ectopic nail with polydactyly

2
Norito Katoh, MD d Saburou Kishimoto, MD d Departments of Dermatology, a Internal Medicine, b and Plastic Surgery c Kyoto Second Red Cross Hospital Department of Dermatology Kyoto Prefectural University of Medicine Graduate School of Medical Science d Reprint requests: Takeshi Nara, MD Department of Dermatology Kyoto Prefectural University of Medicine Graduate School of Medical Science Kawaramachi-Hirokoji, Kamigyo-ku Kyoto 602-8566, Japan REFERENCES 1. Hillmen P, Lewis SM, Bessler M, Besseler M, Luzzatto L, Dacie J. Natural history of paroxysmal nocturnal hemoglobinuria. N Engl J Med 1995;333:1253-8. 2. Gralnick H, Vail M, McKeown L, Meryman P, Wilson O, Chu I, et al. Activated platelets in paroxysmal nocturnal hemoglobi- nuria. Br J Haematol 1995;91:697-702. 3. Sims P, Faioni E, Wieder T, Shattil S. Complement proteins C5b-9 cause release of membrane vesicles from the platelet sur- face that are enriched in the membrane receptor for coagulation factor Va and express prothrombinase activity. J Biol Chem 1988;263:18205-12. 4. Gilbert G, Sims P, Wiedmer T, Furie B, Furie BC, Shattil SJ. Platelet-derived microparticles express high affinity receptor for factor VIII. J Biol Chem 1991;266:17261-8. 5. Wiedmer H, Hall S, Ortel T, Kane W, Rosse W, Sims P. Com- plement-induced vesiculation and exposure of membrane prothrombinase sites in platelets of paroxysmal nocturnal hemoglobinuria. Blood 1993;82:1192-6. doi:10.1016/j.jaad.2005.07.049 Ectopic nail with polydactyly To the Editor: Ectopic nail is an extremely rare condition related to acquired or congenital anoma- lies. It is characterized by development of nail-like tissue in a location other than the nail bed. We observed a case of a 27-year-old man with polydac- tyly and ectopic nail (Fig 1). He was referred with keratotic projection growing vertically out of the back aspect of the proximal phalanx of the accessory toe in his left foot. This lesion had been present since the age of 10 years and he simply clipped it when- ever it grew long. On this presentation, the growth measured 8 3 8 mm at the base and 9 mm high. The surrounding skin was slightly swollen without any signs of inflammation. Besides polydactyly, no skin alteration, hand or foot deformity was evident, nor was there any evidence of previous injury. There was no history of similar abnormalities in his family members. Radiography revealed an accessory digit and an opaque structure corresponding to the ker- atotic horn. The diagnosis of ectopic nail was made after histopathologic examination of the biopsy specimen revealed a fully developed nail unit. After surgical resection of this ectopic nail, there has been no recurrence for 1 year. DISCUSSION Almost 40 cases of ectopic nail are reported in the literature, mostly in Japanese patients. 1-3 The major- ity of the cases were noted on the palmar aspect of fingers (40 localizations in 34 patients); the V digit is most commonly involved 4 (32 localizations in 26 patients), followed by the IV digit (3 cases), the I and III digits (two cases, respectively), and the II digit (one case). Toes are involved more rarely (7 local- izations in 4 patients) with 3 cases of the III digit and two cases each of the II and IV digits. They were associated with acquired or congenital growth anomalies or polydactyly in some cases. Most of the reported cases are of congenital origin. The essential feature of ectopic nail is the continuous growth of the nail plate. Although exactly unknown, various hypotheses proposed to explain the genesis of ectopic nail are: aberration in the long arm of chromosome 6, 5 presence of stray germinal cells in ectopic sites, 6 persistence of a rudimentary nail bed after regression of polydactyly, and traumatic inoc- ulation of onychocytes. 7 Although it is an uncom- mon anomaly, the cases may go unnoticed as mostly they are asymptomatic and may be confused as cutaneous horn or rudimentary digit. Tarun Narang, MD Amrinder Jit Kanwar, MD Department of Dermatology Venereology, and Leprology Postgraduate Institute of Medical Education and Research Fig 1. Close-up view of ectopic nail in this case. JAM ACAD DERMATOL DECEMBER 2005 1092 Letters

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Norito Katoh, MDd

Saburou Kishimoto, MDd

Departments of Dermatology,a

Internal Medicine,b

and Plastic Surgeryc

Kyoto Second Red Cross HospitalDepartment of Dermatology

Kyoto Prefectural University of MedicineGraduate School of Medical Scienced

Reprint requests: Takeshi Nara, MDDepartment of Dermatology

Kyoto Prefectural University of MedicineGraduate School of Medical Science

Kawaramachi-Hirokoji, Kamigyo-kuKyoto 602-8566, Japan

REFERENCES

1. Hillmen P, Lewis SM, Bessler M, Besseler M, Luzzatto L, Dacie J.

Natural history of paroxysmal nocturnal hemoglobinuria.

N Engl J Med 1995;333:1253-8.

2. Gralnick H, Vail M, McKeown L, Meryman P, Wilson O, Chu I,

et al. Activated platelets in paroxysmal nocturnal hemoglobi-

nuria. Br J Haematol 1995;91:697-702.

