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I have been looking at previous posts trying to come up with an answer and was

not able to find what I needed; I am sure this has already been discussed

sometime in the history of this group so I apologize ahead of time for asking

the question again.

Suzanne is getting an ERCP on Monday because of significant blockage of her main

bile duct. Dr. Shneider from Childrens of Pgh showed me her MRCP from last year

and it was the first time I realized that the bile duct that was affected was

the main bile duct, not the smaller " branches " (using Dr. Shneider's

description). I have read that small duct PSC is a slower progressing type. What

about what I am describing in Suzanne?

Thank you,

LINDA

(Mom of Suzanne, Indeterminant Colitis, 1/04; PSC 3/04; j-pouch 2/07)

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Dear ;

I can recommend this article as a source for some of the information that you

are looking for:

LaRusso NF, Shneider BL, Black D, Gores GJ, SP, Doo E, Hoofnagle JH 2006

Primary sclerosing cholangitis: summary of a workshop. Hepatology 44: 746-764

http://www.niddk.nih.gov/fund/other/PSCSept2006.pdf

" PSC typically affects both intra- and extra-hepatic bile ducts. " Small duct

PSC " refers to disease that affects bile ducts that are too small to be

identified by endoscopic retrograde cholangiopancreatography (ERCP).7,8 Among

adults with PSC, 75% have involvement of both small and large ducts, 15% small

ducts only, and 10% large ducts only. Small duct PSC typically presents with

insidious onset of symptoms or with end-stage liver disease. Episodes of

cholangitis are rare. Small duct PSC can be associated with IBD, has a more

favorable prognosis, and is less likely than large duct PSC to lead to

cholangiocarcinoma.9

Many patients with PSC have IBD as well, typically ulcerative colitis and less

commonly Crohn's disease with colonic involvement.10 IBD is diagnosed before PSC

in 75% of cases and afterward in the remainder. Indeed, the diagnosis of IBD may

not become evident until after liver

transplantation for end-stage PSC; and conversely, PSC may not become apparent

in patients with ulcerative colitis until after colectomy. There is little or no

correlation between the severity of PSC and that of the associated IBD.

Furthermore, therapy of IBD has little effect on the course of PSC, and vice

versa. Other less common disease

associations are thyroiditis, ankylosing spondylitis, and celiac disease.

PSC with autoimmune hepatitis-like features has been referred to as autoimmune

cholangitis.11,12 These patients usually present with high serum alanine

aminotransferase (ALT) levels, modest or no elevations in serum alkaline

phosphatase, high titers of antinuclear (ANA) and antismooth muscle antibodies

(SMA) and liver histology typical

of autoimmune hepatitis. Corticosteroid therapy may lead to improvements in

symptoms and liver enzyme abnormalities. Eventually, however, patients become

resistant to therapy and serum enzymes rise again, with prominent elevations in

alkaline phosphatase and gamma

glutamyl transpeptidase (GGT). Thus, PSC is a heterogeneous

condition that can present with distinct clinical patterns that may have

important implications for pathogenesis, prognosis and therapy. "

So, I think they are possibly distinguishing here between small-duct PSC,

large-duct PSC, small-duct + large-duct PSC, and PSC/AIH (autoimmune

cholangitis).

Perhaps these should also be sub-divided further in terms of whether the patient

has celiac disease, ulcerative colitis, Crohn's disease or no IBD at all?

Other types of sclerosing cholangitis (SC) (not discussed to any extent in this

article, but possibly relevant to understanding pathogenesis (?)) include:

Segmental PSC associated with Sjogren's syndrome:

Guerrero P, M, Conde JM, Castro M, MC, Castilla L, Guillen P

1991 Primary segmental sclerosing cholangitis associated with Sjogren's

syndrome. Rev. Esp. Enferm. Dig. 79: 363-366.

http://www.ncbi.nlm.nih.gov/pubmed/1867927

Neonatal sclerosing cholangitis:

Hadj-Rabia S, Baala L, Vabres P, Hamel-Teillac D, Jacquemin E, Fabre M, Lyonnet

S, De Prost Y, Munnich A, Hadchouel M, Smahi A 2004 Claudin-1 gene mutations in

neonatal sclerosing cholangitis associated with ichthyosis: a tight junction

disease. Gastroenterology 127: 1386-1390.

http://www.ncbi.nlm.nih.gov/pubmed/15521008

(P)SC associated with hyperimmunoglobulin M immunodeficiency:

DiPalma JA, Strobel CT, Farrow JG 1986 Primary sclerosing cholangitis associated

with hyperimmunoglobulin M immunodeficiency (dysgammaglobulinemia).

