The evaluation of IgG4 and IgG expression in cutaneous Rosai-Dorfman disease

Zou, Puyu; Zhan, Yi; Xue, Ruzeng; Liu, Yu; Zhang, Guiying

doi:10.1016/j.abd.2022.07.010

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Original Article • An. Bras. Dermatol. 98 (5) • Sep-Oct 2023 • https://doi.org/10.1016/j.abd.2022.07.010 copy

The evaluation of IgG4 and IgG expression in cutaneous Rosai-Dorfman disease^☆ ⋆ Study conducted at the Department of dermatology, Second Xiangya Hospital, Central South University, Changsha, China.

Authorship SCIMAGO INSTITUTIONS RANKINGS

Abstract

Objective

The authors investigated the expression of IgG4 and IgG in cutaneous Rosai-Dorfman Disease (CRDD) to further improve the understanding of this disease.

Methods

The authors retrospectively reviewed the clinicopathological features of 23 CRDD patients. The authors diagnosed CRDD by the presence of emperipolesis and immunohistochemical (IHC) staining of histiocytes consisting of S-100(+)/CD68(+)/CD1a(-) cells. The expressions of IgG and IgG4 in cutaneous specimens were assessed by IHC (EnVision) and quantitatively calculated by a medical image analysis system.

Results

All 23 patients, including 14 males and 9 females, were confirmed to have CRDD. Their ages ranged from 17 to 68 years (mean 47.91 ± 14.16). The most frequently affected skin regions were the face, followed by the trunk, ears, neck, limbs, and genitals. In 16 of these cases, the disease presented as a single lesion. IHC staining of sections showed that IgG was positive (≥ 10 cells/High-Power Field [HPF]) in 22 cases, while IgG4 was positive (≥ 10 cells/HPF) in 18 cases. Moreover, the IgG4/IgG proportion ranged from 1.7% to 85.7% (mean 29.50 ± 24.67%, median 18.4%) in the 18 cases.

Study limitations

In the majority of studies, as well as in the current study, the design. RDD is a rare disease, so the sample size is small. In the next studies to come, the authors will expand the sample for multi-center verification and in-depth study.

Conclusion

The positive rates of IgG4 and IgG and the IgG4/IgG ratio assessed through IHC staining may be important in understanding the pathogenesis of CRDD.

Keywords
Skin; Histiocytosis; Immunoglobulin G; Immunoglobulin G4-related disease

Case^a a Case 1 to case 12 were from Second Xiangya Hospital of Central South University and case 13 to case 23 were from Dermatology Hospital of Southern Medical University.	Age	Gender^b b M, Male; F, Female.	Duration^c c y, Years; m, Months; d, Days.	Skin lesion description	Quantity	Pain	Comorbidity
1	57	M	20d	1 cm × 1 cm Upper right lip red plaque	1	No	None
2	23	F	6m	5 cm × 4 cm Upper right limb brown patch	1	No	None
3	53	M	1y	1 cm × 1 cm Right chest dull-red patch	1	No	None
4	59	F	1y	10 cm × 10 cm Right chest red plaque	1	No	None
5	28	F	6m	0.3 cm × 0.2 cm Right breast red papule	1	Yes	None
6	52	M	6m	15 cm × 10 cm Right lower back Red plaque	1	Yes	None
7	17	M	6y	3 cm × 2 cm Upper left limb red plaque	1	No	None
8	50	M	3m	3 cm × 2.5 cm Left cheek red patch	1	No	None
9	61	M	1y	Generalized skin eruption with red patch papule	many	No	None
10	57	F	6m	0.5 cm × 0.4 cm Right chest brown papule	1	No	None
11	38	F	2m	0.8 cm × 0.8 cm Left calf dull-red papule	1	No	None
12	57	M	4m	0.7 cm × 0.6 cm Left chest red papule	1	No	None
13	68	M	1y	2 cm × 1 cm Left temporal red plaque	1	No	None
14	61	M	1m	2 cm × 2 cm Plaque in chest and abdomen	3	No	None
15	63	F	2y	3 cm × 3 cm Left thigh dull-red patch	1	No	None
16	20	M	50d	1 cm × 1 cm Chest and abdomen plaques	5	No	None
17	59	F	7m	1 cm × 1 cm Left cheek red plaque	1	No	None
18	40	M	2y	3 cm × 3 cm Right thigh dark brown patch	1	No	None
19	54	M	2y	Generalized skin eruption with brown papules	1	No	None
20	39	F	2m	1 cm × 1.5 cm Left cheek red plaque	many	No	None
21	42	F	6m	2 cm × 1 cm Left cheek red plaque	2	No	None
22	52	M	6m	0.3 cm × 0.3 cm Right thigh papules	2	No	None
23	52	M	1y	3 cm × 3 cm Left sub-axillary mass	2	No	None

Case^a a Case 1 to case 12 were from Second Xiangya Hospital of Central South University and case 13 to case 23 were from Dermatology Hospital of Southern Medical University.	Features specific for RDD			Antibodies examined may contribute to diagnosis
	S100^b b The immunohistochemical expression of S100, CD68 and CD1a were collected from existing pathologic reports.	CD68^b b The immunohistochemical expression of S100, CD68 and CD1a were collected from existing pathologic reports.	CD1a^b b The immunohistochemical expression of S100, CD68 and CD1a were collected from existing pathologic reports.	IgG4+/HPF^c c Three high-power fields (400×) with the maximum number of positive cells were counted, and an average number per HPF was calculated and presented in the Table 2.	IgG+/HPF^c c Three high-power fields (400×) with the maximum number of positive cells were counted, and an average number per HPF was calculated and presented in the Table 2.	IgG4/IgG%^c c Three high-power fields (400×) with the maximum number of positive cells were counted, and an average number per HPF was calculated and presented in the Table 2.
1	+	+	−	46	81	56.8%
2	+	+	−	33	40	82.5%
3	+	+	−	11	65	16.9%
4	+	+	−	35	90	38.9%
5	+	+	−	20	220	9.1%
6	+	+	−	101	213	47.4%
7	+	+	−	72	84	85.7%
8	+	+	−	10	265	3.8%
9	+	+	−	1	4	25%
10	+	+	−	9	49	18.4%
11	+	+	−	1	46	2.2%
12	+	+	−	12	12	13.3%
13	+	+	−	10	190	5.3%
14	+	+	−	71	160	44.3%
15	+	+	−	69	116	59.5%
16	+	+	−	19	79	24.1%
17	+	+	−	6	178	3.4%
18	+	+	−	18	182	9.9%
19	+	+	−	65	118	55.1%
20	+	+	−	2	120	1.7%
21	+	+	−	78	218	35.8%
22	+	+	−	10	48	20.8%
23	+	+	−	21	117	17.9%

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E-mail: revista@sbd.org.br

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Study conducted at the Department of dermatology, Second Xiangya Hospital, Central South University, Changsha, China.

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The evaluation of IgG4 and IgG expression in cutaneous Rosai-Dorfman disease☆ ⋆ Study conducted at the Department of dermatology, Second Xiangya Hospital, Central South University, Changsha, China.

Abstract

Objective

Methods

Results

Study limitations

Conclusion

The evaluation of IgG4 and IgG expression in cutaneous Rosai-Dorfman disease^☆ ⋆ Study conducted at the Department of dermatology, Second Xiangya Hospital, Central South University, Changsha, China.