craniopharyngioma masquerading as allergic rhititis: a case report

1
S342 Abstracts J ALLERGY CLIN IMMUNOL JANUARY 2002 '~N Craniopharyngioma Masquerading as Allergic Rhinitis: A li, lP~lPll,ilCase Report Kirk Holden Waibel*, Rohit K Katial§ *Waiter Reed Army Medical Cen- ter, Wheaton, MD §Walter Reed Army Medical Center, Washington, DC INTRODUCTION: Seasonal allergic rhinitis (SAR) and perennial allergic rhinitis (PAR) can present with rhinorrhea, congestion, and headache. While headaches are often attributed to allergic disease, they can be overlooked as a manifestation of other underlying processes to include sinusitis, primary headaches, and intracranial lesions. We present a case of allergic rhinitis that incidentally had intracranial calcifications on sinus imaging, and subsequently was diagnosed with a craniopharyngioma. CASE REPORT: A 13 year-old male presents with a six year history of seasonal ocular pruritis, sneezing, rhinorrhea, perennial congestion, and a one year history of progressive headaches. The patient's history, physical exam, nasal smear, and skin prick testing were supportive of allergic rhini- tis. A coronal sinus CT scan demonstrated normal sinuses but incidental intracranial calcifications. Neurology and endocrinology examinations were normal except for a low somatomedin C of 184.0 ng/mL (n1202-957 ng/mL). A head MRI showed a cystic lesion in the sella consistent with a craniopharyngioma, and subtotal tumor resection was confirmatory. DISCUSSION: Headache can be a presenting symptom of allergic rhinitis but is rarely a prominent complaint. Our patient's history, nasal cytology, and skin tests were consistent with allergic rhinitis, while headache was his only neuroendocrine sign or symptom of craniopharyn- gioma. Thus, this lesion could have easily been missed if not incidentally detected on coronal sinus imaging. In conclusion, health care providers should be cautious to attribute prominent headache complaints to "aller- gies" or "sinus-related disease" and consider other etiologies. 1061 Allergyto Chinchilla John M Kelso*, Richard T Jones§, John W Yunginger§ *Naval Medical Center, San Diego, CA §Mayo Clinic, Rochester, MN A 22 year old Navy Hospital Corpsman had worked in a laboratory ani- mal facility for the past 2 years with several animal species. He stated that within the last month he had noted that when he handled the chinchillas (all female animals) he got an immediate rash (itchy red spots, slightly raised) not only where he actually touched the animals, but also on any exposed skin, e.g. around his neck. In the same setting, he also developed itchy eyes and runny nose and on 3 occasions had developed cough and shortness of breath as well. At 2 AM the night after one such episode, he developed symptoms of shortness of breath and wheeze severe enough to visit the emergency room. He had no previous history of rhinitis or asthma. He had 2 guinea pigs at home as pets but denied symptoms around them. His prick skin tests to a large panel of common aeroallergens were negative. Prick skin tests to commercial extracts of common laboratory animals were also negative except for guinea pig (3 mm wheal and 11 mm flare). A crude extract made by pulverizing shed chinchilla hair in saline (soaked for 10 minutes, filtered through a 0.22 micron filter) gave a negative skin test result. However, subsequent testing with an extract of clipped hair gave a weakly positive result (3 mm wheal and 6 mm flare). A filtered full strength urine prick test gave an 8 mm wheal and 50 mm flare on the was negative on a non-atopic control subject. A RAST using an te from shed chinchilla hair was positive (class 4). A RAST to was also positive (class 3), but RASTs to rabbit, hamster, mouse, mink as well as cat and dog were completely negative. Allergy to animals is quite common, however allergy to chinchilla has been dy rarely. The major source of allergen for rats, mice and guinea e, although some allergen can be found in hair or dander. Chin- ~odents, but are more closely related to guinea pigs than to other , history, skin testing and IgE antibody testing our patient seems rgic to chinchilla, manifesting as contact urticaria, allergic rhini- ma. He appears to have IgE antibody to allergens present in both Lair. Although not tested, it is possible that there is some cross- reactivity with guinea pig allergens. Immunotherapy with laboratory ani- mal allergens has been reported, but our patient was reassigned to duties away from the animals and had no further symptoms. 