U.S. flag

An official website of the United States government

Format

Send to:

Choose Destination

Stuve-Wiedemann syndrome(SJS2)

MedGen UID:
167109
Concept ID:
C0796176
Disease or Syndrome
Synonyms: Neonatal Schwartz-Jampel syndrome type 2; Schwartz-Jampel syndrome neonatal; Schwartz-Jampel syndrome type 2; Stuve-Wiedemann/Schwartz-Jampel type 2 syndrome; Stüve-Wiedemann syndrome
SNOMED CT: Stuve-Wiedemann dysplasia (254097005)
 
Related genes: LIFR, IL6ST
 
Monarch Initiative: MONDO:0031280
OMIM®: 601559
OMIM® Phenotypic series: PS601559

Definition

Stüve-Wiedemann syndrome is a severe condition characterized by bone abnormalities and dysfunction of the autonomic nervous system, which controls involuntary body processes such as the regulation of breathing rate and body temperature. The condition is apparent from birth, and its key features include abnormal curvature (bowing) of the long bones in the legs, difficulty feeding and swallowing, and episodes of dangerously high body temperature (hyperthermia).

In addition to bowed legs, affected infants can have bowed arms, permanently bent fingers and toes (camptodactyly), and joint deformities (contractures) in the elbows and knees that restrict their movement. Other features include abnormalities of the pelvic bones (the ilia) and reduced bone mineral density (osteopenia).

In infants with Stüve-Wiedemann syndrome, dysfunction of the autonomic nervous system typically leads to difficulty feeding and swallowing, breathing problems, and episodes of hyperthermia. Affected infants may also sweat excessively, even when the body temperature is not elevated, or have a reduced ability to feel pain. Many babies with this condition do not survive past infancy because of the problems regulating breathing and body temperature; however, some people with Stüve-Wiedemann syndrome live into adolescence or later.

Problems with breathing and swallowing usually improve in affected children who survive infancy; however, they still have difficulty regulating body temperature. In addition, the leg bowing worsens, and children with Stüve-Wiedemann syndrome may develop prominent joints, an abnormal curvature of the spine (scoliosis), and spontaneous bone fractures. Some affected individuals have a smooth tongue that lacks the bumps that house taste buds (fungiform papillae). Affected children may also lose certain reflexes, particularly the reflex to blink when something touches the eye (corneal reflex) and the knee-jerk reflex (patellar reflex).

Another condition once known as Schwartz-Jampel syndrome type 2 is now considered to be part of Stüve-Wiedemann syndrome. Researchers have recommended that the designation Schwartz-Jampel syndrome type 2 no longer be used. [from MedlinePlus Genetics]

Professional guidelines

PubMed

Injarie AM, Narang A, Idrees Z, Saggar AK, Nischal KK
Cornea 2012 Mar;31(3):269-72. doi: 10.1097/ICO.0b013e3182182089. PMID: 22316651

Recent clinical studies

Etiology

Chen YH, van Zon S, Adams A, Schmidt-Arras D, Laurence ADJ, Uhlig HH
J Clin Immunol 2023 Dec 22;44(1):30. doi: 10.1007/s10875-023-01603-7. PMID: 38133879Free PMC Article
Warnier H, Barrea C, Bethlen S, Schrouff I, Harvengt J
Orphanet J Rare Dis 2022 Apr 23;17(1):174. doi: 10.1186/s13023-022-02323-8. PMID: 35461249Free PMC Article
Injarie AM, Narang A, Idrees Z, Saggar AK, Nischal KK
Cornea 2012 Mar;31(3):269-72. doi: 10.1097/ICO.0b013e3182182089. PMID: 22316651
Jung C, Dagoneau N, Baujat G, Le Merrer M, David A, Di Rocco M, Hamel B, Mégarbané A, Superti-Furga A, Unger S, Munnich A, Cormier-Daire V
Clin Genet 2010 Mar;77(3):266-72. doi: 10.1111/j.1399-0004.2009.01314.x. PMID: 20447141
Gaspar IM, Saldanha T, Cabral P, Vilhena MM, Tuna M, Costa C, Dagoneau N, Daire VC, Hennekam RC
Am J Med Genet A 2008 Jul 1;146A(13):1748-53. doi: 10.1002/ajmg.a.32325. PMID: 18546280

