Comment.—Pruritic urticarial plaques and papules of
pregnancy is a distinct rash of pregnancy that typically oc¬ curs in the later part of the third trimester in primigrávi¬ das. The rash frequently begins in the abdominal striae as
small erythematous papules that coalesce into plaques. The rash often spreads to thighs, buttocks, and extremities, and is intensely pruritic. Most patients obtain symptomatic re¬ lief from topical steroids and antihistamines. Possible hormonal mechanisms have been proposed and investigated but studies of human chorionic gonadotropin levels,2 estradiol, and cortisol have all been unrewarding.3 In those cases in which direct and indirect immunofluorescence have been investigated,4 autoantibodies examined,3 or HLA associations sought,5 results have been unrevealing. It carries no known risk to mother or fetus and does not tend to recur in subsequent pregnancies. In a study of 30 cases of PUPPP, Cohen and coworkers1 noted maternal weight gain was 18.1 ± 0.9 kg but only 14.6 ± 1.0 kg in control subjects. There were three twin pregnancies, representing 10% of the PUPPP group, com¬ pared with an expected twin gestation rate at their institu¬ tion of 1.6%. Based on these findings and the fact that PUPPP occurs almost exclusively in primigrávidas, and in the striae, they suggested that skin distention plays a role in the development of PUPPP.1 Yancey and coworkers5 had suggested this association earlier and noted excess weight gain in their patients with PUPPP as well as an increased incidence of twin pregnancies. The two cases reported herein support this idea. In both cases there was exceptional weight gain: due to the triplet pregnancy in the first case and polyhydramnios in the second. The relationship of PUPPP to in vitro fertilization in our cases may simply be coincidental. There is no evidence that any endocrinologie abnormality plays a role in the develop¬ ment of PUPPP and we know of nothing specific to in vitro pregnancies that should predispose the mothers to PUPPP. Marie Anne Beckett, MD Neil S. Goldberg, MD Department of Dermatology New York Medical College Valhalla, NY 10595 1. Cohen LM, Capeless EL, Krusinski PA, Maloney ME. Pruritic urticarial papules and plaques of pregnancy and its relationship to maternal-fetal weight gain and twin pregnancy. Arch Dermatol. 1989;125:1534-1536. 2. Callen JP, Hanno R. Pruritic urticarial papules and plaques of pregnancy (PUPPP). J Am Acad Dermatol. 1981;5:401-405. 3. Alcalay J, Ingber A, Kafri B, et al. Hormonal evaluation and autoimmune background in pruritic urticarial papules and plaques of pregnancy. Am J Obstet Gynecol. 1988;158:417-420. 4. Alcalay J, Ingber A, David M, Hazaz B, Sandback M. Pruritic urticarial papules and plaques of pregnancy: a review of 21 cases. J Reprod Med.
1987;32:315-316. 5. Yancey KB, Hall RP, Lawley TJ. Pruritic urticarial papules and plaques
of pregnancy: clinical
experience in
The Pinnal Hair
Sign
1984;10:473-480.
25
patients. J
Am Acad Dermatol.
To the Editor.\p=m-\We had earlier reported our observation of absence of graying of pinnal hairs in 250 men.1 On continuing to observe pinnal hairs, we have noticed an interesting feature that we wish to share with your readers.
Report of a Case.\p=m-\A60-year-old man with vitilgo showed graying of a few of his pinnal hairs. This made us carefully observe more patients with pinnal hairs. So far we have examined 482 men, all over 50 years of age, with pinnal hairs. Of these, 28 men were seen to have a few gray pinnal hairs. Dermatologic examination of these 28 men revealed that 12 of them showed vitiliginous patches. The size and number of the patches were variable. The sites involved included both covered and exposed areas of the body. There was no
correlation between these parameters and the number of gray pinnal hairs. The remaining 16 patients showed guttate hypomelanosis. There was no correlation between the site, size, and number of guttäte hypomelanotic macules and the intensity of graying of the pinnal hairs. Among the 454 men who did not have gray pinnal hairs, only eight showed guttäte hypomelanosis, but vitíligo was not present in any of them.
Comment—Our clinical study shows that graying of pin¬ nal hairs in an individual could be a marker of hypopigmentary disorders such as vitíligo or guttäte hypomelano¬ sis. We suggest that whenever a man is seen with gray pin¬ nal hairs it is worthwhile to search for either vitiligo or guttäte hypomelanosis in both exposed and covered areas. This was true in 28 of 482 patients. While we submit that this may be too small a number to draw definite conclusions, we call this observation of ours the pinnal hair sign. Jayakar Thomas, MD, PhD D. Prabhavathy, MBBS, DD S. M. Augustine, MD T. C. Muthuswami, MD Department of Dermatology and Leprosy Government General Hospital Madras 600 003, India 1. Thomas J, Prabhavathy D, Augustine SM, Muthuswami TC. Absence of graying of pinnal hairs. Arch Dermatol. 1989;125:1589.
