- Email: [email protected]
Horizontal sections for the presence of eccrine glands
Letters 369
J AM ACAD DERMATOL VOLUME 52, NUMBER 2
Table I. Summary of findings in 6 patients with vaccine-associated Sweet’s syndrome Clinical symptoms Vaccine
Influenza Small pox Small pox BCG Pneumo-coccal BCG
Age (y), GI or sex URI
44 47 70 23 36 36
F F F F F F
ÿ ÿ ÿ ÿ ÿ ÿ
Fe
1 1 1 1 1 1
MSI
Lesion location
OI
1 1(Oral) ÿ 1(Ocular) 1 1(Ocular) ÿ ÿ 1 ÿ 1 ÿ
ESR Anemia Np Bx (mm/h) (g/dL) PT
B, LE, N, VS, T, UE ÿ 1 T, UE, VS ÿ 1 P, S, T, UE 1 1 FA, UE, VS 1 1 LE, T, UE 1 1 LE, T, UE, VS 1 1
84 35 58 55 NR 4
10.8 NR 83 NR NR NR
ÿ NR NR NR NR NR
K
Study
ÿ NR ÿ NR NR ÿ
CR Gunawardena et al1 Gunawardena et al1 Radeff and Harms2 Maddox and Motley3 Carpentier et al4
B, Back; Bx, biopsy confirmed neutrophilic dermatosis; CR, current report; ESR, erythrocyte sedimentation rate; F, female; FA, face; Fe, fever; GI, gastrointestinal infection; K, abnormal kidney function; LE, lower extremities; MSI, muscule-skeletal involvement; N, neck; Np, neutrophilia; NR, not reported; OI, ocular or oral mucous membrane involvement; P, palms; PT, abnormal platelet count; S, soles; T, trunk; UE, upper extremities; URI, upper respiratory infection; VS, vaccination site.
Correspondence to: Marina Jovanovic´, MD, PhD Clinic for Dermatovenerologic Diseases, Clinical Center, Medical Faculty University of Novi Sad, 21000 Novi Sad Hajduk Veljkova 1-3, Serbia and Montenegro E-mail: [email protected]
REFERENCES 1. Gunawardena DA, Gunawardena KA, Ratnayaka RM, Vasanthanathan NS. The clinical spectrum of Sweet’s syndrome (acute febrile neutrophilic dermatosis)—a report of eighteen cases. Br J Dermatol 1975;92:363-73. 2. Radeff B, Harms M. Acute febrile neutrophilic dermatosis (Sweet’s syndrome) following BCG vaccination. Acta Derm Venereol 1986;66:357-8. 3. Maddox PR, Motley RJ. Sweet’s syndrome: a severe complication of pneumococcal vaccination following emergency splenectomy. Br J Surg 1990;77:809-10. 4. Carpentier O, Piette F, Delaporte E. Sweet’s syndrome after BCG vaccination. Acta Dermatol Venereol 2002;82:221. 5. von den Driesch P. Sweet’s syndrome (acute febrile neutrophilic dermatosis). J Am Acad Dermatol 1994;31:535-56. 6. Cohen PR, Kurzrock R. Sweet’s syndrome and cancer. Clin Dermatol 1993;11:149-57. 7. Walker DC, Cohen PR. Trimethoprim-sulfamethoxazole-associated acute febrile neutrophilic dermatosis: case report and review of drug-induced Sweet’s syndrome. J Am Acad Dermatol 1996;34(5 Pt 2):918-23. 8. Holdiness MR. Adverse cutaneous reactions to influenza vaccination and chemotherapy. Int J Dermatol 2001;40:427-30. doi:10.1016/j.jaad.2004.07.061
Horizontal sections for the presence of eccrine glands To the Editor: The presence of eccrine glands in the skin is often easily assessed by a good physical examination. The presence of sweat confirms the presence of some eccrine gland function. A few
Fig 1. Transverse section through papillary dermis reveals equidistant eccrine ducts throughout specimen, with one cluster marked (*) to left of each duct. Thick stratum corneum that characterizes palmar skin is visible, with occasional clear spaces in horn identifying path of acrosyringia. At level through subcutaneous fat, multiple eccrine gland coils were seen (inset).