3. Sims P, Faioni E, Wieder T, Shattil S. Complement proteins

C5b-9 cause release of membrane vesicles from the platelet sur-

face that are enriched in the membrane receptor for coagulation

factor Va and express prothrombinase activity. J Biol Chem

1988;263:18205-12.

4. Gilbert G, Sims P, Wiedmer T, Furie B, Furie BC, Shattil SJ.

Platelet-derived microparticles express high affinity receptor

for factor VIII. J Biol Chem 1991;266:17261-8.

5. Wiedmer H, Hall S, Ortel T, Kane W, Rosse W, Sims P. Com-

plement-induced vesiculation and exposure of membrane

prothrombinase sites in platelets of paroxysmal nocturnal

hemoglobinuria. Blood 1993;82:1192-6.

doi:10.1016/j.jaad.2005.07.049

Ectopic nail with polydactyly

To the Editor: Ectopic nail is an extremely rarecondition related to acquired or congenital anoma-lies. It is characterized by development of nail-liketissue in a location other than the nail bed. Weobserved a case of a 27-year-old man with polydac-tyly and ectopic nail (Fig 1). He was referred withkeratotic projection growing vertically out of theback aspect of the proximal phalanx of the accessorytoe in his left foot. This lesion had been present sincethe age of 10 years and he simply clipped it when-ever it grew long. On this presentation, the growthmeasured 8 3 8 mm at the base and 9 mm high. Thesurrounding skin was slightly swollen without anysigns of inflammation. Besides polydactyly, no skinalteration, hand or foot deformity was evident, norwas there any evidence of previous injury. Therewas no history of similar abnormalities in his family

members. Radiography revealed an accessory digitand an opaque structure corresponding to the ker-atotic horn.

The diagnosis of ectopic nail was made afterhistopathologic examination of the biopsy specimenrevealed a fully developed nail unit.

After surgical resection of this ectopic nail, therehas been no recurrence for 1 year.

DISCUSSIONAlmost 40 cases of ectopic nail are reported in the

literature, mostly in Japanese patients.1-3 The major-ity of the cases were noted on the palmar aspect offingers (40 localizations in 34 patients); the V digit ismost commonly involved4 (32 localizations in 26patients), followed by the IV digit (3 cases), the I andIII digits (two cases, respectively), and the II digit(one case). Toes are involved more rarely (7 local-izations in 4 patients) with 3 cases of the III digit andtwo cases each of the II and IV digits. They wereassociated with acquired or congenital growthanomalies or polydactyly in some cases. Most ofthe reported cases are of congenital origin. Theessential feature of ectopic nail is the continuousgrowth of the nail plate. Although exactly unknown,various hypotheses proposed to explain the genesisof ectopic nail are: aberration in the long arm ofchromosome 6,5 presence of stray germinal cells inectopic sites,6 persistence of a rudimentary nail bedafter regression of polydactyly, and traumatic inoc-ulation of onychocytes.7 Although it is an uncom-mon anomaly, the cases may go unnoticed as mostlythey are asymptomatic and may be confused ascutaneous horn or rudimentary digit.

Tarun Narang, MDAmrinder Jit Kanwar, MD

Department of DermatologyVenereology, and Leprology

Postgraduate Institute of Medical Educationand Research

Fig 1. Close-up view of ectopic nail in this case.

J AM ACAD DERMATOL

DECEMBER 2005

1092 Letters

Reprint requests: Amrinder Jit Kanwar, MDDepartment of DermatologyVenereology, and Leprology

PGIMER, Chandigarh-160 012, India

E-mail: [email protected]

REFERENCES

1. Kikuchi I, Ono T, Ogata K. Ectopic nail: case reports. Plast

Reconstr Surg 1978;61:781-3.

2. Markinson B, Brenner AR, McGrath M. Congenital ectopic nail:

a case study. J Am Podiatr Med Assoc 1988;78:318-9.

3. Tomita K, Inoue K, Ichikawa H, Shirai S. Congenital ectopic nails.

Plast Reconstr Surg 1997;100:1497-9.

4. Iida N, Fukuya Y, Yoshitane K, Hosaka Y. A case of congenital

ectopic nails on bilateral little fingers. J Dermatol 1997;24:38-42.

5. Kalisman M, Goldberg R, Ship AG. Dorsal skin and fingernails on

the volar aspect of the hand: an unusual anatomic deformity.

Plast Reconstr Surg 1982;69:6946.

6. Egawa T. Congenital claw-like fingers and toes: case report of

two siblings. Plast Reconstr Surg 1977;59:569-74.

7. Kopera D, Soyer HP, Kerl H. Ectopic calcaneal nail. J Am Acad

Dermatol 1996;35:4845.

doi:10.1016/j.jaad.2005.07.065

Rapid onset of CD81 aggressive T-celllymphoma during bexarotene therapyin a patient with Sezary syndrome

To the Editor: Bexarotene, a new retinoid X receptorligand, has been approved for the treatment of

Fig 1. Immunohistologic stainings at first presentation (CD41 cells, 80% positive [A]; CD81

cells, 10% positive [B]). Immunohistologic transformation after 6 months of bexarotene therapy(CD41 cells, 0% positive [C]; CD81 cells, 90% positive [D]). (A and C, CD4 stain, originalmagnification 340; B and D, CD8 stain, original magnification 340.)

J AM ACAD DERMATOL

VOLUME 53, NUMBER 6

Letters 1093