1062 Mucoid Impaction:A LocalizedFormof ABPA Blake G Scheer*, Patricia S Hutcheson§, Julio A Lagos~, John A Wood~, Raymond G Slavin* *St Louis University, St Louis, MO §St Louis Uni- versity, Saint Louis, MO ¥St Luke's Hospital, Chesterfield, MO Mucoid impaction is defined as the obstruction of proximal bronchi by mucous plugs and exudates. Clinically, these patients usually have histories of asthma or chronic bronchitis and present with symptoms of fever, chest pain, hemoptysis, upper respiratory infection, or cough. There are striking similarities between patients with mucoid impaction and those with allergic bronchopulmonary aspergillosis (ABPA). ABPA is characterized by asth- ma, recurrent infiltrates on chest x-ray, positive skin test, precipitating anti- bodies, specific IgG and IgE to Aspergillusfumigatus, and peripheral blood eosinophilia. ABPA, in fact, is occasionally defined as "mucoid microim- paction". We evaluated three patients with mucoid impaction for diagnostic criteria of ABPA, as well as HLA type. All three of these patients had posi- tive skin tests to Aspergillus and had evidence of specific IgE and IgG to Aspergillus. All had a history of asthma and a modest elevation of IgE. After bronchoscopy, microscopic evaluation of the mucous plugs revealed evi- dence of allergic inflammation that included numerous eosinophils, Char- cot-Leyden crystals, and fungal hyphae. Only one of the three patients had precipitating antibodies and none had peripheral blood eosinophilia. All had either HLA-DR 2 or 5 that have been shown to correlate with suscepti- bility to ABPA. Two patients had HLA-DQ 2, which we have previously found contributes to resistance to ABPA. It is possibly the combination of these genetic elements that can determine the severity and presentation of disease. With these similarities, patients with mucoid impaction could rep- resent a localized form of ABPA. 4 ~rli~'j~ Compliance With Anti-Tuberculous Drugs in Asthmatic lUUJ Children Nermin Giiler, Zeynep Tama.~; (llker Ones Istanbul University, Istanbul Medical Faculty, Istanbul, Turkey Numerous studies have shown that patients with chronic diseases do not properly take a remarkable proportion of prescribed medication. This study aimed to evaluate whether the presence of asthma and its severity affects compliance with the treatment of concomitant tuberculosis (TB) in chil- dren. Twenty-seven asthmatic children (14F, 13M) who were consequently diagnosed as TB infection or disease were enrolled in the study. The mean age of children was 6.04 _+ 3.16 years (range: 1-13 years). TB infection was diagnosed in 7 (25.9%) patients due to PPD positivity and/or conversion, and isoniazid was prescribed. TB disease was found in 20 (74.1%) children based on the clinical, radiological findings and PPD positivity. They were diagnosed and treated with isoniazid, rifampicin and pyrazinamide accord- ing to the Guideliness of American Thoracic Society. Eight (29.6%) chil- dren had mild asthma and used inhaled bronchodilators as needed. The rest of the patients were moderate asthmatics and used inhaled anti-inflamma- tory drugs regularly and rescue therapy as needed. A questionnaire was filled by one of the parents and morning urine was checked for the presence of anti-tuberculosis drugs (isoniazid and/or rifampicin and/or pyrazi- narnide) for each child on the visit days. Twenty-one (77.8%) of the parents declared that their children took the drugs regularly. According to urine test it was seen that only 13 (48.1%) of children were receiving anti-tuberculo- sis drugs properly, 9 (33.3%) of them were partially using the drugs. No drug was detected in urine of 5 (18.5%) of the patients. There was no signif- icant correlation between the severity of asthma and compliance with the treatment. Socio-economical factors (such as age, education and job of the parents, number of persons and children in the family, ownership of the house and number of the rooms, social health insurance) did not affect the attitude of the parents. Twenty-two (82%) of the parents believed in the