Gastroenterology 91: 464-468.

http://www.ncbi.nlm.nih.gov/pubmed/3721129

(P)SC associated with isolated IgA deficiency:

Wagner A, Eichmann D 1989 Primary sclerosing cholangitis in isolated IgA

deficiency. Schweiz. Med. Wochenschr. 119: 835-838.

(P)SC associated with Wiskott-Aldrich syndrome:

Kahn K, Sharp H, Hunter D, Kerzner B, Jessurun J, Blaese M 2001 Primary

sclerosing cholangitis in Wiskott-Aldrich syndrome. J. Pediatr. Gastroenterol.

Nutr. 32: 95-99.

http://www.ncbi.nlm.nih.gov/pubmed/11176335

(P)SC associated with Riedel's thyroiditis and/or retroperitoneal fibrosis:

De Boer WA 1993 Riedel's thyroiditis, retroperitoneal fibrosis, and sclerosing

cholangitis: diseases with one pathogenesis? Gut 34: 714.

http://www.ncbi.nlm.nih.gov/pubmed/8504980

Hatanaka H, Suzuki M, Onodera H, Ukai K, Kayaba Y, Sasaki A, Takahashi K, Kagaya

H, Kikuchi T, Oriuchi T, Asano N, Kinouchi Y, Ueno Y 2002 A case of primary

sclerosing cholangitis associated with retroperitoneal fibrosis. Nippon

Shokakibyo Gakkai Zasshi 99: 1360-1365.

http://www.ncbi.nlm.nih.gov/pubmed/12462057

SC associated with systemic lymphadenopathy:

Kazumori H, Hashimoto T, Akagi S, Adachi K, Watanabe M, Kinoshita Y 2001

Sclerosing cholangitis and systemic lymphadenopathy. J. Gastroenterol. 36:

429-432.

http://www.ncbi.nlm.nih.gov/pubmed/11428591

Hypereosinophilic sclerosing cholangitis:

al-Abdulla NA, Schulick RD, Regan F 2000 Hypereosinophilic sclerosing

cholangitis: findings using half-Fourier magnetic resonance imaging.

Hepatogastroenterology 47: 359-361.

http://www.ncbi.nlm.nih.gov/pubmed/10791189

Grauer L, Padilla VM 3rd, Bouza L, Barkin JS 1993 Eosinophilic sclerosing

cholangitis associated with hypereosinophilic syndrome. Am. J. Gastroenterol.

88: 1764-1769.

http://www.ncbi.nlm.nih.gov/pubmed/8213721

Ichikawa N, Taniguchi A, Akama H, Ishiguro H, Kurihara T, Terai C, Hara M,

Kashiwazaki S 1997 Sclerosing cholangitis associated with hypereosinophilic

syndrome. Intern. Med. 36: 561-564.

http://www.ncbi.nlm.nih.gov/pubmed/9260773

(P)SC associated with sarcoidosis:

Alam I, Levenson SD, Ferrell LD, Bass NM 1997 Diffuse intrahepatic biliary

strictures in sarcoidosis resembling sclerosing cholangitis. Case report and

review of the literature. Dig. Dis. Sci. 42: 1295-1301.

http://www.ncbi.nlm.nih.gov/pubmed/9201098

Ilan Y, Rappaport I, Feigin R, Ben-Chetrit E 1993 Primary sclerosing cholangitis

in sarcoidosis. J. Clin. Gastroenterol. 16: 326-328.

http://www.ncbi.nlm.nih.gov/pubmed/8331269

Ishak KG 1998 Sarcoidosis of the liver and bile ducts. Mayo Clin. Proc. 73:

467-472.

http://www.ncbi.nlm.nih.gov/pubmed/9581591

SC associated with cystic fibrosis:

Benett I, Salh B, Haboubi NY, Braganza JM 1989 Sclerosing cholangitis with

hepatic microvesicular steatosis in cystic fibrosis and chronic pancreatitis. J.

