Images in Allergy
Is It Food Allergy or Frey Syndrome? Betul Buyuktiryaki, MD, and Bulent Enis Sekerel, MD Ankara, Turkey
A 6-month-old boy was referred with suspicion of a food allergy. The patient had been born at term via spontaneous vaginal delivery without the use of forceps or vacuum assistance. He had a history of recurrent unilateral erythematous eruption on his face after feeding. The parents first recognized the symptoms at 5 months of age after introducing solid foods to his diet. The symptoms were triggered by yoghurt, carrots, and fruit (apples, pears, oranges) a few minutes after mastication and would resolve spontaneously within 15 to 30 minutes. The rash always appeared in the same area without pruritus, swelling, respiratory, or gastrointestinal symptoms. Although these foods were restricted in the diet by the family physician, the child experienced the same manifestation with each newly added solid food after chewing. On his presentation, clinical examination was normal; skin tests with a commercial battery of foods and also prick-to-prick tests and specific IgE levels with Uni-Cap (Phadia, Uppsala, Sweden) for incriminated foods were negative. An open challenge test with yoghurt and pear on different occasions resulted in flushing within a few minutes, which extended from the right cheek across the preauricular and temporal region to the forehead scalp with no sweating (Figure 1), and disappeared over 20 minutes. The child received a diagnosis of Frey syndrome, which is commonly misinterpreted as a food allergy. In his follow-ups, a decrease in the frequency of the symptoms and the extent along with the intensity of the erythema was observed. Interestingly, flushing occurred on his left cheek after eating cucumber at 8 months of age. At the age of 12 months, flushing still appeared localized to the right cheek with lemon, spicy foods, cucumber, and butter, and was sometimes also seen on the left cheek (Figure 2, A and B). Frey syndrome, otherwise named auriculotemporal nerve (ATN) syndrome, is characterized by unilateral (rarely bilateral) recurrent episodes of flushing and sweating over the parotid area after gustatory stimulus. The syndrome most often occurs in adults after parotid surgery and very uncommon in children. Because the disease often exhibits spontaneous resolution within a few years in children, no treatment is recommended.
Department of Pediatric Allergy, Hacettepe University Faculty of Medicine, Sihhiye, 06100, Ankara, Turkey No funding was received for this work. Conflicts of interest: The authors declare that they have no relevant conflicts. Received for publication August 13, 2014; revised November 13, 2014; accepted for publication November 14, 2014. Available online -Corresponding author: Bulent Enis Sekerel, MD, Professor in Pediatrics, Pediatric Allergy and Asthma Unit, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey. E-mail: [email protected]. 2213-2198 Ó 2015 American Academy of Allergy, Asthma & Immunology http://dx.doi.org/10.1016/j.jaip.2014.11.010
FIGURE 1. Unilateral flushing appearing on the face after an open challenge test with pear at 6 months of age.
It has been proposed that a perinatal birth trauma during delivery may be a possible reason for damage to the auriculotemporal nerve.1 Moreover, congenital cases without any known trauma have also been reported, but only a few.2 Postulated mechanisms for the congenital cases include a congenital aberrant pathway, crossover of the fibers caused by loss of insulation around the neural sheaths, neural irritation due to scar tissue formation, subclinical intrauterine infection, and mild injury to the parotid gland.3 Although the majority of patients have flushing in the preauricular region compatible with the cutaneous distribution of ATN, the reaction of our patient expanded from the cheeks to the middle of the forehead. Additionally, he had no history of trauma and had bilateral symptoms, strengthening the assumption of a central nervous system abnormality or immaturation. We suppose that keeping in mind this rare disease induced by tactile or masticatory stimuli would help physicians to differentiate it from food allergy and obviate unnecessary diagnostic tests and elimination diets. REFERENCES 1. Caulley L, Hong P. Pediatric auriculotemporal nerve (Frey) syndrome. CMAJ 2013;185:504. 2. Beck SA, Burks AW, Woody RC. Auriculotemporal syndrome seen clinically as food allergy. Pediatrics 1989;83:601-3. 3. Moreno-Arias GA, Grimalt R, Llusa M, Cadavid J, Otal C, Ferrando J. Frey’s syndrome. J Pediatr 2001;138:294.
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BUYUKTIRYAKI AND SEKEREL
J ALLERGY CLIN IMMUNOL PRACT MONTH 2015
FIGURE 2. A, Flushing on the left cheek after an open challenge test with cucumber at 12 months of age. B, Flushing on the right cheek after an open challenge test with lemon at 12 months of age.