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Page 1: Craniopharyngioma masquerading as allergic rhititis: A case report

S342 Abst rac ts J ALLERGY CLIN IMMUNOL JANUARY 2002

' ~ N Craniopharyngioma Masquerading as Allergic Rhinitis: A li, lP~lPll,il Case Report

Kirk Holden Waibel*, Rohit K Katial§ *Waiter Reed Army Medical Cen- ter, Wheaton, MD §Walter Reed Army Medical Center, Washington, DC

INTRODUCTION: Seasonal allergic rhinitis (SAR) and perennial allergic rhinitis (PAR) can present with rhinorrhea, congestion, and headache. While headaches are often attributed to allergic disease, they can be overlooked as a manifestation of other underlying processes to include sinusitis, primary headaches, and intracranial lesions. We present a case of allergic rhinitis that incidentally had intracranial calcifications on sinus imaging, and subsequently was diagnosed with a craniopharyngioma.

CASE REPORT: A 13 year-old male presents with a six year history of seasonal ocular pruritis, sneezing, rhinorrhea, perennial congestion, and a one year history of progressive headaches. The patient's history, physical exam, nasal smear, and skin prick testing were supportive of allergic rhini- tis. A coronal sinus CT scan demonstrated normal sinuses but incidental intracranial calcifications. Neurology and endocrinology examinations were normal except for a low somatomedin C of 184.0 ng/mL (n1202-957 ng/mL). A head MRI showed a cystic lesion in the sella consistent with a craniopharyngioma, and subtotal tumor resection was confirmatory.

DISCUSSION: Headache can be a presenting symptom of allergic rhinitis but is rarely a prominent complaint. Our patient's history, nasal cytology, and skin tests were consistent with allergic rhinitis, while headache was his only neuroendocrine sign or symptom of craniopharyn- gioma. Thus, this lesion could have easily been missed if not incidentally detected on coronal sinus imaging. In conclusion, health care providers should be cautious to attribute prominent headache complaints to "aller- gies" or "sinus-related disease" and consider other etiologies.

1061 Allergy to Chinchilla

John M Kelso*, Richard T Jones§, John W Yunginger§ *Naval Medical Center, San Diego, CA §Mayo Clinic, Rochester, MN

A 22 year old Navy Hospital Corpsman had worked in a laboratory ani- mal facility for the past 2 years with several animal species. He stated that within the last month he had noted that when he handled the chinchillas (all female animals) he got an immediate rash (itchy red spots, slightly raised) not only where he actually touched the animals, but also on any exposed skin, e.g. around his neck. In the same setting, he also developed itchy eyes and runny nose and on 3 occasions had developed cough and shortness of breath as well. At 2 AM the night after one such episode, he developed symptoms of shortness of breath and wheeze severe enough to visit the emergency room. He had no previous history of rhinitis or asthma. He had 2 guinea pigs at home as pets but denied symptoms around them. His prick skin tests to a large panel of common aeroallergens were negative. Prick skin tests to commercial extracts of common laboratory animals were also negative except for guinea pig (3 mm wheal and 11 mm flare). A crude extract made by pulverizing shed chinchilla hair in saline (soaked for 10 minutes, filtered through a 0.22 micron filter) gave a negative skin test result. However, subsequent testing with an extract of clipped hair gave a weakly positive result (3 mm wheal and 6 mm flare). A filtered full strength

urine prick test gave an 8 mm wheal and 50 mm flare on the was negative on a non-atopic control subject. A RAST using an te from shed chinchilla hair was positive (class 4). A RAST to was also positive (class 3), but RASTs to rabbit, hamster, mouse, mink as well as cat and dog were completely negative. Allergy to animals is quite common, however allergy to chinchilla has been dy rarely. The major source of allergen for rats, mice and guinea e, although some allergen can be found in hair or dander. Chin- ~odents, but are more closely related to guinea pigs than to other , history, skin testing and IgE antibody testing our patient seems rgic to chinchilla, manifesting as contact urticaria, allergic rhini- ma. He appears to have IgE antibody to allergens present in both Lair. Although not tested, it is possible that there is some cross-

reactivity with guinea pig allergens. Immunotherapy with laboratory ani- mal allergens has been reported, but our patient was reassigned to duties away from the animals and had no further symptoms.