Diagnosis

Cores ML, de Los Bueis AB
Rom J Ophthalmol 2023 Oct-Dec;67(4):412-415. doi: 10.22336/rjo.2023.66. PMID: 38239413Free PMC Article
McDermott H, Simmonds J, Thyagarajan M; Genomics England Research Consortium, Islam L, Naik S, Titheradge H
Eur J Med Genet 2023 Aug;66(8):104788. Epub 2023 Jun 7 doi: 10.1016/j.ejmg.2023.104788. PMID: 37295610
Siccha SM, Cueto AM, Parrón-Pajares M, González-Morán G, Pacio-Miguez M, Del Pozo Á, Solís M, Rodriguez-Jimenez C, Caino S, Fano V, Heath KE, García-Miñaúr S, Palomares-Bralo M, Santos-Simarro F
Am J Med Genet A 2021 Mar;185(3):856-865. Epub 2020 Dec 11 doi: 10.1002/ajmg.a.62010. PMID: 33305909
Yeşil G, Lebre AS, Santos SD, Güran O, Özahi II, Daire VC, Güran T
Am J Med Genet A 2014 Sep;164A(9):2200-5. Epub 2014 Jul 2 doi: 10.1002/ajmg.a.36626. PMID: 24988918
Gaspar IM, Saldanha T, Cabral P, Vilhena MM, Tuna M, Costa C, Dagoneau N, Daire VC, Hennekam RC
Am J Med Genet A 2008 Jul 1;146A(13):1748-53. doi: 10.1002/ajmg.a.32325. PMID: 18546280

Therapy

Chen YH, Zastrow DB, Metcalfe RD, Gartner L, Krause F, Morton CJ, Marwaha S, Fresard L, Huang Y, Zhao C, McCormack C, Bick D, Worthey EA, Eng CM, Gold J, Undiagnosed Diseases Network, Montgomery SB, Fisher PG, Ashley EA, Wheeler MT, Parker MW, Shanmugasundaram V, Putoczki TL, Schmidt-Arras D, Laurence A, Bernstein JA, Griffin MDW, Uhlig HH
J Allergy Clin Immunol 2021 Aug;148(2):585-598. Epub 2021 Mar 23 doi: 10.1016/j.jaci.2021.02.044. PMID: 33771552
Materna-Kiryluk A, Pollak A, Gawalski K, Szczawinska-Poplonyk A, Rydzynska Z, Sosnowska A, Cukrowska B, Gasperowicz P, Konopka E, Pietrucha B, Grzywa TM, Banaszak-Ziemska M, Niedziela M, Skalska-Sadowska J, Stawiński P, Śladowski D, Nowis D, Ploski R
Hum Mol Genet 2021 Apr 26;30(3-4):226-233. doi: 10.1093/hmg/ddab035. PMID: 33517393
Artilheiro V, Portela F, Reis AT
J Appl Genet 2020 Dec;61(4):571-573. Epub 2020 Sep 10 doi: 10.1007/s13353-020-00581-5. PMID: 32910413
Injarie AM, Narang A, Idrees Z, Saggar AK, Nischal KK
Cornea 2012 Mar;31(3):269-72. doi: 10.1097/ICO.0b013e3182182089. PMID: 22316651
Crisponi L, Crisponi G, Meloni A, Toliat MR, Nurnberg G, Usala G, Uda M, Masala M, Hohne W, Becker C, Marongiu M, Chiappe F, Kleta R, Rauch A, Wollnik B, Strasser F, Reese T, Jakobs C, Kurlemann G, Cao A, Nurnberg P, Rutsch F
Am J Hum Genet 2007 May;80(5):971-81. Epub 2007 Mar 30 doi: 10.1086/516843. PMID: 17436252Free PMC Article