abnormalities of the eccrine glands are of importance to dermatologists. The presence of sweating can be confirmed with a starch iodine test. In some patients, such as infants, this test is difficult to perform. Occasionally dermatologists are called on to evaluate infants and children for disorders of sweat glands, namely anhidrotic ectodermal dysplasia and hidrotic ectodermal dysplasia.1,2 Anhidrotic ectodermal dysplasia, also know as hypohidrotic ectodermal dysplasia, is a predominately x-linked recessive genodermatosis. Autosomal recessive patterns of inheritance can also be seen. Patients may have the characteristic facial features (frontal bossing, periorbital hyperpigmentation), abnormal teeth and hair, and hyperpyrexia secondary to severe hypohidrosis or anhidrosis. An important aspect of caring for these patients is to avoid overheating
370 Letters
because they lack eccrine sweat glands. A way to help confirm the diagnosis is to perform a skin biopsy and to assess for eccrine glands. The most logical area to biopsy is the palm or sole, two areas with high concentrations of eccrine glands. We describe a technique of performing horizontal sections of a skin punch biopsy as a way to evaluate for eccrine glands. We feel this method has advantages over the standard vertical sectioning routinely performed to evaluate for eccrine glands, and this can be a useful method for studying ectodermal dysplasia syndromes.
CASE REPORT A 4-month-old Amish child was admitted to the hospital for fevers of unknown origin. He had a significant family history of a brother dying as an infant with similar fevers. A diagnosis was never made for the deceased brother. He was sent to us specifically to look into the possibility of anhidrotic ectodermal dysplasia. We examined the patient and found no obvious cutaneous disorders. He had no clinical features of anhidrotic ectodermal dysplasia. The parents said the child did not sweat. We proceeded to perform a punch biopsy of the palm. In our practice we evaluate scalp biopsy specimens for alopecia using a horizontal sectioning technique. That allows for all the hair follicles in the specimen to be reviewed. We used this method to search for eccrine glands. A routine hematoxylin and eosin stain was used to assess the eccrine glands. The biopsy specimen did indeed show a normalappearing density of eccrine glands, which were evenly spaced throughout (Fig 1). This technique proved to be a simple and rapid way to demonstrate the presence of eccrine glands. We concluded the child did not have anhidrotic ectodermal dysplasia, because of the normal density of eccrine glands. The biopsy specimen, however, did not rule out the possibility of hidrotic ectodermal dysplasia, as the eccrine glands can appear normal in that condition. After further evaluation by his pediatricians no firm diagnosis was made. He was believed to have an immunodeficiency syndrome, but unfortunately he was lost to follow-up. In conclusion, we feel that horizontal sectioning of skin biopsy specimens is an excellent way to evaluate for eccrine glands. Bryan E. Anderson, MD Klaus F. Helm, MD Michael Ioffreda, MD Hershey Medical Center, Hershey, Pennsylvania Correspondence to: Bryan E. Anderson, MD Department of Dermatology Hershey Medical Center
J AM ACAD DERMATOL FEBRUARY 2005
PO Box 850, 500 University Dr, UPC II Suite 4300 Hershey, PA 17033 E-mail: [email protected]
REFERENCES 1. Berg D, Weingold DH, Abson KG, Olsen EA. Sweating in ectodermal dysplasia syndromes. Arch Dermatol 1990;126: 1075-9. 2. Wisniewski SA, Kobielak A, Trzeciak WH, Kobielak K. Recent advances in understanding of the molecular basis of anhidrotic ectodermal dysplasia: discovery of a ligand, ectodyslpasian A and its two receptors. J Appl Genet 2002;43:97-107. doi:10.1016/j.jaad.2004.08.048