1 0 6 2 Mucoid Impaction: A Localized Form of ABPA

Blake G Scheer*, Patricia S Hutcheson§, Julio A Lagos~, John A Wood~, Raymond G Slavin* *St Louis University, St Louis, MO §St Louis Uni- versity, Saint Louis, MO ¥St Luke's Hospital, Chesterfield, MO

Mucoid impaction is defined as the obstruction of proximal bronchi by mucous plugs and exudates. Clinically, these patients usually have histories of asthma or chronic bronchitis and present with symptoms of fever, chest pain, hemoptysis, upper respiratory infection, or cough. There are striking similarities between patients with mucoid impaction and those with allergic bronchopulmonary aspergillosis (ABPA). ABPA is characterized by asth- ma, recurrent infiltrates on chest x-ray, positive skin test, precipitating anti- bodies, specific IgG and IgE to Aspergillusfumigatus, and peripheral blood eosinophilia. ABPA, in fact, is occasionally defined as "mucoid microim- paction". We evaluated three patients with mucoid impaction for diagnostic criteria of ABPA, as well as HLA type. All three of these patients had posi- tive skin tests to Aspergillus and had evidence of specific IgE and IgG to Aspergillus. All had a history of asthma and a modest elevation of IgE. After bronchoscopy, microscopic evaluation of the mucous plugs revealed evi- dence of allergic inflammation that included numerous eosinophils, Char- cot-Leyden crystals, and fungal hyphae. Only one of the three patients had precipitating antibodies and none had peripheral blood eosinophilia. All had either HLA-DR 2 or 5 that have been shown to correlate with suscepti- bility to ABPA. Two patients had HLA-DQ 2, which we have previously found contributes to resistance to ABPA. It is possibly the combination of these genetic elements that can determine the severity and presentation of disease. With these similarities, patients with mucoid impaction could rep- resent a localized form of ABPA.

4 ~rli~'j~ Compliance With Anti-Tuberculous Drugs in Asthmatic l U U J Children

Nermin Giiler, Zeynep Tama.~; (llker Ones Istanbul University, Istanbul Medical Faculty, Istanbul, Turkey

Numerous studies have shown that patients with chronic diseases do not properly take a remarkable proportion of prescribed medication. This study aimed to evaluate whether the presence of asthma and its severity affects compliance with the treatment of concomitant tuberculosis (TB) in chil- dren. Twenty-seven asthmatic children (14F, 13M) who were consequently diagnosed as TB infection or disease were enrolled in the study. The mean age of children was 6.04 _+ 3.16 years (range: 1-13 years). TB infection was diagnosed in 7 (25.9%) patients due to PPD positivity and/or conversion, and isoniazid was prescribed. TB disease was found in 20 (74.1%) children based on the clinical, radiological findings and PPD positivity. They were diagnosed and treated with isoniazid, rifampicin and pyrazinamide accord- ing to the Guideliness of American Thoracic Society. Eight (29.6%) chil- dren had mild asthma and used inhaled bronchodilators as needed. The rest of the patients were moderate asthmatics and used inhaled anti-inflamma- tory drugs regularly and rescue therapy as needed. A questionnaire was filled by one of the parents and morning urine was checked for the presence of anti-tuberculosis drugs (isoniazid and/or rifampicin and/or pyrazi- narnide) for each child on the visit days. Twenty-one (77.8%) of the parents declared that their children took the drugs regularly. According to urine test it was seen that only 13 (48.1%) of children were receiving anti-tuberculo- sis drugs properly, 9 (33.3%) of them were partially using the drugs. No drug was detected in urine of 5 (18.5%) of the patients. There was no signif- icant correlation between the severity of asthma and compliance with the treatment. Socio-economical factors (such as age, education and job of the parents, number of persons and children in the family, ownership of the house and number of the rooms, social health insurance) did not affect the attitude of the parents. Twenty-two (82%) of the parents believed in the