Journal of Dermatology 2015; 42: 1101–1135
LETTERS TO THE EDITOR
Use of propranolol on a nasal hemangioma in an extremely low birthweight premature infant Dear Editor, We describe an extremely low birthweight infant of 700 g of 25 weeks’ gestation who presented on day 28 of age (11 weeks corrected) with infantile hemangioma (IH) on his nose, and was successfully treated with oral propranolol. The lesion was first noticed as a small red spot but had grown quickly, covering the entire nose on day 56 (7 weeks corrected) (Fig. 1a). Although clinically indicated for propranolol treatment, it was not commenced because of the potential side-effects on the cardiorespiratory system of this 1000-g infant. On day 90 (2 weeks corrected) at a bodyweight of 1500 g, with normal general condition, the baby was finally commenced on oral propranolol at 0.25 mg/kg per day b.i.d. in the neonatal intensive care unit. During the first 2 weeks of treatment, the heart rate dropped slightly between 140– 155 b.p.m. to 110–120 b.p.m. while blood pressure and serum glucose levels remained stable. Then, the dose was gradually increased to 2 mg/kg (full dose) per day on 5 months (Fig. 1f), and his IH had lightened and flattened over time (Fig. 1b–d). When the patient was 2 years old, the tumor had disappeared, leaving no observable nasal deformity and no defect in the underlying nasal cartilage (Fig. 1e). Therefore, propranolol was gradually stopped over 3 weeks; the baby is still in follow up and no recurrence was found at the last visit when he was 2 years and 3 months of age. At this time, the baby was 83 cm in height, 11 kg in weight, 48 cm in head circumference and intelligence development was normal. Thus, there was no obvious growth and development abnormality at the last visit. For preterm infants, evidence for IH treatment is lacking despite their high incidence.1 Despite propranolol becoming the mainstay of therapy in IH since first reported in 2008,2 case reports of extremely low birthweight infants with IH who were treated with propranolol at the age younger than 0 weeks’ corrected age are rare.3,4 This case is of interest because propranolol was started at an age younger than 0 weeks’ corrected age in an extremely Figure 1. (a) Fifty-six days (7 weeks corrected) of age, hemangioma on the nose. (b) Three months (2 weeks corrected), the initial day of propranolol therapy, hemangioma covered his full nose. (c) Four months (2 weeks corrected), after 1 month of propranolol therapy, hemangioma was lightened and flattened. (d) Fifteen months (11.5 months corrected), after 12 months of propranolol therapy, marked improvement in his hemangioma. (e) Two years old (20.5 months corrected), after 21 months of propranolol therapy, hemangioma had disappeared. (f) Patient’s medication dose, age and corrected age.
(a)
(b)
(c)
(d)
(e)
(f)
Correspondence: Lin Ma, M.D., Department of Dermatology, Beijing Children’s Hospital, Capital Medical University, 56 South Li Shi Road, Xi Cheng District, Beijing 100045, China. Email: [email protected]
© 2015 Japanese Dermatological Association
1101
Letters to the Editor
low birthweight. In order to avoid side-effects, we evaluated the general condition of the patient before treatment, monitored him closely when propranolol was given in the first 2 months, and the drug was administrated at 0.125 mg/kg per dose from the beginning. Although the patient had experienced myocardial enzyme level increase which was treated by oral fructose sodium diphosphate solution by our pediatric cardiologist, the side-effect was believed to be temporary and would not cause long-term adverse influence on the patient; thus, propranolol was used successfully without obvious sideeffects. In this case, a cautious and gradual approach in using propranolol for our preterm and low birthweight neonate with IH was adopted. A low initiation dose, gradual dose increase and close monitoring were employed and the patient was successfully treated.