A Peculiar Case of Linear
IgA Bullous Dermatosis
To the Editor.\p=m-\Weobserved an adult patient with sequential development of mucosal lesions typical of cicatricial pemphigoid (CP) and skin lesions clinically suggestive of early, urticarioid bullous pemphigoid (BP). Direct immunofluorescence showed a linear IgA deposition at the dermoepidermal junction in both lesions. The patient did not respond either to the classic combined treatment with diamino-dimethyl-sulfone (DDS) and steroids or to immunosuppressive agents. Interestingly, on the contrary, the mucosal and cutaneous lesions showed a specifically different response to different treatments, ie, the lesions were satisfactorily cured by low doses of tetracyclines and oral
steroids, respectively.
Report of a Case.\p=m-\A61-year-old man was first seen in 1986 with erythematous and erosive lesions of the conjunctival, oral (Fig 1), prepucial, and epiglottal mucosa for 8 months. Major symptoms consisted of burning and clear reduction of smell and taste sensitivity. Histologic examination, performed on a prepucial biopsy specimen, showed a subepithelial bulla and a perivascular infiltrate of mononuclear cells and neutrophils in the lamina propria. Direct immunofluorescence showed a linear deposition of IgA and C3 at the dermoepidermal junction. Indirect immunofluorescence on normal human skin was negative. The diagnosis of linear IgACP was made, and the patient was treated with prednisone (50 mg/d orally) and diamino-dimethyl-sulfone (DDS) (50 to 75 mg/d orally); the above treatment was discontinued after 10 months be¬ cause of minimal improvement of the disease. A new treatment with prednisone (50 mg/d orally), and immunosuppressive agents (azatioprine, 0.5 to 1 mg/kg per day orally) did not produce any significant improvement. This treatment was thus discontinued after 2 months. The patient was left untreated. Seven months later (19 months from the first examination), the patient progressively developed small, erythematous and edematous, itching papules on the limbs (Fig 2), clinically suggesting BP in its early, urticarioid phase. Histologie examination of a skin biopsy specimen showed edema and perivascular lymphohistiocytic infiltrate in the upper dermis; the epidermis and dermoepidermal junction were histologically normal. Direct immunofluorescence showed a linear dep¬ osition of IgA and C3 at the dermoepidermal junction. The cutane¬ ous lesions were rapidly cured by low doses of oral steroids (triamcinolone, 8 mg/d orally, then gradually reduced to 2 mg/d). Due to
Downloaded From: http://archderm.jamanetwork.com/ by a University of Iowa User on 06/11/2015
pregnancy is a distinct rash of pregnancy that typically oc¬ curs in the later part of the third trimester in primigrávi¬ das. The rash frequently begins in the abdominal striae as
small erythematous papules that coalesce into plaques. The rash often spreads to thighs, buttocks, and extremities, and is intensely pruritic. Most patients obtain symptomatic re¬ lief from topical steroids and antihistamines. Possible hormonal mechanisms have been proposed and investigated but studies of human chorionic gonadotropin levels,2 estradiol, and cortisol have all been unrewarding.3 In those cases in which direct and indirect immunofluorescence have been investigated,4 autoantibodies examined,3 or HLA associations sought,5 results have been unrevealing. It carries no known risk to mother or fetus and does not tend to recur in subsequent pregnancies. In a study of 30 cases of PUPPP, Cohen and coworkers1 noted maternal weight gain was 18.1 ± 0.9 kg but only 14.6 ± 1.0 kg in control subjects. There were three twin pregnancies, representing 10% of the PUPPP group, com¬ pared with an expected twin gestation rate at their institu¬ tion of 1.6%. Based on these findings and the fact that PUPPP occurs almost exclusively in primigrávidas, and in the striae, they suggested that skin distention plays a role in the development of PUPPP.1 Yancey and coworkers5 had suggested this association earlier and noted excess weight gain in their patients with PUPPP as well as an increased incidence of twin pregnancies. The two cases reported herein support this idea. In both cases there was exceptional weight gain: due to the triplet pregnancy in the first case and polyhydramnios in the second. The relationship of PUPPP to in vitro fertilization in our cases may simply be coincidental. There is no evidence that any endocrinologie abnormality plays a role in the develop¬ ment of PUPPP and we know of nothing specific to in vitro pregnancies that should predispose the mothers to PUPPP. Marie Anne Beckett, MD Neil S. Goldberg, MD Department of Dermatology New York Medical College Valhalla, NY 10595 1. Cohen LM, Capeless EL, Krusinski PA, Maloney ME. Pruritic urticarial papules and plaques of pregnancy and its relationship to maternal-fetal weight gain and twin pregnancy. Arch Dermatol. 1989;125:1534-1536. 2. Callen JP, Hanno R. Pruritic urticarial papules and plaques of pregnancy (PUPPP). J Am Acad Dermatol. 1981;5:401-405. 3. Alcalay J, Ingber A, Kafri B, et al. Hormonal evaluation and autoimmune background in pruritic urticarial papules and plaques of pregnancy. Am J Obstet Gynecol. 1988;158:417-420. 4. Alcalay J, Ingber A, David M, Hazaz B, Sandback M. Pruritic urticarial papules and plaques of pregnancy: a review of 21 cases. J Reprod Med.