Prognosis

Chen YH, van Zon S, Adams A, Schmidt-Arras D, Laurence ADJ, Uhlig HH
J Clin Immunol 2023 Dec 22;44(1):30. doi: 10.1007/s10875-023-01603-7. PMID: 38133879Free PMC Article
Warnier H, Barrea C, Bethlen S, Schrouff I, Harvengt J
Orphanet J Rare Dis 2022 Apr 23;17(1):174. doi: 10.1186/s13023-022-02323-8. PMID: 35461249Free PMC Article
Siccha SM, Cueto AM, Parrón-Pajares M, González-Morán G, Pacio-Miguez M, Del Pozo Á, Solís M, Rodriguez-Jimenez C, Caino S, Fano V, Heath KE, García-Miñaúr S, Palomares-Bralo M, Santos-Simarro F
Am J Med Genet A 2021 Mar;185(3):856-865. Epub 2020 Dec 11 doi: 10.1002/ajmg.a.62010. PMID: 33305909
Yeşil G, Lebre AS, Santos SD, Güran O, Özahi II, Daire VC, Güran T
Am J Med Genet A 2014 Sep;164A(9):2200-5. Epub 2014 Jul 2 doi: 10.1002/ajmg.a.36626. PMID: 24988918
Mikelonis D, Jorcyk CL, Tawara K, Oxford JT
Orphanet J Rare Dis 2014 Mar 12;9:34. doi: 10.1186/1750-1172-9-34. PMID: 24618404Free PMC Article

Clinical prediction guides

Chen YH, van Zon S, Adams A, Schmidt-Arras D, Laurence ADJ, Uhlig HH
J Clin Immunol 2023 Dec 22;44(1):30. doi: 10.1007/s10875-023-01603-7. PMID: 38133879Free PMC Article
Siccha SM, Cueto AM, Parrón-Pajares M, González-Morán G, Pacio-Miguez M, Del Pozo Á, Solís M, Rodriguez-Jimenez C, Caino S, Fano V, Heath KE, García-Miñaúr S, Palomares-Bralo M, Santos-Simarro F
Am J Med Genet A 2021 Mar;185(3):856-865. Epub 2020 Dec 11 doi: 10.1002/ajmg.a.62010. PMID: 33305909
Corona-Rivera JR, Cormier-Daire V, Dagoneau N, Coello-Ramírez P, López-Marure E, Romo-Huerta CO, Silva-Baez H, Aguirre-Salas LM, Estrada-Solorio MI
Eur J Med Genet 2009 Jul-Aug;52(4):242-6. Epub 2009 Apr 14 doi: 10.1016/j.ejmg.2009.04.001. PMID: 19371797
Dagoneau N, Scheffer D, Huber C, Al-Gazali LI, Di Rocco M, Godard A, Martinovic J, Raas-Rothschild A, Sigaudy S, Unger S, Nicole S, Fontaine B, Taupin JL, Moreau JF, Superti-Furga A, Le Merrer M, Bonaventure J, Munnich A, Legeai-Mallet L, Cormier-Daire V
Am J Hum Genet 2004 Feb;74(2):298-305. Epub 2004 Jan 21 doi: 10.1086/381715. PMID: 14740318Free PMC Article
Raas-Rothschild A, Ergaz-Schaltiel Z, Bar-Ziv J, Rein AJ
Am J Med Genet A 2003 Aug 30;121A(2):156-8. doi: 10.1002/ajmg.a.20066. PMID: 12910496

Recent systematic reviews

Warnier H, Barrea C, Bethlen S, Schrouff I, Harvengt J
Orphanet J Rare Dis 2022 Apr 23;17(1):174. doi: 10.1186/s13023-022-02323-8. PMID: 35461249Free PMC Article

Supplemental Content

Table of contents

    Clinical resources

    Practice guidelines

    • PubMed
      See practice and clinical guidelines in PubMed. The search results may include broader topics and may not capture all published guidelines. See the FAQ for details.

    Recent activity

    Your browsing activity is empty.

    Activity recording is turned off.

    Turn recording back on

    See more...