CONFLICT OF INTEREST:
Li LI, Lin MA Department of Dermatology, Beijing Children’s Hospital, Capital Medical University, Beijing, China doi: 10.1111/1346-8138.13029
REFERENCES 1 Goelz R, Poets CF. Incidence and treatment of infantile haemangioma in preterm infants. Arch Dis Child Fetal Neonatal Ed 2015; 100 (1): F85–F91. aute -Labre ze C, Dumas de la Roque E, Hubiche T et al. Proprano2 Le lol for severe hemangiomas of infancy [J]. N Engl J Med 2008; 358 (24): 2649–2651. 3 Erbay A, Sarialioglu F, Malbora B et al. Propranolol for infantile hemangiomas: a preliminary report on efficacy and safety in very low birth weight infants. Turk J Pediatr 2010; 52: 450–456. 4 Frost G, Relic J. Dangers of propranolol in preterm infants. Australas J Dermatol 2013; 54: 237–238.
None.
Case of basosquamous carcinoma: Dermoscopic and immunohistochemical findings Dear Editor, Basosquamous carcinoma (BSC) is a rare subtype of basal cell carcinoma (BCC) that exhibits squamoid cell differentiation and has worse prognosis than other types of BCC.1 There have been only few reports about dermoscopic and immunohistochemical findings of BSC. Here, we report a case of BSC on the leg with interesting dermoscopic and immunohistochemical findings. A 76-year-old man visited our outpatient clinic complaining of an expanding ulcer on his leg for 2 months (Fig. 1a). Physical examination at first visit revealed a 20 mm 9 12 mm skin ulcer with peripheral pigmentation on the anterior aspect of the left lower leg. Dermoscopic examination exhibited multifocal blue-gray pigmentation inside of an extensive ulcer and blue-gray structures in a radial fashion at the periphery of the ulcer (Fig. 1b). Many dotted, glomerular and linear blood vessels were observed in the ulcerated area. White fibrous area and thin scales were also observed at the periphery of the ulcer (Fig. 1b). A pathological diagnosis of the biopsy specimen taken from the lesion was BCC or BSC. The lesion was excised with a 5-mm margin. Pathological findings of the excised specimen revealed a nodular lesion in the dermis with ulceration at the low-power view (Fig. 1c). The peripheral tumor cell nests showed basophilic staining, while the central area showed eosinophilic staining. There was continuity between those two areas. In the basophilic area, neoplastic cells with basaloid features formed nests with nuclear palisad-
ing at the periphery and connected to the overlying epidermis. Mucin deposition was observed within and around the tumor cell nests (Fig. 1e). In the central eosinophilic area, the lesion was composed of a proliferation of squamoid cells with nuclear atypism (Fig. 1f). The basophilic area was positive with immunostaining using anti-Ber-EP4 antibody, although eosinophilic area was negative (Fig. 1d). We diagnosed this case as BSC based on those findings. There has been no distant metastasis or local recurrence 1 year after the resection. Giacomel et al.2 reported that dermoscopic findings of BSC revealed both those of BCC such as unfocused (peripheral) arborizing vessels, ulceration, blood crusts, blue-gray blotches and pigmentation (brown dots and leaf-like areas), and those of squamous cell carcinoma (SCC) such as keratin mass, white structureless structure, blood spots in the keratin masses and dotted, linear, irregular hairpin vessels. In our case, dermoscopic examination also exhibited the findings compatible with both BCC and SCC as mentioned above. Retrospectively, dermoscopy revealed combination of BCC and SCC in our case. Those findings are the characteristics of BSC as Giacomel et al. reported.2 Immunohistochemical examination using Ber-EP4 has been reported to be useful in diagnosing BCC.3 In BSC, several reports mentioned the usefulness of such a method,4,5 however, detail of distribution of Ber-EP4-positive cells has not been reported. Our case clearly exhibited the loss of immunoreactivity
Correspondence: Shin-ichi Ansai, M.D., Ph.D., Division of Dermatology, Nippon Medical School Musashi Kosugi Hospital, 1-396 Kosugi-cho, Nakahara-ku, Kawasaki, Kanagawa 211-8533, Japan. Email: [email protected]