Clin. Pathol. 42: 466-469.

http://www.ncbi.nlm.nih.gov/pubmed/2732340

Durieu I, Pellet O, Simonot L, Durupt S, Bellon G, Durand DV, Minh VA 1999

Sclerosing cholangitis in adults with cystic fibrosis: a magnetic resonance

cholangiographic prospective study. J. Hepatol. 30: 1052-1056.

http://www.ncbi.nlm.nih.gov/pubmed/10406183

IgG4-associated (P)SC:

Bjornsson E 2008 Immunoglobulin G4-associated cholangitis. Curr. Opin.

Gastroenterol. 24: 389-394.

http://www.ncbi.nlm.nih.gov/pubmed/18408470

Erdogan D, Kloek JJ, Ten Kate FJ, Rauws EA, Busch OR, Gouma DJ, van Gulik TM

2008 Immunoglobulin G4-related sclerosing cholangitis in patients resected for

presumed malignant bile duct strictures. Br. J. Surg. 95: 727-734.

http://www.ncbi.nlm.nih.gov/pubmed/18418862

Frossard JL, Spahr L, Rubbia-Brandt L, Hadengue A 2008 Biliary, pancreatic and

systemic diseases associated with IgG4. Rev. Med. Suisse. 4: 1856-1858.

http://www.ncbi.nlm.nih.gov/pubmed/18831404

Iida Y, Onitsuka A, Katagiri Y 2009 Immunoglobulin G4-related sclerosing

cholangitis without pancreatic involvement. Dig. Surg. 26: 117-118.

http://www.ncbi.nlm.nih.gov/pubmed/19262061

(P)SC associated with autoimmune pancreatitis:

Deshpande V, Mino-Kenudson M, Brugge W, Lauwers GY 2005 Autoimmune pancreatitis:

more than just a pancreatic disease? A contemporary review of its pathology.

Arch. Pathol. Lab. Med. 129: 1148-1154.

http://www.ncbi.nlm.nih.gov/pubmed/16119989

Eerens I, Vanbeckevoort D, Vansteenbergen W, Van Hoe L 2001 Autoimmune

pancreatitis associated with primary sclerosing cholangitis: MR imaging

findings. Eur. Radiol. 11: 1401-1404.

http://www.ncbi.nlm.nih.gov/pubmed/11519549

Hamano H, Kawa S, Uehara T, Ochi Y, Takayama M, Komatsu K, Muraki T, Umino J,

Kiyosawa K, Miyagawa S 2005 Immunoglobulin G4-related lymphoplasmacytic

sclerosing cholangitis that mimics infiltrating hilar cholangiocarcinoma: part

of a spectrum of autoimmune pancreatitis? Gastrointest. Endosc. 62: 152-157.

http://www.ncbi.nlm.nih.gov/pubmed/15990840

SC associated with Langerhans cell histiocytosis:

Braier J, Ciocca M, Latella A, de Davila MG, Drajer M, Imventarza O 2002

Cholestasis, sclerosing cholangitis, and liver transplantation in Langerhans

cell histiocytosis. Med. Pediatr. Oncol. 38: 178-182.

http://www.ncbi.nlm.nih.gov/pubmed/11836717

Gey T, Bergoin C, Just N, Paupard T, Cazals-Hatem D, Xuan KH, Tavernier JY,

Wallaert B 2004 Langerhans cell histiocytosis and sclerosing cholangitis in

adults. Rev. Mal. Respir. 21: 997-1000.

http://www.ncbi.nlm.nih.gov/pubmed/15622348

and this is by no means a complete list!

Best regards,

Dave

(father of (23); PSC 07/03; UC 08/03)

Link to comment
Share on other sites

Guest guest

Dear ;

I can recommend this article as a source for some of the information that you

are looking for:

LaRusso NF, Shneider BL, Black D, Gores GJ, SP, Doo E, Hoofnagle JH 2006

Primary sclerosing cholangitis: summary of a workshop. Hepatology 44: 746-764

http://www.niddk.nih.gov/fund/other/PSCSept2006.pdf

" PSC typically affects both intra- and extra-hepatic bile ducts. " Small duct

PSC " refers to disease that affects bile ducts that are too small to be

identified by endoscopic retrograde cholangiopancreatography (ERCP).7,8 Among

adults with PSC, 75% have involvement of both small and large ducts, 15% small

ducts only, and 10% large ducts only. Small duct PSC typically presents with

insidious onset of symptoms or with end-stage liver disease. Episodes of

cholangitis are rare. Small duct PSC can be associated with IBD, has a more

favorable prognosis, and is less likely than large duct PSC to lead to

cholangiocarcinoma.9

Many patients with PSC have IBD as well, typically ulcerative colitis and less

commonly Crohn's disease with colonic involvement.10 IBD is diagnosed before PSC

in 75% of cases and afterward in the remainder. Indeed, the diagnosis of IBD may

not become evident until after liver

transplantation for end-stage PSC; and conversely, PSC may not become apparent

in patients with ulcerative colitis until after colectomy. There is little or no

correlation between the severity of PSC and that of the associated IBD.