1987;32:315-316. 5. Yancey KB, Hall RP, Lawley TJ. Pruritic urticarial papules and plaques
of pregnancy: clinical
experience in
The Pinnal Hair
Sign
1984;10:473-480.
25
patients. J
Am Acad Dermatol.
To the Editor.\p=m-\We had earlier reported our observation of absence of graying of pinnal hairs in 250 men.1 On continuing to observe pinnal hairs, we have noticed an interesting feature that we wish to share with your readers.
Report of a Case.\p=m-\A60-year-old man with vitilgo showed graying of a few of his pinnal hairs. This made us carefully observe more patients with pinnal hairs. So far we have examined 482 men, all over 50 years of age, with pinnal hairs. Of these, 28 men were seen to have a few gray pinnal hairs. Dermatologic examination of these 28 men revealed that 12 of them showed vitiliginous patches. The size and number of the patches were variable. The sites involved included both covered and exposed areas of the body. There was no
correlation between these parameters and the number of gray pinnal hairs. The remaining 16 patients showed guttate hypomelanosis. There was no correlation between the site, size, and number of guttäte hypomelanotic macules and the intensity of graying of the pinnal hairs. Among the 454 men who did not have gray pinnal hairs, only eight showed guttäte hypomelanosis, but vitíligo was not present in any of them.
Comment—Our clinical study shows that graying of pin¬ nal hairs in an individual could be a marker of hypopigmentary disorders such as vitíligo or guttäte hypomelano¬ sis. We suggest that whenever a man is seen with gray pin¬ nal hairs it is worthwhile to search for either vitiligo or guttäte hypomelanosis in both exposed and covered areas. This was true in 28 of 482 patients. While we submit that this may be too small a number to draw definite conclusions, we call this observation of ours the pinnal hair sign. Jayakar Thomas, MD, PhD D. Prabhavathy, MBBS, DD S. M. Augustine, MD T. C. Muthuswami, MD Department of Dermatology and Leprosy Government General Hospital Madras 600 003, India 1. Thomas J, Prabhavathy D, Augustine SM, Muthuswami TC. Absence of graying of pinnal hairs. Arch Dermatol. 1989;125:1589.
A Peculiar Case of Linear
IgA Bullous Dermatosis
To the Editor.\p=m-\Weobserved an adult patient with sequential development of mucosal lesions typical of cicatricial pemphigoid (CP) and skin lesions clinically suggestive of early, urticarioid bullous pemphigoid (BP). Direct immunofluorescence showed a linear IgA deposition at the dermoepidermal junction in both lesions. The patient did not respond either to the classic combined treatment with diamino-dimethyl-sulfone (DDS) and steroids or to immunosuppressive agents. Interestingly, on the contrary, the mucosal and cutaneous lesions showed a specifically different response to different treatments, ie, the lesions were satisfactorily cured by low doses of tetracyclines and oral
steroids, respectively.
Report of a Case.\p=m-\A61-year-old man was first seen in 1986 with erythematous and erosive lesions of the conjunctival, oral (Fig 1), prepucial, and epiglottal mucosa for 8 months. Major symptoms consisted of burning and clear reduction of smell and taste sensitivity. Histologic examination, performed on a prepucial biopsy specimen, showed a subepithelial bulla and a perivascular infiltrate of mononuclear cells and neutrophils in the lamina propria. Direct immunofluorescence showed a linear deposition of IgA and C3 at the dermoepidermal junction. Indirect immunofluorescence on normal human skin was negative. The diagnosis of linear IgACP was made, and the patient was treated with prednisone (50 mg/d orally) and diamino-dimethyl-sulfone (DDS) (50 to 75 mg/d orally); the above treatment was discontinued after 10 months be¬ cause of minimal improvement of the disease. A new treatment with prednisone (50 mg/d orally), and immunosuppressive agents (azatioprine, 0.5 to 1 mg/kg per day orally) did not produce any significant improvement. This treatment was thus discontinued after 2 months. The patient was left untreated. Seven months later (19 months from the first examination), the patient progressively developed small, erythematous and edematous, itching papules on the limbs (Fig 2), clinically suggesting BP in its early, urticarioid phase. Histologie examination of a skin biopsy specimen showed edema and perivascular lymphohistiocytic infiltrate in the upper dermis; the epidermis and dermoepidermal junction were histologically normal. Direct immunofluorescence showed a linear dep¬ osition of IgA and C3 at the dermoepidermal junction. The cutane¬ ous lesions were rapidly cured by low doses of oral steroids (triamcinolone, 8 mg/d orally, then gradually reduced to 2 mg/d). Due to
Downloaded From: http://archderm.jamanetwork.com/ by a University of Iowa User on 06/11/2015