1102
© 2015 Japanese Dermatological Association
LETTERS TO THE EDITOR
Use of propranolol on a nasal hemangioma in an extremely low birthweight premature infant Dear Editor, We describe an extremely low birthweight infant of 700 g of 25 weeks’ gestation who presented on day 28 of age (11 weeks corrected) with infantile hemangioma (IH) on his nose, and was successfully treated with oral propranolol. The lesion was first noticed as a small red spot but had grown quickly, covering the entire nose on day 56 (7 weeks corrected) (Fig. 1a). Although clinically indicated for propranolol treatment, it was not commenced because of the potential side-effects on the cardiorespiratory system of this 1000-g infant. On day 90 (2 weeks corrected) at a bodyweight of 1500 g, with normal general condition, the baby was finally commenced on oral propranolol at 0.25 mg/kg per day b.i.d. in the neonatal intensive care unit. During the first 2 weeks of treatment, the heart rate dropped slightly between 140– 155 b.p.m. to 110–120 b.p.m. while blood pressure and serum glucose levels remained stable. Then, the dose was gradually increased to 2 mg/kg (full dose) per day on 5 months (Fig. 1f), and his IH had lightened and flattened over time (Fig. 1b–d). When the patient was 2 years old, the tumor had disappeared, leaving no observable nasal deformity and no defect in the underlying nasal cartilage (Fig. 1e). Therefore, propranolol was gradually stopped over 3 weeks; the baby is still in follow up and no recurrence was found at the last visit when he was 2 years and 3 months of age. At this time, the baby was 83 cm in height, 11 kg in weight, 48 cm in head circumference and intelligence development was normal. Thus, there was no obvious growth and development abnormality at the last visit. For preterm infants, evidence for IH treatment is lacking despite their high incidence.1 Despite propranolol becoming the mainstay of therapy in IH since first reported in 2008,2 case reports of extremely low birthweight infants with IH who were treated with propranolol at the age younger than 0 weeks’ corrected age are rare.3,4 This case is of interest because propranolol was started at an age younger than 0 weeks’ corrected age in an extremely Figure 1. (a) Fifty-six days (7 weeks corrected) of age, hemangioma on the nose. (b) Three months (2 weeks corrected), the initial day of propranolol therapy, hemangioma covered his full nose. (c) Four months (2 weeks corrected), after 1 month of propranolol therapy, hemangioma was lightened and flattened. (d) Fifteen months (11.5 months corrected), after 12 months of propranolol therapy, marked improvement in his hemangioma. (e) Two years old (20.5 months corrected), after 21 months of propranolol therapy, hemangioma had disappeared. (f) Patient’s medication dose, age and corrected age.
(a)
(b)
(c)
(d)
(e)
(f)
Correspondence: Lin Ma, M.D., Department of Dermatology, Beijing Children’s Hospital, Capital Medical University, 56 South Li Shi Road, Xi Cheng District, Beijing 100045, China. Email: [email protected]
© 2015 Japanese Dermatological Association
1101
Letters to the Editor
low birthweight. In order to avoid side-effects, we evaluated the general condition of the patient before treatment, monitored him closely when propranolol was given in the first 2 months, and the drug was administrated at 0.125 mg/kg per dose from the beginning. Although the patient had experienced myocardial enzyme level increase which was treated by oral fructose sodium diphosphate solution by our pediatric cardiologist, the side-effect was believed to be temporary and would not cause long-term adverse influence on the patient; thus, propranolol was used successfully without obvious sideeffects. In this case, a cautious and gradual approach in using propranolol for our preterm and low birthweight neonate with IH was adopted. A low initiation dose, gradual dose increase and close monitoring were employed and the patient was successfully treated.