Furthermore, therapy of IBD has little effect on the course of PSC, and vice

versa. Other less common disease

associations are thyroiditis, ankylosing spondylitis, and celiac disease.

PSC with autoimmune hepatitis-like features has been referred to as autoimmune

cholangitis.11,12 These patients usually present with high serum alanine

aminotransferase (ALT) levels, modest or no elevations in serum alkaline

phosphatase, high titers of antinuclear (ANA) and antismooth muscle antibodies

(SMA) and liver histology typical

of autoimmune hepatitis. Corticosteroid therapy may lead to improvements in

symptoms and liver enzyme abnormalities. Eventually, however, patients become

resistant to therapy and serum enzymes rise again, with prominent elevations in

alkaline phosphatase and gamma

glutamyl transpeptidase (GGT). Thus, PSC is a heterogeneous

condition that can present with distinct clinical patterns that may have

important implications for pathogenesis, prognosis and therapy. "

So, I think they are possibly distinguishing here between small-duct PSC,

large-duct PSC, small-duct + large-duct PSC, and PSC/AIH (autoimmune

cholangitis).

Perhaps these should also be sub-divided further in terms of whether the patient

has celiac disease, ulcerative colitis, Crohn's disease or no IBD at all?

Other types of sclerosing cholangitis (SC) (not discussed to any extent in this

article, but possibly relevant to understanding pathogenesis (?)) include:

Segmental PSC associated with Sjogren's syndrome:

Guerrero P, M, Conde JM, Castro M, MC, Castilla L, Guillen P

1991 Primary segmental sclerosing cholangitis associated with Sjogren's

syndrome. Rev. Esp. Enferm. Dig. 79: 363-366.

http://www.ncbi.nlm.nih.gov/pubmed/1867927

Neonatal sclerosing cholangitis:

Hadj-Rabia S, Baala L, Vabres P, Hamel-Teillac D, Jacquemin E, Fabre M, Lyonnet

S, De Prost Y, Munnich A, Hadchouel M, Smahi A 2004 Claudin-1 gene mutations in

neonatal sclerosing cholangitis associated with ichthyosis: a tight junction

disease. Gastroenterology 127: 1386-1390.

http://www.ncbi.nlm.nih.gov/pubmed/15521008

(P)SC associated with hyperimmunoglobulin M immunodeficiency:

DiPalma JA, Strobel CT, Farrow JG 1986 Primary sclerosing cholangitis associated

with hyperimmunoglobulin M immunodeficiency (dysgammaglobulinemia).

Gastroenterology 91: 464-468.

http://www.ncbi.nlm.nih.gov/pubmed/3721129

(P)SC associated with isolated IgA deficiency:

Wagner A, Eichmann D 1989 Primary sclerosing cholangitis in isolated IgA

deficiency. Schweiz. Med. Wochenschr. 119: 835-838.

(P)SC associated with Wiskott-Aldrich syndrome:

Kahn K, Sharp H, Hunter D, Kerzner B, Jessurun J, Blaese M 2001 Primary

sclerosing cholangitis in Wiskott-Aldrich syndrome. J. Pediatr. Gastroenterol.

Nutr. 32: 95-99.

http://www.ncbi.nlm.nih.gov/pubmed/11176335

(P)SC associated with Riedel's thyroiditis and/or retroperitoneal fibrosis:

De Boer WA 1993 Riedel's thyroiditis, retroperitoneal fibrosis, and sclerosing

cholangitis: diseases with one pathogenesis? Gut 34: 714.

http://www.ncbi.nlm.nih.gov/pubmed/8504980

Hatanaka H, Suzuki M, Onodera H, Ukai K, Kayaba Y, Sasaki A, Takahashi K, Kagaya

H, Kikuchi T, Oriuchi T, Asano N, Kinouchi Y, Ueno Y 2002 A case of primary

sclerosing cholangitis associated with retroperitoneal fibrosis. Nippon

Shokakibyo Gakkai Zasshi 99: 1360-1365.

http://www.ncbi.nlm.nih.gov/pubmed/12462057

SC associated with systemic lymphadenopathy:

Kazumori H, Hashimoto T, Akagi S, Adachi K, Watanabe M, Kinoshita Y 2001

Sclerosing cholangitis and systemic lymphadenopathy. J. Gastroenterol. 36:

429-432.

http://www.ncbi.nlm.nih.gov/pubmed/11428591

Hypereosinophilic sclerosing cholangitis:

al-Abdulla NA, Schulick RD, Regan F 2000 Hypereosinophilic sclerosing

cholangitis: findings using half-Fourier magnetic resonance imaging.

