|
| SHORT COMMUNICATIONS |
|
|
|
| Year : 2000 | Volume
: 66
| Issue : 3 | Page : 139-140 |
Reticulate Acropigmentation of Dohi - a Report of Two Unrelated Families
Rajeev Sharma, Mithilesh Chandra
Correspondence Address:
Rajeev Sharma
 Source of Support: None, Conflict of Interest: None  | Check |
PMID: 20877056 
Reticulate acropigmentation of Dohi is a rarely reported disorder especially in the Indian literature. We report our findings in eight cases belonging to two different families. It will create awareness about its occurrence in our country.
Keywords: Acropigmentation Dohi, Pigmentation
How to cite this article:
Sharma R, Chandra M. Reticulate Acropigmentation of Dohi - a Report of Two Unrelated Families. Indian J Dermatol Venereol Leprol 2000;66:139-40 |
How to cite this URL:
Sharma R, Chandra M. Reticulate Acropigmentation of Dohi - a Report of Two Unrelated Families. Indian J Dermatol Venereol Leprol [serial online] 2000 [cited 2021 Jan 17];66:139-40. Available from: https://www.ijdvl.com/text.asp?2000/66/3/139/4898 |
| Introduction | |  |
Reticulate acropigmentation of Dohi (symmetrical dyschromatosis of the extremities or dyschromatosis symmetrica hereditaria) is a condition uncommonly encountered in people outside Japan. [1] It is characterized by presence of both hyperpigmented and hypopigmented macules mixed in a reticulate pattern on the extremities. [2] Though it was first described in patients from Japan in 1924, [3] only a few cases have been reported from other parts of the world. [1],[2],[3],[4],[5],[6] Eight cases belonging to two unrelated Indian families, who had typical features of reticulate acropigmentation of Dohi, are being reported.
Case Report
Casel: A 6-year-old boy was brought for altered pigmentation on the hands and feet since the age of 3 1/2 years. The lesions initially appeared as hyperpigmented macules on the feet. Subsequently similar lesions were noticed on the hands 6 months later. Hypopigmented macules appeared in between the hyperpigmented lesions in the next few years. On examination he had a combination of hypopigmented and hyperpigmented macules arranged in a reticulate pattern on the dorsa of the hands and feet. The sides of the soles and palms, fingers and toes were also similarly affected. There were no associated pits or breaks in the ridges on the palms and soles. There were multiple hyperpigmented macules, 1 to 2 mm in size, on the central part of the face. A skin biopsy taken from the hypopigmented lesion showed elongation of rete ridges with focal loss of melanin from the basal layer without pigmentary incontinence.
Case 2: A 26-year old woman, mother of case 1 had similar clinical presentation with similar age at onset and histological features. The extent of her involvement was more than her son . She had involvement of the knees and elbows in addition to hands and feet. Hyperpigmented macules like son,where present on the central part of the face. Four other members, maternal grandmother, maternal great grandmother and two maternal second cousins, were found to be similarly affected on examination.
Case 3: A 38- year- old man presented with itchy rash all over the body including the genitalia. He was diagnosed as a case of scabies. During examination, he was found to have hyperpigmented macules arranged in a reticulate manner on the dorsa as well as the sides of hands and feet, knees and elbows with intermingled hypopigmented macules.
Histological examination of a skin biopsy involving both hypopigmented and hyperpigmented lesions showed features similar to Case 1. His 23year-old younger sister was similarly affected. No other member of the family had similar lesions.
| Discussion | | |
Reticulate acropigmentation of Dohi is accepted to be an autosomal dominant condition, [5],[6],[7] though sporadic cases are known to occur. [1] It has a characteristic appearance in every family with hyperpigmented and hypopigmented macules forming a reticulate pattern on the dorsa of the hands and feet, varying only in its intensity and the extent of involvement. The onset is normally reported to occur in the first decade, [1],[2],[3],[4],[5],[6],[7] but occasionally onset at later age may occur. [1] The lesions may gradually spread proximally to involve sides of the neck and the supra-clavicular region. [1] None of our patients had involvement of the neck or supra-clavicular region. None of our patients had atrophic pigmented lesions, which have been reported earlier [1],[2],[3],[4],[5],[6],[7],[8],[9]
Reticulate acropigmentation of Dohi is mostly reported in the Japanese. [1] Few cases have been described from Europe and India. [1],[2],[3],[4],[5],[6]
Treatment of reticulate acropigmentation of Dohi is difficult. Only split-skin grafting had been reported to benefit the patients but is not normally carried out, as it is not accepTable to most of them. Camouflage is the most reasonable approach if accepTable to the patient. The exact pathomechanism of this disease is not yet understood though a resemblance to reptilian skin, [5] and an evolutionary process mediated by embryonic neural reflexes which is expressed only in genetically predisposed individuals has been suggested. [10]
The occurrence of acropigmentation of Dohi in two members of the same generation in the second family supports an autosomal recessive mode of transmission though a paradominant trait may be responsible.
The presence of lentigenes on the central part of the face, in members of the first family may be an example of co-incidental occurrence of centrofacial lentiginosis with acropigmentation of Dohi.
| References | | |
| 1. | Ostlere LS, Rantavel RC, Lawlor F, et al. Reticulate acropigmentation of Dohi. Clin Exp Dermatol 1995;20:477-479.  |
| 2. | Griffiths WAD. Reticulate pigmentary disorders. Clin Exp Dermatol 1984 ; 9 : 439 - 450. |
| 3. | Komaya G. Symmetrische Pigmentanomile der Extremitaten. Arch Dermatol Syph 1924 ; 147 : 389 - 393. |
| 4. | Bleehen SS, Ebling FJG, Champion RH. Disorders of skin colour. In:Champion RH, Burton JL, Ebling FJG, Eds.Textbook of Dermatology,5th edn. Oxford: Blackwell Scientfic Publications 1992;1561. |
| 5. | Gharpuray MB, Tolat SN, Patwardhan SP. Dyschromatosis:Its occurrence in two Indian families with unusual features. Int J Dermatol 1994 ; 33 : 391 - 392. |
| 6. | Dhar S, Kanwar AI, Jebraili R, et al. Spectrum of reticulate flexural and acral pigmentary disorders in Northern India. J Dermatol 1994; 21:598-603. |
| 7. | Kim NI, Park SY, Youn II, et al. Dyschromatosis symmetrica hereditaria affecting two families. Korean J Dermatol 1980;18:585-589. |
| 8. | Dohi K. Dyschromatosis symmetrica hereditaria affecting two families.Korean J Dermatology.Tokyo:University of Tokyo press. 1982: 142. |
| 9. | Gartmann. Akropigmentation symmetrica Dohi mit angeborenen Fundus Verandergungen be! Mutter and Tochter. Dermatol Wochneschr 1952 ; 125 : 534 - 535. |
| 10. | WhimsterW. Symmetry in Dermatology. In Borrie P, ed. Modern Trends in Dermatology.4th ed. London : Butterworths, 1971 : 20. |
|
|
|
|
|
|
|
|
