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Dowling-Degos disease 2(DDD2)

MedGen UID:
815477
Concept ID:
C3809147
Disease or Syndrome
Synonym: DDD2
 
Gene (location): POFUT1 (20q11.21)
 
Monarch Initiative: MONDO:0014130
OMIM®: 615327

Definition

Dowling-Degos disease (DDD) is an autosomal dominant genodermatosis characterized by reticular pigmentation, usually in a flexural distribution. However, generalized DDD can also occur, with numerous hypopigmented or erythematous macules and papules on the neck, chest, and abdomen. The histopathology of DDD shows characteristic thin branch-like patterns of epidermal downgrowth (summary by Li et al., 2013). Review of Reticulate Pigment Disorders Muller et al. (2012) reviewed the spectrum of reticulate pigment disorders of the skin, tabulating all reported cases of patients with Dowling-Degos disease, reticulate acropigmentation of Kitamura (RAK), reticulate acropigmentation of Dohi (DSH, RAD; 127400), Galli-Galli disease (GGD), and Haber syndrome (HS). Of 82 cases, 26 (31.7%) were clinically diagnosed as DDD, 13 (15.9%) as RAD, 11 (13.4%) as GGD, 8 (9.8%) as RAK, and 8 (9.8%) as HS; in addition, 16 (19.5%) of the cases showed overlap between DDD and RAK. Muller et al. (2012) also published photographs of an affected individual exhibiting an overlap of clinical features of DDD, GGD, RAD, and RAK. The authors noted that in reticulate disorders of the skin, the main disease entity is DDD, with a subset of cases exhibiting acantholysis (GGD), facial erythema (HS), or an acral distribution (RAD; RAK). Muller et al. (2012) concluded that all reticulate pigment diseases of the skin are varying manifestations of a single entity. For a discussion of genetic heterogeneity of reticulate pigment disorders, see 179850. [from OMIM]

Additional description

From MedlinePlus Genetics
Dowling-Degos disease is a skin condition characterized by a lacy or net-like (reticulate) pattern of abnormally dark skin coloring (hyperpigmentation), particularly in the body's folds and creases. These skin changes typically first appear in the armpits and groin area and can later spread to other skin folds such as the crook of the elbow, back of the knee, and under the breasts. Less commonly, pigmentation changes can also occur on the neck, wrists, back of the hands, face, scalp, scrotum, and vulva. These areas of hyperpigmentation typically cause no health problems.

Individuals with Dowling-Degos disease may also have dark spots (lesions) on the face and back that resemble blackheads, red bumps around the mouth that resemble acne, or pitted scars on the face similar to acne scars but with no history of acne. Fluid-filled sacs within the hair follicle (pilar cysts) may develop, most commonly on the scalp. Rarely, affected individuals have patches of skin that are unusually light in color (hypopigmented).

In rare cases, individuals with Dowling-Degos disease experience itching (pruritus) or burning sensations on the skin. These feelings can be triggered by UV light, sweating, or friction on the skin.

The pigmentation changes characteristic of Dowling-Degos disease typically begin in late childhood or in adolescence, although in some individuals, features of the condition do not appear until adulthood. New areas of hyperpigmentation tend to develop over time, and the other skin lesions tend to increase in number as well. While the skin changes associated with Dowling-Degos disease may cause distress or anxiety, they typically cause no other health problems.

A condition called Galli-Galli disease has signs and symptoms similar to those of Dowling-Degos disease. In addition to pigmentation changes, individuals with Galli-Galli disease also have a breakdown of cells in the outer layer of skin (acantholysis). Acantholysis can cause skin irritation and itchiness and lead to reddened or missing patches of skin (erosions). These conditions used to be considered two separate disorders, but Galli-Galli disease and Dowling-Degos disease are now regarded as the same condition.  https://medlineplus.gov/genetics/condition/dowling-degos-disease

Clinical features

From HPO
Phrynoderma
MedGen UID:
83101
Concept ID:
C0334013
Disease or Syndrome
A skin condition characterized by excessive development of keratin in hair follicles, resulting in rough, cone-shaped, elevated papules resulting from closure of hair follicles with a white plug of sebum.
Reticular hyperpigmentation
MedGen UID:
338832
Concept ID:
C1851972
Finding
Increased pigmentation of the skin with a netlike (reticular) pattern.
Hyperkeratotic papule
MedGen UID:
852209
Concept ID:
C2047516
Finding
A circumscribed, solid elevation of skin with no visible fluid, varying in size from a pinhead to less than 10mm in diameter at the widest point that is composed of localized hyperkeratosis (the latter may be demonstrated histopathologically).
Hypomelanotic macule
MedGen UID:
869790
Concept ID:
C4024220
Finding
Hypomelanotic macules ("ash leaf spots") are white or lighter patches of skin that may appear anywhere on the body and are caused by a lack of melanin. White ash leaf-shaped macules are considered to be characteristic of tuberous sclerosis.