Hepatogastroenterology 47: 359-361.

http://www.ncbi.nlm.nih.gov/pubmed/10791189

Grauer L, Padilla VM 3rd, Bouza L, Barkin JS 1993 Eosinophilic sclerosing

cholangitis associated with hypereosinophilic syndrome. Am. J. Gastroenterol.

88: 1764-1769.

http://www.ncbi.nlm.nih.gov/pubmed/8213721

Ichikawa N, Taniguchi A, Akama H, Ishiguro H, Kurihara T, Terai C, Hara M,

Kashiwazaki S 1997 Sclerosing cholangitis associated with hypereosinophilic

syndrome. Intern. Med. 36: 561-564.

http://www.ncbi.nlm.nih.gov/pubmed/9260773

(P)SC associated with sarcoidosis:

Alam I, Levenson SD, Ferrell LD, Bass NM 1997 Diffuse intrahepatic biliary

strictures in sarcoidosis resembling sclerosing cholangitis. Case report and

review of the literature. Dig. Dis. Sci. 42: 1295-1301.

http://www.ncbi.nlm.nih.gov/pubmed/9201098

Ilan Y, Rappaport I, Feigin R, Ben-Chetrit E 1993 Primary sclerosing cholangitis

in sarcoidosis. J. Clin. Gastroenterol. 16: 326-328.

http://www.ncbi.nlm.nih.gov/pubmed/8331269

Ishak KG 1998 Sarcoidosis of the liver and bile ducts. Mayo Clin. Proc. 73:

467-472.

http://www.ncbi.nlm.nih.gov/pubmed/9581591

SC associated with cystic fibrosis:

Benett I, Salh B, Haboubi NY, Braganza JM 1989 Sclerosing cholangitis with

hepatic microvesicular steatosis in cystic fibrosis and chronic pancreatitis. J.

Clin. Pathol. 42: 466-469.

http://www.ncbi.nlm.nih.gov/pubmed/2732340

Durieu I, Pellet O, Simonot L, Durupt S, Bellon G, Durand DV, Minh VA 1999

Sclerosing cholangitis in adults with cystic fibrosis: a magnetic resonance

cholangiographic prospective study. J. Hepatol. 30: 1052-1056.

http://www.ncbi.nlm.nih.gov/pubmed/10406183

IgG4-associated (P)SC:

Bjornsson E 2008 Immunoglobulin G4-associated cholangitis. Curr. Opin.

Gastroenterol. 24: 389-394.

http://www.ncbi.nlm.nih.gov/pubmed/18408470

Erdogan D, Kloek JJ, Ten Kate FJ, Rauws EA, Busch OR, Gouma DJ, van Gulik TM

2008 Immunoglobulin G4-related sclerosing cholangitis in patients resected for

presumed malignant bile duct strictures. Br. J. Surg. 95: 727-734.

http://www.ncbi.nlm.nih.gov/pubmed/18418862

Frossard JL, Spahr L, Rubbia-Brandt L, Hadengue A 2008 Biliary, pancreatic and

systemic diseases associated with IgG4. Rev. Med. Suisse. 4: 1856-1858.

http://www.ncbi.nlm.nih.gov/pubmed/18831404

Iida Y, Onitsuka A, Katagiri Y 2009 Immunoglobulin G4-related sclerosing

cholangitis without pancreatic involvement. Dig. Surg. 26: 117-118.

http://www.ncbi.nlm.nih.gov/pubmed/19262061

(P)SC associated with autoimmune pancreatitis:

Deshpande V, Mino-Kenudson M, Brugge W, Lauwers GY 2005 Autoimmune pancreatitis:

more than just a pancreatic disease? A contemporary review of its pathology.