Fixed food eruption caused by lactose identified after oral administration of four unrelated drugs To the Editor: Lactose is a common dietary constituent. We describe a patient with fixed food eruption caused by lactose identified after administrating four unrelated drugs. A 54-year-old woman visited the outpatient clinic because of eyelid pruritus after administrating cilazapril for hypertension. Physical examination revealed a hyperpigmented edematous erythema on both eyelids. She reported a similar eruption after consuming dairy products (milk or instant potage soup) during the last 10 years. Therefore, she reported to have avoided dairy products as much as possible. Because we considered the diagnosis as drug eruption, we recommended her to avoid cilazapril. The eruption remitted after corticosteroid therapy. The lesion, however, recurred after separate administration of estriol for menopausal syndrome, clotiazepam for insomnia, and enalapril maleate for hypertension. Because these drugs are unrelated, we considered that excipient common to these drugs might have induced this eruption. Because the single common excipient was lactose, we considered the possible involvement of lactose. Results of patch tests for cilazapril, estriol, clotiazepam, enalapril, and lactose were negative. Radioallergosorbent test to lacto-albumin/lacto-globulin was normal. An oral challenge test with 0.5 g of lactose provoked similar lesion on the eyelids after 24 hours (Fig 1). Although one tablet of cilazapril also provoked the same lesion, corn starch and wheat produced no reaction. The patient had not taken over-the-counter drugs for more than 10 years. She reported that her deceased mother had developed lip swelling and her son develops abdominal distension, increased
J AM ACAD DERMATOL VOLUME 52, NUMBER 2
Table I. Summary of findings in 6 patients with vaccine-associated Sweet’s syndrome Clinical symptoms Vaccine
Influenza Small pox Small pox BCG Pneumo-coccal BCG
Age (y), GI or sex URI
44 47 70 23 36 36
F F F F F F
ÿ ÿ ÿ ÿ ÿ ÿ
Fe
1 1 1 1 1 1
MSI
Lesion location
OI
1 1(Oral) ÿ 1(Ocular) 1 1(Ocular) ÿ ÿ 1 ÿ 1 ÿ
ESR Anemia Np Bx (mm/h) (g/dL) PT
B, LE, N, VS, T, UE ÿ 1 T, UE, VS ÿ 1 P, S, T, UE 1 1 FA, UE, VS 1 1 LE, T, UE 1 1 LE, T, UE, VS 1 1
84 35 58 55 NR 4
10.8 NR 83 NR NR NR
ÿ NR NR NR NR NR
K
Study
ÿ NR ÿ NR NR ÿ
CR Gunawardena et al1 Gunawardena et al1 Radeff and Harms2 Maddox and Motley3 Carpentier et al4
B, Back; Bx, biopsy confirmed neutrophilic dermatosis; CR, current report; ESR, erythrocyte sedimentation rate; F, female; FA, face; Fe, fever; GI, gastrointestinal infection; K, abnormal kidney function; LE, lower extremities; MSI, muscule-skeletal involvement; N, neck; Np, neutrophilia; NR, not reported; OI, ocular or oral mucous membrane involvement; P, palms; PT, abnormal platelet count; S, soles; T, trunk; UE, upper extremities; URI, upper respiratory infection; VS, vaccination site.
Correspondence to: Marina Jovanovic´, MD, PhD Clinic for Dermatovenerologic Diseases, Clinical Center, Medical Faculty University of Novi Sad, 21000 Novi Sad Hajduk Veljkova 1-3, Serbia and Montenegro E-mail: [email protected]
REFERENCES 1. Gunawardena DA, Gunawardena KA, Ratnayaka RM, Vasanthanathan NS. The clinical spectrum of Sweet’s syndrome (acute febrile neutrophilic dermatosis)—a report of eighteen cases. Br J Dermatol 1975;92:363-73. 2. Radeff B, Harms M. Acute febrile neutrophilic dermatosis (Sweet’s syndrome) following BCG vaccination. Acta Derm Venereol 1986;66:357-8. 3. Maddox PR, Motley RJ. Sweet’s syndrome: a severe complication of pneumococcal vaccination following emergency splenectomy. Br J Surg 1990;77:809-10. 4. Carpentier O, Piette F, Delaporte E. Sweet’s syndrome after BCG vaccination. Acta Dermatol Venereol 2002;82:221. 5. von den Driesch P. Sweet’s syndrome (acute febrile neutrophilic dermatosis). J Am Acad Dermatol 1994;31:535-56. 6. Cohen PR, Kurzrock R. Sweet’s syndrome and cancer. Clin Dermatol 1993;11:149-57. 7. Walker DC, Cohen PR. Trimethoprim-sulfamethoxazole-associated acute febrile neutrophilic dermatosis: case report and review of drug-induced Sweet’s syndrome. J Am Acad Dermatol 1996;34(5 Pt 2):918-23. 8. Holdiness MR. Adverse cutaneous reactions to influenza vaccination and chemotherapy. Int J Dermatol 2001;40:427-30. doi:10.1016/j.jaad.2004.07.061
Horizontal sections for the presence of eccrine glands To the Editor: The presence of eccrine glands in the skin is often easily assessed by a good physical examination. The presence of sweat confirms the presence of some eccrine gland function. A few
Fig 1. Transverse section through papillary dermis reveals equidistant eccrine ducts throughout specimen, with one cluster marked (*) to left of each duct. Thick stratum corneum that characterizes palmar skin is visible, with occasional clear spaces in horn identifying path of acrosyringia. At level through subcutaneous fat, multiple eccrine gland coils were seen (inset).