CONFLICT OF INTEREST:
Li LI, Lin MA Department of Dermatology, Beijing Children’s Hospital, Capital Medical University, Beijing, China doi: 10.1111/1346-8138.13029
REFERENCES 1 Goelz R, Poets CF. Incidence and treatment of infantile haemangioma in preterm infants. Arch Dis Child Fetal Neonatal Ed 2015; 100 (1): F85–F91. aute -Labre ze C, Dumas de la Roque E, Hubiche T et al. Proprano2 Le lol for severe hemangiomas of infancy [J]. N Engl J Med 2008; 358 (24): 2649–2651. 3 Erbay A, Sarialioglu F, Malbora B et al. Propranolol for infantile hemangiomas: a preliminary report on efficacy and safety in very low birth weight infants. Turk J Pediatr 2010; 52: 450–456. 4 Frost G, Relic J. Dangers of propranolol in preterm infants. Australas J Dermatol 2013; 54: 237–238.
None.
Case of basosquamous carcinoma: Dermoscopic and immunohistochemical findings Dear Editor, Basosquamous carcinoma (BSC) is a rare subtype of basal cell carcinoma (BCC) that exhibits squamoid cell differentiation and has worse prognosis than other types of BCC.1 There have been only few reports about dermoscopic and immunohistochemical findings of BSC. Here, we report a case of BSC on the leg with interesting dermoscopic and immunohistochemical findings. A 76-year-old man visited our outpatient clinic complaining of an expanding ulcer on his leg for 2 months (Fig. 1a). Physical examination at first visit revealed a 20 mm 9 12 mm skin ulcer with peripheral pigmentation on the anterior aspect of the left lower leg. Dermoscopic examination exhibited multifocal blue-gray pigmentation inside of an extensive ulcer and blue-gray structures in a radial fashion at the periphery of the ulcer (Fig. 1b). Many dotted, glomerular and linear blood vessels were observed in the ulcerated area. White fibrous area and thin scales were also observed at the periphery of the ulcer (Fig. 1b). A pathological diagnosis of the biopsy specimen taken from the lesion was BCC or BSC. The lesion was excised with a 5-mm margin. Pathological findings of the excised specimen revealed a nodular lesion in the dermis with ulceration at the low-power view (Fig. 1c). The peripheral tumor cell nests showed basophilic staining, while the central area showed eosinophilic staining. There was continuity between those two areas. In the basophilic area, neoplastic cells with basaloid features formed nests with nuclear palisad-
ing at the periphery and connected to the overlying epidermis. Mucin deposition was observed within and around the tumor cell nests (Fig. 1e). In the central eosinophilic area, the lesion was composed of a proliferation of squamoid cells with nuclear atypism (Fig. 1f). The basophilic area was positive with immunostaining using anti-Ber-EP4 antibody, although eosinophilic area was negative (Fig. 1d). We diagnosed this case as BSC based on those findings. There has been no distant metastasis or local recurrence 1 year after the resection. Giacomel et al.2 reported that dermoscopic findings of BSC revealed both those of BCC such as unfocused (peripheral) arborizing vessels, ulceration, blood crusts, blue-gray blotches and pigmentation (brown dots and leaf-like areas), and those of squamous cell carcinoma (SCC) such as keratin mass, white structureless structure, blood spots in the keratin masses and dotted, linear, irregular hairpin vessels. In our case, dermoscopic examination also exhibited the findings compatible with both BCC and SCC as mentioned above. Retrospectively, dermoscopy revealed combination of BCC and SCC in our case. Those findings are the characteristics of BSC as Giacomel et al. reported.2 Immunohistochemical examination using Ber-EP4 has been reported to be useful in diagnosing BCC.3 In BSC, several reports mentioned the usefulness of such a method,4,5 however, detail of distribution of Ber-EP4-positive cells has not been reported. Our case clearly exhibited the loss of immunoreactivity
Correspondence: Shin-ichi Ansai, M.D., Ph.D., Division of Dermatology, Nippon Medical School Musashi Kosugi Hospital, 1-396 Kosugi-cho, Nakahara-ku, Kawasaki, Kanagawa 211-8533, Japan. Email: [email protected]
1102
© 2015 Japanese Dermatological Association