Arch. Pathol. Lab. Med. 129: 1148-1154.

http://www.ncbi.nlm.nih.gov/pubmed/16119989

Eerens I, Vanbeckevoort D, Vansteenbergen W, Van Hoe L 2001 Autoimmune

pancreatitis associated with primary sclerosing cholangitis: MR imaging

findings. Eur. Radiol. 11: 1401-1404.

http://www.ncbi.nlm.nih.gov/pubmed/11519549

Hamano H, Kawa S, Uehara T, Ochi Y, Takayama M, Komatsu K, Muraki T, Umino J,

Kiyosawa K, Miyagawa S 2005 Immunoglobulin G4-related lymphoplasmacytic

sclerosing cholangitis that mimics infiltrating hilar cholangiocarcinoma: part

of a spectrum of autoimmune pancreatitis? Gastrointest. Endosc. 62: 152-157.

http://www.ncbi.nlm.nih.gov/pubmed/15990840

SC associated with Langerhans cell histiocytosis:

Braier J, Ciocca M, Latella A, de Davila MG, Drajer M, Imventarza O 2002

Cholestasis, sclerosing cholangitis, and liver transplantation in Langerhans

cell histiocytosis. Med. Pediatr. Oncol. 38: 178-182.

http://www.ncbi.nlm.nih.gov/pubmed/11836717

Gey T, Bergoin C, Just N, Paupard T, Cazals-Hatem D, Xuan KH, Tavernier JY,

Wallaert B 2004 Langerhans cell histiocytosis and sclerosing cholangitis in

adults. Rev. Mal. Respir. 21: 997-1000.

http://www.ncbi.nlm.nih.gov/pubmed/15622348

and this is by no means a complete list!

Best regards,

Dave

(father of (23); PSC 07/03; UC 08/03)

Link to comment
Share on other sites

Guest guest

Dear ;

I can recommend this article as a source for some of the information that you

are looking for:

LaRusso NF, Shneider BL, Black D, Gores GJ, SP, Doo E, Hoofnagle JH 2006

Primary sclerosing cholangitis: summary of a workshop. Hepatology 44: 746-764

http://www.niddk.nih.gov/fund/other/PSCSept2006.pdf

" PSC typically affects both intra- and extra-hepatic bile ducts. " Small duct

PSC " refers to disease that affects bile ducts that are too small to be

identified by endoscopic retrograde cholangiopancreatography (ERCP).7,8 Among

adults with PSC, 75% have involvement of both small and large ducts, 15% small

ducts only, and 10% large ducts only. Small duct PSC typically presents with

insidious onset of symptoms or with end-stage liver disease. Episodes of

cholangitis are rare. Small duct PSC can be associated with IBD, has a more

favorable prognosis, and is less likely than large duct PSC to lead to

cholangiocarcinoma.9

Many patients with PSC have IBD as well, typically ulcerative colitis and less

commonly Crohn's disease with colonic involvement.10 IBD is diagnosed before PSC

in 75% of cases and afterward in the remainder. Indeed, the diagnosis of IBD may

not become evident until after liver

transplantation for end-stage PSC; and conversely, PSC may not become apparent

in patients with ulcerative colitis until after colectomy. There is little or no

correlation between the severity of PSC and that of the associated IBD.

Furthermore, therapy of IBD has little effect on the course of PSC, and vice

versa. Other less common disease

associations are thyroiditis, ankylosing spondylitis, and celiac disease.

PSC with autoimmune hepatitis-like features has been referred to as autoimmune

cholangitis.11,12 These patients usually present with high serum alanine

aminotransferase (ALT) levels, modest or no elevations in serum alkaline

phosphatase, high titers of antinuclear (ANA) and antismooth muscle antibodies

(SMA) and liver histology typical

of autoimmune hepatitis. Corticosteroid therapy may lead to improvements in

symptoms and liver enzyme abnormalities. Eventually, however, patients become

resistant to therapy and serum enzymes rise again, with prominent elevations in

alkaline phosphatase and gamma

glutamyl transpeptidase (GGT). Thus, PSC is a heterogeneous

condition that can present with distinct clinical patterns that may have

important implications for pathogenesis, prognosis and therapy. "

So, I think they are possibly distinguishing here between small-duct PSC,

large-duct PSC, small-duct + large-duct PSC, and PSC/AIH (autoimmune

cholangitis).

Perhaps these should also be sub-divided further in terms of whether the patient

has celiac disease, ulcerative colitis, Crohn's disease or no IBD at all?

