria	30 minutes	2,156	NA	Enlarged pancreas	NA	3 days	5 hours

FA: food allergy; GI: gastrointestinal; AMY: serum amylase concentration; LIP: serum lipase concentration; US: ultrasonography; CT: computed tomography; NA: not available; P-AMY: serum pancreatic amylase concentration.

FA is not a common cause of AP^{4, 5)}. Ogura et al. reported that two of 2,598 patients with FA with abdominal symptoms on allergen oral challenge testing developed AP, both of whom were under 10 years of age¹¹⁾. However, AP itself is rare in children, with an estimated prevalence of 1/10,000 per year according to a US survey study⁴⁾. Therefore, FA may be a contributing factor to AP in children. Moreover, gastrointestinal symptoms, including anaphylaxis due to CN, are relatively common^18,19,20,21). Furthermore, the number of patients with CN allergies has been increasing^{19, 22)}. These factors suggest that the number of patients with CN-induced AP may increase in the future. To test this hypothesis, it is necessary to investigate the epidemiology of FA, with a focus on the complications of AP, especially for patients with FA suspected to have several gastrointestinal symptoms, such as CN allergy.

CN is known for its antioxidant and anti-inflammatory effects, and has been demonstrated to have potential therapeutic applications for AP in animal studies²³⁾. However, CN allergy frequently results in anaphylaxis²⁴⁾, and AP may develop as a symptom of CN allergy. Therefore, allergies to CN should be considered when using CN to treat AP in humans.

The only gastrointestinal symptom in our case was vomiting without abdominal pain. However, 80–95% of pediatric patients with AP experience abdominal pain⁴⁾. Therefore, the presence of gastrointestinal symptoms, even if just vomiting, should raise the suspicion of AP complications.

However, the natural history of FA-associated AP remains unclear. Some cases in which gastrointestinal symptoms disappeared within a few hours have been reported (Table 1). In the present case, gastrointestinal symptoms disappeared within 5 h of onset. Although non-specific symptoms such as abdominal pain and vomiting are insufficient to distinguish between occasional AP and FA-associated AP, considering the reported average hospitalization duration of 19.5 days for infants with AP⁴⁾, AP that develops shortly after food intake and presents with gastrointestinal symptoms within a few days may be more likely to be caused by FA.

Some cases have been clinically diagnosed with AP based on gastrointestinal symptoms and elevated pancreatic enzyme activities without evidence of pancreatic enlargement, possibly due to pancreatic fluid congestion associated with mucosal edema near Vater’s papillae causing AP, as suggested by Inamura et al¹⁶⁾. Moreover, Suzuki et al. reported a case of thickening of the intestinal wall on computed tomography and mentioned the possibility that obstruction of the pancreatic or biliary duct due to thickening of the gastroduodenal wall with eosinophilic infiltration may trigger AP⁷⁾. Rather than direct inflammation of the pancreatic parenchyma, pancreatic fluid retention due to eosinophilic thickening of the duodenal mucosa, including Vater’s papilla, is a more acceptable pathogenesis in cases of AP associated with FA, considering the rapid resolution of digestive symptoms. However, the presence of gastroduodenal wall thickening was not confirmed in this case, leaving the cause of AP unknown, which is a limitation of this study.

Conclusions

AP should be considered in patients with FA and gastrointestinal symptoms, even in the presence of vomiting as the only symptom. However, AP associated with FA may be partly due to pancreatic fluid retention associated with intestinal mucosal edema and may resolve within a few days.

Conflict of interest

The authors declare no conflicts of interest.

Funding

No funding was received for this study.

Ethics approval and consent to participate

The parents provided informed consent for the publication of this study and accompanying data.

Consent for publication

All the authors provided consent for the publication of this study.

Author contributions

ST and RS designed the study and prepared the manuscript. MS and AM revised the manuscript. All the authors have read and approved the final version of this manuscript.

Data availability statement

The data supporting the findings of this study are available from the corresponding author upon reasonable request.