Is It Food Allergy or Frey Syndrome? Betul Buyuktiryaki, MD, and Bulent Enis Sekerel, MD Ankara, Turkey
A 6-month-old boy was referred with suspicion of a food allergy. The patient had been born at term via spontaneous vaginal delivery without the use of forceps or vacuum assistance. He had a history of recurrent unilateral erythematous eruption on his face after feeding. The parents first recognized the symptoms at 5 months of age after introducing solid foods to his diet. The symptoms were triggered by yoghurt, carrots, and fruit (apples, pears, oranges) a few minutes after mastication and would resolve spontaneously within 15 to 30 minutes. The rash always appeared in the same area without pruritus, swelling, respiratory, or gastrointestinal symptoms. Although these foods were restricted in the diet by the family physician, the child experienced the same manifestation with each newly added solid food after chewing. On his presentation, clinical examination was normal; skin tests with a commercial battery of foods and also prick-to-prick tests and specific IgE levels with Uni-Cap (Phadia, Uppsala, Sweden) for incriminated foods were negative. An open challenge test with yoghurt and pear on different occasions resulted in flushing within a few minutes, which extended from the right cheek across the preauricular and temporal region to the forehead scalp with no sweating (Figure 1), and disappeared over 20 minutes. The child received a diagnosis of Frey syndrome, which is commonly misinterpreted as a food allergy. In his follow-ups, a decrease in the frequency of the symptoms and the extent along with the intensity of the erythema was observed. Interestingly, flushing occurred on his left cheek after eating cucumber at 8 months of age. At the age of 12 months, flushing still appeared localized to the right cheek with lemon, spicy foods, cucumber, and butter, and was sometimes also seen on the left cheek (Figure 2, A and B). Frey syndrome, otherwise named auriculotemporal nerve (ATN) syndrome, is characterized by unilateral (rarely bilateral) recurrent episodes of flushing and sweating over the parotid area after gustatory stimulus. The syndrome most often occurs in adults after parotid surgery and very uncommon in children. Because the disease often exhibits spontaneous resolution within a few years in children, no treatment is recommended.
Department of Pediatric Allergy, Hacettepe University Faculty of Medicine, Sihhiye, 06100, Ankara, Turkey No funding was received for this work. Conflicts of interest: The authors declare that they have no relevant conflicts. Received for publication August 13, 2014; revised November 13, 2014; accepted for publication November 14, 2014. Available online -Corresponding author: Bulent Enis Sekerel, MD, Professor in Pediatrics, Pediatric Allergy and Asthma Unit, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey. E-mail: [email protected]. 2213-2198 Ó 2015 American Academy of Allergy, Asthma & Immunology http://dx.doi.org/10.1016/j.jaip.2014.11.010
FIGURE 1. Unilateral flushing appearing on the face after an open challenge test with pear at 6 months of age.
It has been proposed that a perinatal birth trauma during delivery may be a possible reason for damage to the auriculotemporal nerve.1 Moreover, congenital cases without any known trauma have also been reported, but only a few.2 Postulated mechanisms for the congenital cases include a congenital aberrant pathway, crossover of the fibers caused by loss of insulation around the neural sheaths, neural irritation due to scar tissue formation, subclinical intrauterine infection, and mild injury to the parotid gland.3 Although the majority of patients have flushing in the preauricular region compatible with the cutaneous distribution of ATN, the reaction of our patient expanded from the cheeks to the middle of the forehead. Additionally, he had no history of trauma and had bilateral symptoms, strengthening the assumption of a central nervous system abnormality or immaturation. We suppose that keeping in mind this rare disease induced by tactile or masticatory stimuli would help physicians to differentiate it from food allergy and obviate unnecessary diagnostic tests and elimination diets. REFERENCES 1. Caulley L, Hong P. Pediatric auriculotemporal nerve (Frey) syndrome. CMAJ 2013;185:504. 2. Beck SA, Burks AW, Woody RC. Auriculotemporal syndrome seen clinically as food allergy. Pediatrics 1989;83:601-3. 3. Moreno-Arias GA, Grimalt R, Llusa M, Cadavid J, Otal C, Ferrando J. Frey’s syndrome. J Pediatr 2001;138:294.
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BUYUKTIRYAKI AND SEKEREL
J ALLERGY CLIN IMMUNOL PRACT MONTH 2015
FIGURE 2. A, Flushing on the left cheek after an open challenge test with cucumber at 12 months of age. B, Flushing on the right cheek after an open challenge test with lemon at 12 months of age.