Other types of sclerosing cholangitis (SC) (not discussed to any extent in this

article, but possibly relevant to understanding pathogenesis (?)) include:

Segmental PSC associated with Sjogren's syndrome:

Guerrero P, M, Conde JM, Castro M, MC, Castilla L, Guillen P

1991 Primary segmental sclerosing cholangitis associated with Sjogren's

syndrome. Rev. Esp. Enferm. Dig. 79: 363-366.

http://www.ncbi.nlm.nih.gov/pubmed/1867927

Neonatal sclerosing cholangitis:

Hadj-Rabia S, Baala L, Vabres P, Hamel-Teillac D, Jacquemin E, Fabre M, Lyonnet

S, De Prost Y, Munnich A, Hadchouel M, Smahi A 2004 Claudin-1 gene mutations in

neonatal sclerosing cholangitis associated with ichthyosis: a tight junction

disease. Gastroenterology 127: 1386-1390.

http://www.ncbi.nlm.nih.gov/pubmed/15521008

(P)SC associated with hyperimmunoglobulin M immunodeficiency:

DiPalma JA, Strobel CT, Farrow JG 1986 Primary sclerosing cholangitis associated

with hyperimmunoglobulin M immunodeficiency (dysgammaglobulinemia).

Gastroenterology 91: 464-468.

http://www.ncbi.nlm.nih.gov/pubmed/3721129

(P)SC associated with isolated IgA deficiency:

Wagner A, Eichmann D 1989 Primary sclerosing cholangitis in isolated IgA

deficiency. Schweiz. Med. Wochenschr. 119: 835-838.

(P)SC associated with Wiskott-Aldrich syndrome:

Kahn K, Sharp H, Hunter D, Kerzner B, Jessurun J, Blaese M 2001 Primary

sclerosing cholangitis in Wiskott-Aldrich syndrome. J. Pediatr. Gastroenterol.

Nutr. 32: 95-99.

http://www.ncbi.nlm.nih.gov/pubmed/11176335

(P)SC associated with Riedel's thyroiditis and/or retroperitoneal fibrosis:

De Boer WA 1993 Riedel's thyroiditis, retroperitoneal fibrosis, and sclerosing

cholangitis: diseases with one pathogenesis? Gut 34: 714.

http://www.ncbi.nlm.nih.gov/pubmed/8504980

Hatanaka H, Suzuki M, Onodera H, Ukai K, Kayaba Y, Sasaki A, Takahashi K, Kagaya

H, Kikuchi T, Oriuchi T, Asano N, Kinouchi Y, Ueno Y 2002 A case of primary

sclerosing cholangitis associated with retroperitoneal fibrosis. Nippon

Shokakibyo Gakkai Zasshi 99: 1360-1365.

http://www.ncbi.nlm.nih.gov/pubmed/12462057

SC associated with systemic lymphadenopathy:

Kazumori H, Hashimoto T, Akagi S, Adachi K, Watanabe M, Kinoshita Y 2001

Sclerosing cholangitis and systemic lymphadenopathy. J. Gastroenterol. 36:

429-432.

http://www.ncbi.nlm.nih.gov/pubmed/11428591

Hypereosinophilic sclerosing cholangitis:

al-Abdulla NA, Schulick RD, Regan F 2000 Hypereosinophilic sclerosing

cholangitis: findings using half-Fourier magnetic resonance imaging.

Hepatogastroenterology 47: 359-361.

http://www.ncbi.nlm.nih.gov/pubmed/10791189

Grauer L, Padilla VM 3rd, Bouza L, Barkin JS 1993 Eosinophilic sclerosing

cholangitis associated with hypereosinophilic syndrome. Am. J. Gastroenterol.

88: 1764-1769.

http://www.ncbi.nlm.nih.gov/pubmed/8213721

Ichikawa N, Taniguchi A, Akama H, Ishiguro H, Kurihara T, Terai C, Hara M,

Kashiwazaki S 1997 Sclerosing cholangitis associated with hypereosinophilic

syndrome. Intern. Med. 36: 561-564.

http://www.ncbi.nlm.nih.gov/pubmed/9260773

(P)SC associated with sarcoidosis:

Alam I, Levenson SD, Ferrell LD, Bass NM 1997 Diffuse intrahepatic biliary

strictures in sarcoidosis resembling sclerosing cholangitis. Case report and

review of the literature. Dig. Dis. Sci. 42: 1295-1301.