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  

Recent clinical studies

Etiology

Yang A, Cheung K, Kossard S, Murrell DF
Am J Dermatopathol 2020 Jul;42(7):484-490. doi: 10.1097/DAD.0000000000001467. PMID: 31449063
Agut-Busquet E, Romaní J, Ribera M, Luelmo J
J Dermatol 2019 Feb;46(2):149-153. Epub 2018 Dec 18 doi: 10.1111/1346-8138.14722. PMID: 30561771
Lestringant GG, Masouyé I, Frossard PM, Adeghate E, Galadari IH
Dermatology 1997;195(4):337-43. doi: 10.1159/000245984. PMID: 9529553

Diagnosis

Belamarić M, Ljubojević Hadžavdić S
Acta Dermatovenerol Croat 2022 Dec;30(4):261-262. PMID: 36919394
Yang A, Cheung K, Kossard S, Murrell DF
Am J Dermatopathol 2020 Jul;42(7):484-490. doi: 10.1097/DAD.0000000000001467. PMID: 31449063
McSweeney SM, Woolf RT, Calonje E, Lewis F
Clin Exp Dermatol 2020 Mar;45(2):232-234. Epub 2019 May 3 doi: 10.1111/ced.13982. PMID: 31054170
Hohmann CB, Köche B, Bonamigo RR, Dornelles ST, Cattani CA
An Bras Dermatol 2010 Mar-Apr;85(2):241-3. doi: 10.1590/s0365-05962010000200020. PMID: 20520945
Kikuchi I, Crovato F, Rebora A
Int J Dermatol 1988 Mar;27(2):96-7. doi: 10.1111/j.1365-4362.1988.tb01279.x. PMID: 2966131

Therapy

Seitz AT, Sterz H, Strehlow V, Nagel S, Dumann K, Grunewald S, Simon JC, Kunz M
Lasers Surg Med 2019 Apr;51(4):321-324. Epub 2018 Oct 3 doi: 10.1002/lsm.23021. PMID: 30281812
Yun JH, Kim JH, Choi JS, Roh JY, Lee JR
J Cosmet Laser Ther 2013 Dec;15(6):336-9. Epub 2013 Mar 6 doi: 10.3109/14764172.2013.764437. PMID: 23464495
Kikuchi I, Crovato F, Rebora A
Int J Dermatol 1988 Mar;27(2):96-7. doi: 10.1111/j.1365-4362.1988.tb01279.x. PMID: 2966131

Clinical prediction guides

Belamarić M, Ljubojević Hadžavdić S
Acta Dermatovenerol Croat 2022 Dec;30(4):261-262. PMID: 36919394
Cario M, Pain C, Kaulanjan-Checkmodine P, Masia D, Delia G, Casoli V, Costet P, Goussot JF, Guyonnet-Duperat V, Bibeyran A, Ezzedine K, Reymermier C, Andre-Frei V, Taieb A
Pigment Cell Melanoma Res 2020 May;33(3):435-445. Epub 2019 Nov 26 doi: 10.1111/pcmr.12844. PMID: 31692218
Agut-Busquet E, Romaní J, Ribera M, Luelmo J
J Dermatol 2019 Feb;46(2):149-153. Epub 2018 Dec 18 doi: 10.1111/1346-8138.14722. PMID: 30561771
Seitz AT, Sterz H, Strehlow V, Nagel S, Dumann K, Grunewald S, Simon JC, Kunz M
Lasers Surg Med 2019 Apr;51(4):321-324. Epub 2018 Oct 3 doi: 10.1002/lsm.23021. PMID: 30281812
Lestringant GG, Masouyé I, Frossard PM, Adeghate E, Galadari IH
Dermatology 1997;195(4):337-43. doi: 10.1159/000245984. PMID: 9529553

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