abnormalities of the eccrine glands are of importance to dermatologists. The presence of sweating can be confirmed with a starch iodine test. In some patients, such as infants, this test is difficult to perform. Occasionally dermatologists are called on to evaluate infants and children for disorders of sweat glands, namely anhidrotic ectodermal dysplasia and hidrotic ectodermal dysplasia.1,2 Anhidrotic ectodermal dysplasia, also know as hypohidrotic ectodermal dysplasia, is a predominately x-linked recessive genodermatosis. Autosomal recessive patterns of inheritance can also be seen. Patients may have the characteristic facial features (frontal bossing, periorbital hyperpigmentation), abnormal teeth and hair, and hyperpyrexia secondary to severe hypohidrosis or anhidrosis. An important aspect of caring for these patients is to avoid overheating
370 Letters
because they lack eccrine sweat glands. A way to help confirm the diagnosis is to perform a skin biopsy and to assess for eccrine glands. The most logical area to biopsy is the palm or sole, two areas with high concentrations of eccrine glands. We describe a technique of performing horizontal sections of a skin punch biopsy as a way to evaluate for eccrine glands. We feel this method has advantages over the standard vertical sectioning routinely performed to evaluate for eccrine glands, and this can be a useful method for studying ectodermal dysplasia syndromes.
CASE REPORT A 4-month-old Amish child was admitted to the hospital for fevers of unknown origin. He had a significant family history of a brother dying as an infant with similar fevers. A diagnosis was never made for the deceased brother. He was sent to us specifically to look into the possibility of anhidrotic ectodermal dysplasia. We examined the patient and found no obvious cutaneous disorders. He had no clinical features of anhidrotic ectodermal dysplasia. The parents said the child did not sweat. We proceeded to perform a punch biopsy of the palm. In our practice we evaluate scalp biopsy specimens for alopecia using a horizontal sectioning technique. That allows for all the hair follicles in the specimen to be reviewed. We used this method to search for eccrine glands. A routine hematoxylin and eosin stain was used to assess the eccrine glands. The biopsy specimen did indeed show a normalappearing density of eccrine glands, which were evenly spaced throughout (Fig 1). This technique proved to be a simple and rapid way to demonstrate the presence of eccrine glands. We concluded the child did not have anhidrotic ectodermal dysplasia, because of the normal density of eccrine glands. The biopsy specimen, however, did not rule out the possibility of hidrotic ectodermal dysplasia, as the eccrine glands can appear normal in that condition. After further evaluation by his pediatricians no firm diagnosis was made. He was believed to have an immunodeficiency syndrome, but unfortunately he was lost to follow-up. In conclusion, we feel that horizontal sectioning of skin biopsy specimens is an excellent way to evaluate for eccrine glands. Bryan E. Anderson, MD Klaus F. Helm, MD Michael Ioffreda, MD Hershey Medical Center, Hershey, Pennsylvania Correspondence to: Bryan E. Anderson, MD Department of Dermatology Hershey Medical Center
J AM ACAD DERMATOL FEBRUARY 2005
PO Box 850, 500 University Dr, UPC II Suite 4300 Hershey, PA 17033 E-mail: [email protected]
REFERENCES 1. Berg D, Weingold DH, Abson KG, Olsen EA. Sweating in ectodermal dysplasia syndromes. Arch Dermatol 1990;126: 1075-9. 2. Wisniewski SA, Kobielak A, Trzeciak WH, Kobielak K. Recent advances in understanding of the molecular basis of anhidrotic ectodermal dysplasia: discovery of a ligand, ectodyslpasian A and its two receptors. J Appl Genet 2002;43:97-107. doi:10.1016/j.jaad.2004.08.048
Fixed food eruption caused by lactose identified after oral administration of four unrelated drugs To the Editor: Lactose is a common dietary constituent. We describe a patient with fixed food eruption caused by lactose identified after administrating four unrelated drugs. A 54-year-old woman visited the outpatient clinic because of eyelid pruritus after administrating cilazapril for hypertension. Physical examination revealed a hyperpigmented edematous erythema on both eyelids. She reported a similar eruption after consuming dairy products (milk or instant potage soup) during the last 10 years. Therefore, she reported to have avoided dairy products as much as possible. Because we considered the diagnosis as drug eruption, we recommended her to avoid cilazapril. The eruption remitted after corticosteroid therapy. The lesion, however, recurred after separate administration of estriol for menopausal syndrome, clotiazepam for insomnia, and enalapril maleate for hypertension. Because these drugs are unrelated, we considered that excipient common to these drugs might have induced this eruption. Because the single common excipient was lactose, we considered the possible involvement of lactose. Results of patch tests for cilazapril, estriol, clotiazepam, enalapril, and lactose were negative. Radioallergosorbent test to lacto-albumin/lacto-globulin was normal. An oral challenge test with 0.5 g of lactose provoked similar lesion on the eyelids after 24 hours (Fig 1). Although one tablet of cilazapril also provoked the same lesion, corn starch and wheat produced no reaction. The patient had not taken over-the-counter drugs for more than 10 years. She reported that her deceased mother had developed lip swelling and her son develops abdominal distension, increased