http://www.ncbi.nlm.nih.gov/pubmed/9201098

Ilan Y, Rappaport I, Feigin R, Ben-Chetrit E 1993 Primary sclerosing cholangitis

in sarcoidosis. J. Clin. Gastroenterol. 16: 326-328.

http://www.ncbi.nlm.nih.gov/pubmed/8331269

Ishak KG 1998 Sarcoidosis of the liver and bile ducts. Mayo Clin. Proc. 73:

467-472.

http://www.ncbi.nlm.nih.gov/pubmed/9581591

SC associated with cystic fibrosis:

Benett I, Salh B, Haboubi NY, Braganza JM 1989 Sclerosing cholangitis with

hepatic microvesicular steatosis in cystic fibrosis and chronic pancreatitis. J.

Clin. Pathol. 42: 466-469.

http://www.ncbi.nlm.nih.gov/pubmed/2732340

Durieu I, Pellet O, Simonot L, Durupt S, Bellon G, Durand DV, Minh VA 1999

Sclerosing cholangitis in adults with cystic fibrosis: a magnetic resonance

cholangiographic prospective study. J. Hepatol. 30: 1052-1056.

http://www.ncbi.nlm.nih.gov/pubmed/10406183

IgG4-associated (P)SC:

Bjornsson E 2008 Immunoglobulin G4-associated cholangitis. Curr. Opin.

Gastroenterol. 24: 389-394.

http://www.ncbi.nlm.nih.gov/pubmed/18408470

Erdogan D, Kloek JJ, Ten Kate FJ, Rauws EA, Busch OR, Gouma DJ, van Gulik TM

2008 Immunoglobulin G4-related sclerosing cholangitis in patients resected for

presumed malignant bile duct strictures. Br. J. Surg. 95: 727-734.

http://www.ncbi.nlm.nih.gov/pubmed/18418862

Frossard JL, Spahr L, Rubbia-Brandt L, Hadengue A 2008 Biliary, pancreatic and

systemic diseases associated with IgG4. Rev. Med. Suisse. 4: 1856-1858.

http://www.ncbi.nlm.nih.gov/pubmed/18831404

Iida Y, Onitsuka A, Katagiri Y 2009 Immunoglobulin G4-related sclerosing

cholangitis without pancreatic involvement. Dig. Surg. 26: 117-118.

http://www.ncbi.nlm.nih.gov/pubmed/19262061

(P)SC associated with autoimmune pancreatitis:

Deshpande V, Mino-Kenudson M, Brugge W, Lauwers GY 2005 Autoimmune pancreatitis:

more than just a pancreatic disease? A contemporary review of its pathology.

Arch. Pathol. Lab. Med. 129: 1148-1154.

http://www.ncbi.nlm.nih.gov/pubmed/16119989

Eerens I, Vanbeckevoort D, Vansteenbergen W, Van Hoe L 2001 Autoimmune

pancreatitis associated with primary sclerosing cholangitis: MR imaging

findings. Eur. Radiol. 11: 1401-1404.

http://www.ncbi.nlm.nih.gov/pubmed/11519549

Hamano H, Kawa S, Uehara T, Ochi Y, Takayama M, Komatsu K, Muraki T, Umino J,

Kiyosawa K, Miyagawa S 2005 Immunoglobulin G4-related lymphoplasmacytic

sclerosing cholangitis that mimics infiltrating hilar cholangiocarcinoma: part

of a spectrum of autoimmune pancreatitis? Gastrointest. Endosc. 62: 152-157.

http://www.ncbi.nlm.nih.gov/pubmed/15990840

SC associated with Langerhans cell histiocytosis:

Braier J, Ciocca M, Latella A, de Davila MG, Drajer M, Imventarza O 2002

Cholestasis, sclerosing cholangitis, and liver transplantation in Langerhans

cell histiocytosis. Med. Pediatr. Oncol. 38: 178-182.

http://www.ncbi.nlm.nih.gov/pubmed/11836717

Gey T, Bergoin C, Just N, Paupard T, Cazals-Hatem D, Xuan KH, Tavernier JY,

Wallaert B 2004 Langerhans cell histiocytosis and sclerosing cholangitis in

adults. Rev. Mal. Respir. 21: 997-1000.

http://www.ncbi.nlm.nih.gov/pubmed/15622348

and this is by no means a complete list!

Best regards,

Dave

(father of (23); PSC 07/03; UC 08/03)

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